1. bookVolume 9 (2022): Issue 1 (January 2022)
Journal Details
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Format
Journal
eISSN
2055-3390
First Published
22 Dec 2017
Publication timeframe
1 time per year
Languages
English
access type Open Access

Discrete choice experiments: An overview of experience to date in haemophilia

Published Online: 20 May 2022
Volume & Issue: Volume 9 (2022) - Issue 1 (January 2022)
Page range: 50 - 63
Journal Details
License
Format
Journal
eISSN
2055-3390
First Published
22 Dec 2017
Publication timeframe
1 time per year
Languages
English
Abstract Background

The patient voice is an important consideration in the availability and choice of pharmaceuticals – however, how to capture this complex area and apply it formally within regulation, health technology assessment and reimbursement remains subject to ongoing debate. Patient preference studies such as discrete choice experiments (DCEs) are being utilised more frequently in healthcare and it is anticipated that patient preference data will be incorporated more frequently into regulatory submissions moving forward.

Aim

The aim of this review is to provide an overview of DCEs conducted within haemophilia to date and to consider the key issues in response to a rapidly evolving therapeutic pathway.

Methods

A systematic literature search was undertaken via Ovid MEDLINE and EMBASE CLASSIC + EMBASE. Abstracts were uploaded and analysed via Rayyan systematic review software. Results: Of 478 records identified from the database searches, 12 full text journal articles met the inclusion criteria with a date range from 2005–2021. There have been two published studies exploring haemophilia patient preferences in relation to gene therapy: one DCE and one utilising a threshold technique. Surveyed audiences included physicians, patients, pharmacists, healthcare professionals and caregivers. 50% of the included studies (n=6) were exclusively conducted in the US, whilst 3 recruited participants across multiple countries. The sample size varied considerably between studies with the total sample size ranging from 30 participants to 505 participants. For the studies involving patients and their caregivers, the mean patient age range was 8.2–41.4 years. There was diversity in (a) the scale of the qualitative work undertaken to support the DCEs, (b) the undertaking of pilots, and (c) how extensively these elements were reported in the included studies. There is a notable trend towards using an online web-based format, with 3 out of 4 DCEs since 2019 utilising this approach. The number of attributes observed per DCE ranged from 5–12 with a median of 6 attributes from the included studies. The number of levels per attribute was relatively consistent (range 2–5) with 2–3 (n=4) and 2–4 levels (n=4) being utilised most frequently.

Conclusion

Patient preferences and the methods for capturing these are likely to be subject to ongoing debate as the haemophilia care pathway evolves to offer more therapeutic options with a range of risks and benefits. Whilst techniques such as DCE are effective at quantifying patient preferences, they tell us little about the reasons driving these decisions and the likelihood that they will change in response to temporal or external factors. DCEs could be particularly useful for estimating the uptake of new products and assessing potential budget impact. Accelerated and reformed regulatory processes are likely to increase demand for patient preference studies. There is therefore an increased requirement to ensure that patient advocacy groups (PAGs) are resourced and have the expertise to support these studies alongside other research commitments, and that manufacturers consider collaborative approaches when formally capturing patient preferences.

As more therapeutic options become available in haemophilia care, discrete choice experiment may be a useful means of gauging patient preference

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Keywords

<p>The patient voice is an important consideration in the availability and choice of pharmaceuticals – however, how to capture this complex area and apply it formally within regulation, health technology assessment and reimbursement remains subject to ongoing debate <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_001">1</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_002">2</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_003">3</a>]</sup>.</p> <p>The 21st Century Cures Act in the USA highlights the importance of considering the patient experience during the drug development process <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_004">4</a>]</sup>. The Act facilitates the submission of patient experience information and ‘real world evidence’ to enable more rapid drug and device approval <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_005">5</a>]</sup>. However, there remains ambiguity surrounding what constitutes real evidence and concerns that overreliance on this data may potentially mislead clinicians and expose patients to unsafe/ineffective treatments <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_005">5</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_006">6</a>]</sup>. Despite these evidential challenges, it is likely that a rise in patient preference data being incorporated in regulatory submissions will be observed moving forward <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_007">7</a>]</sup>.</p> <p>Pharmaceutical regulators such as the Food and Drug Administration (FDA), European Medicines Agency (EMA), and Medicines & Healthcare products Regulatory Agency (MHRA) are accelerating the availability of medicines which target an unmet need <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_008">8</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_009">9</a>]</sup>. Recent examples of this include Project Orbis <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_010">10</a>]</sup> and the Innovative Licensing and Access Pathway (ILAP) <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_011">11</a>]</sup>. As health technology assessment (HTA) evolves to meet this scenario <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_012">12</a>]</sup>, accelerated access pathways can also exacerbate the challenge of how to capture patient preferences within HTA and the role patient advocacy groups (PAGs) play within this process. <a ref-type="fig" href="#j_jhp-2022-0006_fig_001">Figure 1</a> highlights the role played by patients and PAGs in product availability and choice across the lifecycle of a pharmaceutical product. As accelerated initiatives and revised regulatory frameworks will require the formal capture of patient input at an earlier stage, PAGs will have increasing opportunities to campaign and provide support over a wider timeframe.</p> <figure id="j_jhp-2022-0006_fig_001" position="float" fig-type="figure"><h2>Figure 1</h2><figCaption><p>Impact of regulatory developments on capturing patient preferences across the product lifecycle</p></figCaption><img xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="graphic/j_jhp-2022-0006_fig_001.jpg" src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_001.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=8aeedcd05980675b18c6ed3725351c069d257ee7eb6b15bddae28d03ac225851" class="mw-100"></img></figure> <sec id="j_jhp-2022-0006_s_001_s_001"><div>Patient preference studies</div> <p>Patient preference studies can be either qualitative or quantitative and seek to capture the desirability of particular characteristics which are associated with a product in a given healthcare scenario <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_003">3</a>]</sup>. The Medical Device Innovation Consortium (MDIC) have developed a framework for incorporating information on patient preferences regarding benefit and risk into regulatory assessments of new medical technologies <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_013">13</a>]</sup>. There is no algorithmic approach to determine which patient preference method to use; method selection is a complex issue which depends on the research question being addressed, the population being studied, and time/budgetary constraints <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_014">14</a>]</sup>. The MDIC report helpfully provides a catalogue of patient preference methods and groups them by the type of information provided, namely, structured weighting, health-state utility, stated preference and revealed preference <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_013">13</a>]</sup>.</p> </sec> <sec id="j_jhp-2022-0006_s_001_s_002"><div>Discrete choice experiments</div> <p>Discrete choice experiments (DCEs) are a stated preference technique which allows researchers to uncover how individuals value selected attributes of a programme, product or service by asking them to state their choice over different hypothetical alternatives <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_015">15</a>]</sup>. Stated preference techniques such as DCEs utilise hypothetical examples, typically in the form of a questionnaire, and rely on respondents making choices based on these; revealed preferences analyse patient choices and behaviours in the real world, with examples including patient preference trials or direct questions within clinical trials <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_013">13</a>]</sup>.</p> <p>A DCE is a quantitative technique for eliciting individual preferences. It is grounded in random utility theory and relies on the assumptions of economic rationality and utility maximisation <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_015">15</a>]</sup>. This means there is a core underlying assumption that participants can rationally select the choice which gives them the most benefit. The outputs from DCEs show the strength of relative preferences of the characteristics under evaluation and the rate at which they are traded off. The results are often expressed in terms of utilities or marginal rates of substitution. For example, a DCE could investigate the strength of preference for a treatment that is considered more effective than an existing treatment but requires more frequent administration, and the balance between the two that is considered optimal by recipients.</p> <p>Establishing the attributes and their associated levels is one of the most important and challenging steps in designing a DCE. Attributes represent a technology's key characteristic (e.g. frequency of infusion) whereas levels refer to potential options for a specific attribute (e.g. daily, once a week, once a month). The underlying validity of the study rests on specifying these correctly. Once the attributes are established, the associated levels need to be assigned. These should reflect the range of situations that respondents might be likely to experience <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_015">15</a>]</sup>. Once the attribute levels are established the next step is to generate a set of hypothetical choice sets <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_015">15</a>]</sup>. An example from a DCE in haemophilia, with the attributes and levels highlighted, is given in <a ref-type="fig" href="#j_jhp-2022-0006_fig_002">Figure 2</a> <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_016">16</a>]</sup>.</p> <figure id="j_jhp-2022-0006_fig_002" position="float" fig-type="figure"><h2>Figure 2</h2><figCaption><p>Example of a DCE in haemophilia with attributes and levels highlighted <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_016">16</a>]</sup></p></figCaption><img xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="graphic/j_jhp-2022-0006_fig_002.jpg" src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_002.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=4f306027e74c33449db3d4f6f7618371bfa687d15351d27834073df2d286a9c8" class="mw-100"></img></figure> <p>DCEs are being utilised widely in healthcare and health economics and the method is being used with increasing sophistication in design alongside enhanced analytical techniques which are contributing to a higher quality of output <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_017">17</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_018">18</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_019">19</a>]</sup>.</p> </sec> <sec id="j_jhp-2022-0006_s_001_s_003"><div>DCEs in an evolving haemophilia pathway</div> <p>Treatment with haemophilia in the mainstream consists of replacement therapy with coagulation factor VIII or factor IX, with a complication being the development of inhibitory antibodies against the infused factor VIII or IX <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_020">20</a>]</sup>. The journey to the safe and routine infusion of factor VIII and IX therapies has been a challenging one and the freeze-dried powdered concentrates emerging in the 1970s were found to be contaminated by the transmission of HIV and hepatitis C in blood products <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_021">21</a>]</sup>. As a result of this tragic phenomenon, tighter screening methods were implemented and recombinant (non-plasma derived) technologies were developed <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_021">21</a>]</sup>. Treatments have continued to evolve in haemophilia with bypassing agents, long-acting coagulator factors, biological therapies and the emergence of gene therapy. Horizon scanning from the Specialist Pharmacy Service (SPS) <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_022">22</a>]</sup> has recently identified a number of forthcoming technologies in haemophilia (<a ref-type="table" href="#j_jhp-2022-0006_tab_001">Table 1</a>).</p> <table-wrap id="j_jhp-2022-0006_tab_001" position="float"><label>Table 1</label><caption><p>Potential future treatments in haemophilia<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_001"><sup>*</sup></a></p></caption> <table frame="hsides" rules="all"> <thead> <tr> <th align="left" valign="bottom"><bold>PRODUCT NAME</bold></th> <th align="left" valign="bottom"><bold>THERAPEUTIC FOCUS</bold></th> <th align="left" valign="bottom"><bold>PRODUCT TYPE</bold></th> <th align="left" valign="bottom"><bold>PHASE OF CLINICAL DEVELOPMENT</bold></th> </tr> </thead> <tbody> <tr> <td align="left" valign="top">Serpin PC</td> <td align="left" valign="top">Haemophilia A and Haemophilia B</td> <td align="left" valign="top">Specific inhibitor of activated protein C (APC)</td> <td align="left" valign="top">Phase II</td> </tr> <tr> <td align="left" valign="top">Mim8</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">Next generation FVIII mimetic antibody</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Etranacogene dezaparvovec</td> <td align="left" valign="top">Haemophilia B</td> <td align="left" valign="top">AAV5 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Fidanacogene elaparvovec</td> <td align="left" valign="top">Haemophilia B</td> <td align="left" valign="top">AAV8 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Valoctocogene roxaparvovec</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">AAV5 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Concizumab</td> <td align="left" valign="top">Haemophilia A and Haemophilia B</td> <td align="left" valign="top">Monoclonal antibody directed against tissue factor pathway inhibitor (TFPI)</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Fitusiran</td> <td align="left" valign="top">Haemophilia A and Haemophilia B</td> <td align="left" valign="top">RNAi therapeutic targeting antithrombin</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Efanesoctocog alfa</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">Fully recombinant factor VIII therapy independent of von Willebrand factor</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Eptacog beta activated</td> <td align="left" valign="top">Haemophilia A and Haemophilia B (in patients with inhibitory antibodies to factor VIII or IX)</td> <td align="left" valign="top">Transgenically produced recombinant human factor VIIa</td> <td align="left" valign="top">Phase III<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_002"><sup>**</sup></a></td> </tr> <tr> <td align="left" valign="top">Giroctocogene fitelparvovec</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">AAV2/6 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Dirloctocogene samoparvovec</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">AAV-LK03 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> </tbody> </table> <table-wrap-foot> <fn-group> <fn id="j_jhp-2022-0006_table-fn_001"><label>*</label><p>Horizon scanning data as of April 2022</p></fn> <fn id="j_jhp-2022-0006_table-fn_002"><label>**</label><p>Marketed in the US</p></fn> </fn-group> </table-wrap-foot> </table-wrap> <p>As care pathways such as those in haemophilia become increasingly dynamic, there is a key challenge that patient preferences for advanced therapy medicinal products (ATMPs) in relation to current or future therapeutic options are unknown <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_012">12</a>]</sup>. Understanding this complex interplay between therapeutic interventions will therefore be vital to inform future healthcare investment decisions. Understanding patient preferences and the impact on uptake of current and future interventions is regarded as a core challenge in budget impact analysis (BIA) and there is an acknowledgement that little data may exist to support assumptions which might be highly sensitive in the assessment <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_023">23</a>]</sup>. ISPOR guidelines recommend that the mix of interventions over time should be based on past changes, market research, or clinical expert opinion <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_023">23</a>]</sup>. Whilst sensitivity analysis and a commitment to transparency can assist with the interpretation of BIAs <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_023">23</a>]</sup>, models which rely on historical data, observed uptake of comparable interventions, or expert opinion, fail to account for the dynamic and heterogeneous manner in which individuals make decisions <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_024">24</a>]</sup>. DCEs could be particularly useful for predicting the uptake of new products where observational data from trials or pilot projects are not available <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_025">25</a>]</sup>, and can potentially improve models that parameterise uptake solely based on expert opinion <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_024">24</a>]</sup>.</p> </sec> </sec> <sec id="j_jhp-2022-0006_s_002"><div>METHODS</div> <p>A systematic literature search was undertaken via Ovid MEDLINE and EMBASE CLASSIC + EMBASE with the full search terms set out in <a ref-type="fig" href="#j_jhp-2022-0006_fig_003">Figure 3</a>. Abstracts were uploaded and analysed via Rayyan systematic review software <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_026">26</a>]</sup>.</p> <figure id="j_jhp-2022-0006_fig_003" position="float" fig-type="figure"><h2>Figure 3</h2><figCaption><p>Search terms for literature search on DCEs in haemophilia care</p></figCaption><img xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="graphic/j_jhp-2022-0006_fig_003.jpg" src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_003.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=ceebd12e9ce6351080b3c40d09b0fcb5803a3838ab1bc7a1e3dfa74415f717e7" class="mw-100"></img></figure> <p>Articles were included if they concerned a DCE in haemophilia care, if they were a full article, and if they had a pharmacological focus. Articles were excluded for being background information, non-haemophilia specific, biological/other or non-English language.</p> <p>DCEs in relevant articles were analysed in respect of the audience surveyed, sample size, geography, mean age of patients, format (e.g. paper or online), approach (including extent to which qualitative work supported the DCE, piloting and how these elements were reported), and the attributes and levels per attribute included in the DCE. The study topic and funding source were also documented. Product attributes were mapped over time to investigate temporal patterns.</p> </sec> <sec id="j_jhp-2022-0006_s_003"><div>RESULTS</div> <p>As set out in the PRISMA diagram in <a ref-type="fig" href="#j_jhp-2022-0006_fig_004">Figure 4</a>, 478 records were identified with 320 records available once duplicates had been removed. A further 200 articles were excluded for being background information (n=146), non-haemophilia specific (n=26), biological/other (n=21) or non-English language (n=7). Of the 120 full-text articles assessed for eligibility, 108 were deemed ineligible due to not being a DCE (n=89), not being a full-journal article (n=18) or not having a pharmacological focus (n=1). This led to 12 full text articles being included, with a date range from 2005–2021 <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_016">16</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_027">27</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_028">28</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_029">29</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_030">30</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_031">31</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_032">32</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_033">33</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_034">34</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_035">35</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_036">36</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_037">37</a>]</sup>. The results are summarised in <a ref-type="table" href="#j_jhp-2022-0006_tab_002">Table 2</a>. This table has been cross-referenced to update previous work conducted on this topic <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_038">38</a>]</sup>. The review also identified 3 literature reviews <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_039">39</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_040">40</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_041">41</a>]</sup> assessing patient preferences in haemophilia, all of which were not full journal articles. There have been two published studies exploring haemophilia patient preferences in relation to gene therapy: one DCE <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_037">37</a>]</sup> and one utilising a threshold technique <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_042">42</a>]</sup>.</p> <figure id="j_jhp-2022-0006_fig_004" position="float" fig-type="figure"><h2>Figure 4</h2><figCaption><p>PRISMA diagram for literature search on DCEs in haemophilia care</p></figCaption><img xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="graphic/j_jhp-2022-0006_fig_004.jpg" src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_004.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=ebd03085a1e2b21d6ec2e1e489b2378f9b2470b5ff343da3b72f2aab35c9f6b6" class="mw-100"></img></figure> <table-wrap id="j_jhp-2022-0006_tab_002" position="float"><label>Table 2</label><caption><p>Overview of DCEs within haemophilia</p></caption> <table frame="hsides" rules="all"> <thead> <tr> <th align="left" valign="bottom"><bold>AUTHOR</bold></th> <th align="left" valign="bottom"><bold>YEAR</bold></th> <th align="left" valign="bottom"><bold>SURVEY AUDIENCE</bold></th> <th align="left" valign="bottom"><bold>REGION</bold></th> <th align="left" valign="bottom"><bold>STUDY TOPIC</bold></th> <th align="left" valign="bottom"><bold>SAMPLE SIZE (N)</bold></th> <th align="left" valign="bottom"><bold>MEAN PATIENT AGE (YEARS)</bold></th> <th align="left" valign="bottom"><bold>SCALE OF QUALITATIVE WORK</bold></th> <th align="left" valign="bottom"><bold>NO. OF ATTRIBUTES</bold></th> <th align="left" valign="bottom"><bold>LEVELS PER ATTRIBUTE</bold></th> <th align="left" valign="bottom"><bold>INDUSTRY FUNDING</bold></th> <th align="left" valign="bottom"><bold>SURVEY METHODS</bold></th> </tr> </thead> <tbody> <tr> <td align="left" valign="top">Mantovani et al.<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_027">27</a>]</sup></td> <td align="left" valign="top">2005</td> <td align="left" valign="top">Physicians, patients and pharmacists</td> <td align="left" valign="top">Italy</td> <td align="left" valign="top">Treatment products in haemophilia</td> <td align="left" valign="top">Total: 305<break/>Physicians: 69<break/>Patients: 178<break/>Pharmacists: 58</td> <td align="left" valign="top">35.9</td> <td align="left" valign="top">Focus group (n=N/R) + Pilot (n=15)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Bayer Italia S.p.A.</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Lee et al. <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_028">28</a>]</sup></td> <td align="left" valign="top">2008</td> <td align="left" valign="top">Physicians</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Coagulation factor concentrates<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_003"><sup>†</sup></a></td> <td align="left" valign="top">Total: 30</td> <td align="left" valign="top">N/A</td> <td align="left" valign="top">Physician engagement (n & format N/R) + testing (N/R)</td> <td align="left" valign="top">12</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Novo Nordisk Inc</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Scalone et al.<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_029">29</a>]</sup></td> <td align="left" valign="top">2009</td> <td align="left" valign="top">Physicians, patients and pharmacists</td> <td align="left" valign="top">Italy</td> <td align="left" valign="top">Coagulation factor concentrates<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_003"><sup>†</sup></a></td> <td align="left" valign="top">Total: 101<break/>Physicians: 39<break/>Patients: 37<break/>Pharmacists: 25</td> <td align="left" valign="top">Adults = 41.4<break/>Paedatric = 8.2</td> <td align="left" valign="top">Focus group (n=N/R) + Pilot (n=35)</td> <td align="left" valign="top">8</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Novo Nordisk Denmark</td> <td align="left" valign="top">N/R</td> </tr> <tr> <td align="left" valign="top">Brown et al.<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_016">16</a>]</sup></td> <td align="left" valign="top">2011</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Treatment products in haemophilia<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_003"><sup>†</sup></a></td> <td align="left" valign="top">Total: 53<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_004"><sup>*</sup></a><break/>Patients: 23<break/>Caregivers: 30</td> <td align="left" valign="top">20.7</td> <td align="left" valign="top">Used same as Lee et al. (2008)<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_028">28</a>]</sup></td> <td align="left" valign="top">12</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Novo Nordisk Inc</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Mohamed et al.<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_030">30</a>]</sup></td> <td align="left" valign="top">2011</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 147<break/>Patients: 77<break/>Caregivers: 70</td> <td align="left" valign="top">Adults = 38.7<break/>Juvenile = 12.4</td> <td align="left" valign="top">Face-to-face interviews with adult patients and caregivers (n=8)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Baxter Biosciences</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Gelhorn et al.<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_031">31</a>]</sup></td> <td align="left" valign="top">2013</td> <td align="left" valign="top">Physicians</td> <td align="left" valign="top">US and EU</td> <td align="left" valign="top">Treatment products in haemophilia A<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_003"><sup>†</sup></a></td> <td align="left" valign="top">Total: 36<break/>Physicians EU: 17<break/>Physicians US: 19</td> <td align="left" valign="top">N/A</td> <td align="left" valign="top">Qualitative interviews (n=4) + Pilot (n=5)</td> <td align="left" valign="top">5</td> <td align="left" valign="top">3</td> <td align="left" valign="top">Baxter Biosciences</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Chaugule et al.<sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_032">32</a>]</sup></td> <td align="left" valign="top">2015</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Willingness to pay for treatment products in haemophilia</td> <td align="left" valign="top">Total: 79</td> <td align="left" valign="top">40.0</td> <td align="left" valign="top">Assistance from Haem author + Pilot (n=5)</td> <td align="left" valign="top">5</td> <td align="left" valign="top">2–5</td> <td align="left" valign="top">None</td> <td align="left" valign="top">iPad at conference</td> </tr> <tr> <td align="left" valign="top">Lock et al. <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_033">33</a>]</sup></td> <td align="left" valign="top">2016</td> <td align="left" valign="top">Patients, caregivers and HCPs</td> <td align="left" valign="top">Multi-country</td> <td align="left" valign="top">PK-guided dosing of prophylaxis</td> <td align="left" valign="top">Total: 224<break/>HCPs: 91<break/>Patients: 114<break/>Caregivers: 19</td> <td align="left" valign="top">38.0</td> <td align="left" valign="top">Qualitative interviews + Pilot (n=10)</td> <td align="left" valign="top">5</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Pfizer</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Fifer et al. <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_034">34</a>]</sup></td> <td align="left" valign="top">2019</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">Multi-country</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 54<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_007"><sup>^</sup></a><break/>Patients: 24<break/>Caregivers: 30</td> <td align="left" valign="top">N/R</td> <td align="left" valign="top">Qualitative interviews (n=10)</td> <td align="left" valign="top">11</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Roche</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Su et al. <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_035">35</a>]</sup></td> <td align="left" valign="top">2020</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 209<break/>Patients: 113<break/>Caregivers: 96</td> <td align="left" valign="top">35.5</td> <td align="left" valign="top">Qualitative interviews (n=10)<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_005"><sup>**</sup></a> + Pilot (n=6)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Sanofi Genzyme</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Park et al. <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_036">36</a>]</sup></td> <td align="left" valign="top">2021</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">South Korea</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 505<a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_006"><sup>***</sup></a></td> <td align="left" valign="top">31.5</td> <td align="left" valign="top">N/R</td> <td align="left" valign="top">5</td> <td align="left" valign="top">3</td> <td align="left" valign="top">Pfizer Pharmaceuticals Korea Ltd</td> <td align="left" valign="top">Hospital-based survey</td> </tr> <tr> <td align="left" valign="top">Witkop et al. <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_037">37</a>]</sup></td> <td align="left" valign="top">2021</td> <td align="left" valign="top">Patients</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Haemophilia gene therapy</td> <td align="left" valign="top">Total: 183</td> <td align="left" valign="top">38.5</td> <td align="left" valign="top">Qualitative interviews (n=7) <a ref-type="table-fn" href="#j_jhp-2022-0006_table-fn_005"><sup>**</sup></a> + Pilot (n=14)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">3–4</td> <td align="left" valign="top">uniQure Inc.</td> <td align="left" valign="top">Web-based survey</td> </tr> </tbody> </table> <table-wrap-foot> <fn-group> <fn><p>HCPs: Healthcare professionals</p></fn> <fn><p>N/A: Not applicable</p></fn> <fn><p>N/R: Not reported</p></fn> <fn><p>PK: Pharmacokinetic</p></fn> <fn id="j_jhp-2022-0006_table-fn_003"><label>†</label><p>Inhibitor-specific study</p></fn> <fn id="j_jhp-2022-0006_table-fn_004"><label>*</label><p>The sample size per question varied from 51 to 53, as one or two patients did not respond to certain questions</p></fn> <fn id="j_jhp-2022-0006_table-fn_005"><label>**</label><p>Noting more extensive work was carried out but not fully reported</p></fn> <fn id="j_jhp-2022-0006_table-fn_006"><label>***</label><p>Not possible to obtain number breakdown of caregivers or patients</p></fn> <fn id="j_jhp-2022-0006_table-fn_007"><label>^</label><p>Three respondents were removed as they reported they could not understand the experiment – but not reported if caregiver or patient</p></fn> </fn-group> </table-wrap-foot> </table-wrap> <p>Surveyed audiences included physicians, patients, pharmacists, healthcare professionals and caregivers (either alone or in combination). Two studies focused exclusively on physicians, whilst 50% of the included studies (n=6) surveyed patients and their caregivers. Half of the studies (n=6) were conducted in the US, whilst 3 studies recruited participants across multiple countries. Eleven (92%) studies reported funding by manufacturers with a commercial interest in haemophilia. The sample size between studies varied considerably, with the total sample size ranging from 30 participants to 505 participants. For studies involving patients and their caregivers, the mean patient age range was 8.2–41.4 years; removing the juvenile/paediatric patient population figures narrows this range to 20.7–40.0 years. There was diversity in (a) the scale of the qualitative work undertaken to support the DCEs, (b) the undertaking of pilots, and (c) how extensively this was reported in the included studies. One study did not report undertaking qualitative work <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_036">36</a>]</sup> and in a number of studies the extent of qualitative work or piloting was difficult to establish. It has been previously reported that inadequate information about methodological detail is hindering assessment of quality <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_018">18</a>]</sup>. Given the crucial role that qualitative work and piloting plays in establishing the validity of the DCE <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_015">15</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_017">17</a>]</sup>, it is vital that future DCEs report the full details of qualitative preparatory work and piloting. Three main methods were employed: web-based, paper-based and in-person survey methods. Whilst paper-based surveys made up the earliest DCEs in this area, there is a clear trend to switching to an online web-based approach, with 3 out of 4 DCEs since 2019 utilising this format. The number of attributes observed per DCE ranged from 5–12, with a median of 6 attributes from the included studies. The levels per attribute was relatively consistent (range 2–5) with 2–3 (n=4) and 2–4 levels (n=4) being utilised most frequently. The development of recombinant products over plasma products was seen as a major therapeutic advance in haemophilia; as shown in <a ref-type="fig" href="#j_jhp-2022-0006_fig_005">Figure 5</a>, a shift of attribute focus from safety towards reduction of bleeding risk has been observed in more recent DCE studies <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_038">38</a>]</sup>.</p> <figure id="j_jhp-2022-0006_fig_005" position="float" fig-type="figure"><h2>Figure 5</h2><figCaption><p>Product attributes mapped over time</p></figCaption><img xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="graphic/j_jhp-2022-0006_fig_005.jpg" src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_005.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=f69a52dedfdf9f8788faeb89b44866ed259dfddcdf5ba3c5207e7e98aedd9f39" class="mw-100"></img></figure> <sec id="j_jhp-2022-0006_s_003_s_001"><div>Head-to-head comparison of gene therapy studies</div> <p>As shown in <a ref-type="table" href="#j_jhp-2022-0006_tab_001">Table 1</a>, a number of gene therapies are being developed for both haemophilia A and B. Two published studies to date have explored haemophilia patient preferences in relation to gene therapy: one DCE <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_037">37</a>]</sup> and one utilising a threshold technique <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_042">42</a>]</sup>. Whilst DCEs have been explored extensively in this paper, the threshold technique is a method that determines the maximal change in one attribute respondents are willing to accept to achieve a given change in another attribute <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_043">43</a>]</sup>. Van Overbeeke and colleagues ruled out the utilisation of DCE methodology in the development of the study protocol as they estimated that it would be challenging to recruit over 100 participants, which are generally required for DCEs <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_044">44</a>]</sup>. It is widely acknowledged that method selection is a complex issue <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_014">14</a>]</sup> and that both methods are stated preference studies that can be utilised to quantify patient preferences <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_013">13</a>]</sup>. <a ref-type="fig" href="#j_jhp-2022-0006_fig_006">Figure 6</a> sets out a comparison of the two studies: sample sizes for both were within the range observed with other DCEs to date and, particularly impressive, both studies had to deal with disruption associated with Covid-19. Both studies reported literature reviews, qualitative interviews and piloting to develop and test study attributes and levels. Patients with both haemophilia A and B were included across the studies; sampling in the threshold technique study was aligned to the Belgian haemophilia patient population, whilst over a third of the sampled population in the DCE were haemophilia B patients. Sample severity differed considerably between the studies. The DCE study provided a relatively even split between moderate and severe patients, whilst the threshold technique study had sampling which heavily favoured severe patients. Attributes were consistent between studies with annual bleed rate (ABR), dose frequency, safety issues and quality of life (QoL) being examined, following general trends observed in this area <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_038">38</a>]</sup>. The DCE also explored the impact on mental health and post-treatment effects. It has been shown previously that training materials result in more choice consistency and facilitate more complex designs <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_045">45</a>]</sup> and the threshold technique study included an educational tool to assist with understanding of gene therapy. The educational tool was shown to have a significant impact on both the ABR and QoL threshold. The threshold technique study excluded non-factor therapies such as emicizumab, despite 15% (n=17) of the sample being treated with the product.</p> <figure id="j_jhp-2022-0006_fig_006" position="float" fig-type="figure"><h2>Figure 6</h2><figCaption><p>Comparison of haemophilia gene therapy patient preference studies</p></figCaption><img xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="graphic/j_jhp-2022-0006_fig_006.jpg" src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_006.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=138a26df76c3fe0185914e844e14f97ce223707540232da81804efdebd63e529" class="mw-100"></img></figure> </sec> </sec> <sec id="j_jhp-2022-0006_s_004"><div>DISCUSSION</div> <p>The haemophilia care pathway is constantly evolving, and this phenomenon must be considered carefully when undertaking a DCE to establish patient preferences for product attributes and levels. One of the included studies in the review encountered this issue directly: they were only able to include patients with FVIII administered intravenously, as the subcutaneous emicizumab had not been launched in Korea at the time of patient enrolment <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_036">36</a>]</sup>. Whilst horizon scanning information is dynamic, and may not be available to all researchers, qualitative engagement with clinicians to future proof study design against pathway changes should be considered as part of the experimental design.</p> <p>The sample sizes in the included studies varied considerably and recruitment challenges should be actively considered when choosing the study design to capture stakeholder preferences. Methods such as the threshold technique may be more appropriate than a DCE if recruitment to the study will be challenging or the target population is small <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_044">44</a>]</sup>. One study in the review had a recruitment period of 21 months <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_034">34</a>]</sup>; as acknowledged in the paper, this length of recruitment may also lead to potential bias.</p> <p>A recent review highlighted the vital requirement to undertake qualitative research to determine attributes and levels within DCEs, but echoed concerns that inadequate information about methodological detail was hindering assessment of quality <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_018">18</a>]</sup>. Training materials remain a key but under-developed component of DCEs and the development of interactive tools can potentially improve the quality of choice data if participants are better engaged <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_045">45</a>]</sup>.</p> <p>There is an increasing trend for DCEs to present attribute descriptions and content using online survey-based methods <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_045">45</a>]</sup> and videos <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_046">46</a>]</sup>. One study compared respondents’ understanding of attribute information based on text or video and found that although there was no systematic difference between video or text arms in the study, the information provided by video may better engage survey participants and improve their retention of content <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_046">46</a>]</sup>. Another study looked at animation training materials, and whilst providing these did not change the preferences of respondents, they did result in more choice consistency which may facilitate more complex experiment designs <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_045">45</a>]</sup>.</p> <p>A key challenge with DCEs is that they are cross-sectional surveys, which provide a snapshot at a single point of time. This approach has advantages including being inexpensive, simple to conduct and able to reach large audiences rapidly <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_047">47</a>]</sup>. However, a key limitation is that they are unable to investigate temporal effects <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_047">47</a>]</sup>. There are a range of temporal and external influences on patient choice including treatment history, family and friends, evolving life situation and clinical opinion, and these factors can change over time, yet the understanding of behavioural impacts on DCEs is currently limited <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_025">25</a>,<a ref-type="bibr" href="#j_jhp-2022-0006_ref_048">48</a>–<a ref-type="bibr" href="#j_jhp-2022-0006_ref_049">49</a>]</sup>. Consistency of attributes is likely to be a key issue when considering standardisation of outputs and the routine acceptance of DCE-derived patient data by HTA bodies. Key external events may also heavily influence preferences, for example, after the forthcoming UK report on the infected blood inquiry <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_050">50</a>]</sup> or a major side effect with an emerging technology, as seen with Covid-19 vaccinations <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_051">51</a>]</sup>.</p> <p>A key statistic from the review is that all but one of the studies was either directly or indirectly funded by pharmaceutical manufacturers with a commercial interest in haemophilia. If patient preference studies are going to become a formal part of regulatory and pricing and reimbursement submissions, then it is likely that manufacturers will be expected to fund these as part of an evidential package to place before the relevant authorities. Consideration needs to be given on how to standardise the study design/outputs and evaluate quality. The alternative would have to be that PAGs, or independent institutions would have to fill this gap, which may be financially challenging and/or practically unfeasible. Consideration should be given towards a collaborative approach to undertaking DCEs involving multiple pharmaceutical manufacturers to (a) optimise PAG resource and (b) avoid potential suggestions that attributes, and levels, are selected to support individual product benefits.</p> </sec> <sec id="j_jhp-2022-0006_s_005"><div>CONCLUSION</div> <p>Patient preferences and the methods for capturing these are likely to be subject to ongoing debate as the haemophilia care pathway evolves to offer more therapeutic options with a range of risks and benefits. In addition to challenges with sampling, bias, methodological choice/rigour and manufacturer influence, the question of how best to use the information from patient preference studies remains unclear. Whilst techniques such as DCEs are effective at quantifying patient preferences, they tell us very little about the reasons driving these decisions and the likelihood that they will change in response to temporal or external factors. Given the evolution of the care pathway in haemophilia and the emergence of gene therapy, DCEs could be particularly useful for estimating the uptake of new products and assessing potential budget impact <sup>[<a ref-type="bibr" href="#j_jhp-2022-0006_ref_025">25</a>]</sup>. Several DCE studies in the literature review reported here highlighted the challenge of recruiting participants in rare diseases such as haemophilia, and engaging with PAGs to assist with recruitment is therefore likely to be crucial. Accelerated and reformed regulatory processes are likely to increase demand for patient preference studies and therefore there is an increased requirement to ensure that PAGs are resourced and have the expertise to support these studies alongside other research commitments, and that manufacturers consider collaborative approaches when formally capturing patient preferences.</p> </sec> </div></div></div></div><div id="pane-4" class="SeriesTab_card__26XnC SeriesTab_tab-pane__3pc7y card tab-pane" role="tabpanel" aria-labelledby="tab-4"><div class="SeriesTab_card-header__1DTAS card-header d-md-none pl-0" role="tab" id="heading-4"><h4 class="mb-0"><a data-toggle="collapse" href="#collapse-4" data-parent="#content" aria-expanded="false" aria-controls="collapse-4" style="padding:24px 0">Figures & Tables<svg aria-hidden="true" focusable="false" data-prefix="fas" data-icon="chevron-down" class="svg-inline--fa fa-chevron-down fa-w-14 " role="img" xmlns="http://www.w3.org/2000/svg" viewBox="0 0 448 512"><path fill="currentColor" d="M207.029 381.476L12.686 187.132c-9.373-9.373-9.373-24.569 0-33.941l22.667-22.667c9.357-9.357 24.522-9.375 33.901-.04L224 284.505l154.745-154.021c9.379-9.335 24.544-9.317 33.901.04l22.667 22.667c9.373 9.373 9.373 24.569 0 33.941L240.971 381.476c-9.373 9.372-24.569 9.372-33.942 0z"></path></svg></a></h4></div><div id="collapse-4" class="SeriesTab_seriesTabCollapse__2csiF collapse" role="tabpanel" aria-labelledby="heading-4" data-parent="#content"><div class="SeriesTab_series-tab-body__1tZ1H SeriesTab_card-body__31JEh card-body Article_figures-tables__2SC5X"><figure><h4 class="mb-4"></h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_007.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=79af89e716693e30c6e318b59e340f745e3eed815e332204d05fde70d8f6438d" alt="As more therapeutic options become available in haemophilia care, discrete choice experiment may be a useful means of gauging patient preference© Shutterstock" class="mw-100"/><figcaption class="fw-500">As more therapeutic options become available in haemophilia care, discrete choice experiment may be a useful means of gauging patient preference© Shutterstock</figcaption></figure><figure><h4 class="mb-4">Figure 1</h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_001.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=8aeedcd05980675b18c6ed3725351c069d257ee7eb6b15bddae28d03ac225851" alt="Impact of regulatory developments on capturing patient preferences across the product lifecycle" class="mw-100"/><figcaption class="fw-500">Impact of regulatory developments on capturing patient preferences across the product lifecycle</figcaption></figure><figure><h4 class="mb-4">Figure 2</h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_002.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=4f306027e74c33449db3d4f6f7618371bfa687d15351d27834073df2d286a9c8" alt="Example of a DCE in haemophilia with attributes and levels highlighted [16]" class="mw-100"/><figcaption class="fw-500">Example of a DCE in haemophilia with attributes and levels highlighted [16]</figcaption></figure><figure><h4 class="mb-4">Figure 3</h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_003.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=ceebd12e9ce6351080b3c40d09b0fcb5803a3838ab1bc7a1e3dfa74415f717e7" alt="Search terms for literature search on DCEs in haemophilia care" class="mw-100"/><figcaption class="fw-500">Search terms for literature search on DCEs in haemophilia care</figcaption></figure><figure><h4 class="mb-4">Figure 4</h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_004.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=ebd03085a1e2b21d6ec2e1e489b2378f9b2470b5ff343da3b72f2aab35c9f6b6" alt="PRISMA diagram for literature search on DCEs in haemophilia care" class="mw-100"/><figcaption class="fw-500">PRISMA diagram for literature search on DCEs in haemophilia care</figcaption></figure><figure><h4 class="mb-4">Figure 5</h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_005.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=f69a52dedfdf9f8788faeb89b44866ed259dfddcdf5ba3c5207e7e98aedd9f39" alt="Product attributes mapped over time" class="mw-100"/><figcaption class="fw-500">Product attributes mapped over time</figcaption></figure><figure><h4 class="mb-4">Figure 6</h4><img src="https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_006.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256&X-Amz-Date=20220811T015651Z&X-Amz-SignedHeaders=host&X-Amz-Expires=18000&X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request&X-Amz-Signature=138a26df76c3fe0185914e844e14f97ce223707540232da81804efdebd63e529" alt="Comparison of haemophilia gene therapy patient preference studies" class="mw-100"/><figcaption class="fw-500">Comparison of haemophilia gene therapy patient preference studies</figcaption></figure><h4 class="mb-4 mt-4">Overview of DCEs within haemophilia</h4><table frame="hsides" rules="all"> <thead> <tr> <th align="left" valign="bottom"><bold>AUTHOR</bold></th> <th align="left" valign="bottom"><bold>YEAR</bold></th> <th align="left" valign="bottom"><bold>SURVEY AUDIENCE</bold></th> <th align="left" valign="bottom"><bold>REGION</bold></th> <th align="left" valign="bottom"><bold>STUDY TOPIC</bold></th> <th align="left" valign="bottom"><bold>SAMPLE SIZE (N)</bold></th> <th align="left" valign="bottom"><bold>MEAN PATIENT AGE (YEARS)</bold></th> <th align="left" valign="bottom"><bold>SCALE OF QUALITATIVE WORK</bold></th> <th align="left" valign="bottom"><bold>NO. OF ATTRIBUTES</bold></th> <th align="left" valign="bottom"><bold>LEVELS PER ATTRIBUTE</bold></th> <th align="left" valign="bottom"><bold>INDUSTRY FUNDING</bold></th> <th align="left" valign="bottom"><bold>SURVEY METHODS</bold></th> </tr> </thead> <tbody> <tr> <td align="left" valign="top">Mantovani et al.<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_027">27</xref>]</sup></td> <td align="left" valign="top">2005</td> <td align="left" valign="top">Physicians, patients and pharmacists</td> <td align="left" valign="top">Italy</td> <td align="left" valign="top">Treatment products in haemophilia</td> <td align="left" valign="top">Total: 305<break/>Physicians: 69<break/>Patients: 178<break/>Pharmacists: 58</td> <td align="left" valign="top">35.9</td> <td align="left" valign="top">Focus group (n=N/R) + Pilot (n=15)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Bayer Italia S.p.A.</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Lee et al. <sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_028">28</xref>]</sup></td> <td align="left" valign="top">2008</td> <td align="left" valign="top">Physicians</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Coagulation factor concentrates<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_003"><sup>†</sup></xref></td> <td align="left" valign="top">Total: 30</td> <td align="left" valign="top">N/A</td> <td align="left" valign="top">Physician engagement (n & format N/R) + testing (N/R)</td> <td align="left" valign="top">12</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Novo Nordisk Inc</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Scalone et al.<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_029">29</xref>]</sup></td> <td align="left" valign="top">2009</td> <td align="left" valign="top">Physicians, patients and pharmacists</td> <td align="left" valign="top">Italy</td> <td align="left" valign="top">Coagulation factor concentrates<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_003"><sup>†</sup></xref></td> <td align="left" valign="top">Total: 101<break/>Physicians: 39<break/>Patients: 37<break/>Pharmacists: 25</td> <td align="left" valign="top">Adults = 41.4<break/>Paedatric = 8.2</td> <td align="left" valign="top">Focus group (n=N/R) + Pilot (n=35)</td> <td align="left" valign="top">8</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Novo Nordisk Denmark</td> <td align="left" valign="top">N/R</td> </tr> <tr> <td align="left" valign="top">Brown et al.<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_016">16</xref>]</sup></td> <td align="left" valign="top">2011</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Treatment products in haemophilia<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_003"><sup>†</sup></xref></td> <td align="left" valign="top">Total: 53<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_004"><sup>*</sup></xref><break/>Patients: 23<break/>Caregivers: 30</td> <td align="left" valign="top">20.7</td> <td align="left" valign="top">Used same as Lee et al. (2008)<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_028">28</xref>]</sup></td> <td align="left" valign="top">12</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Novo Nordisk Inc</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Mohamed et al.<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_030">30</xref>]</sup></td> <td align="left" valign="top">2011</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 147<break/>Patients: 77<break/>Caregivers: 70</td> <td align="left" valign="top">Adults = 38.7<break/>Juvenile = 12.4</td> <td align="left" valign="top">Face-to-face interviews with adult patients and caregivers (n=8)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Baxter Biosciences</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Gelhorn et al.<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_031">31</xref>]</sup></td> <td align="left" valign="top">2013</td> <td align="left" valign="top">Physicians</td> <td align="left" valign="top">US and EU</td> <td align="left" valign="top">Treatment products in haemophilia A<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_003"><sup>†</sup></xref></td> <td align="left" valign="top">Total: 36<break/>Physicians EU: 17<break/>Physicians US: 19</td> <td align="left" valign="top">N/A</td> <td align="left" valign="top">Qualitative interviews (n=4) + Pilot (n=5)</td> <td align="left" valign="top">5</td> <td align="left" valign="top">3</td> <td align="left" valign="top">Baxter Biosciences</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Chaugule et al.<sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_032">32</xref>]</sup></td> <td align="left" valign="top">2015</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Willingness to pay for treatment products in haemophilia</td> <td align="left" valign="top">Total: 79</td> <td align="left" valign="top">40.0</td> <td align="left" valign="top">Assistance from Haem author + Pilot (n=5)</td> <td align="left" valign="top">5</td> <td align="left" valign="top">2–5</td> <td align="left" valign="top">None</td> <td align="left" valign="top">iPad at conference</td> </tr> <tr> <td align="left" valign="top">Lock et al. <sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_033">33</xref>]</sup></td> <td align="left" valign="top">2016</td> <td align="left" valign="top">Patients, caregivers and HCPs</td> <td align="left" valign="top">Multi-country</td> <td align="left" valign="top">PK-guided dosing of prophylaxis</td> <td align="left" valign="top">Total: 224<break/>HCPs: 91<break/>Patients: 114<break/>Caregivers: 19</td> <td align="left" valign="top">38.0</td> <td align="left" valign="top">Qualitative interviews + Pilot (n=10)</td> <td align="left" valign="top">5</td> <td align="left" valign="top">2–3</td> <td align="left" valign="top">Pfizer</td> <td align="left" valign="top">Paper-based survey</td> </tr> <tr> <td align="left" valign="top">Fifer et al. <sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_034">34</xref>]</sup></td> <td align="left" valign="top">2019</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">Multi-country</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 54<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_007"><sup>^</sup></xref><break/>Patients: 24<break/>Caregivers: 30</td> <td align="left" valign="top">N/R</td> <td align="left" valign="top">Qualitative interviews (n=10)</td> <td align="left" valign="top">11</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Roche</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Su et al. <sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_035">35</xref>]</sup></td> <td align="left" valign="top">2020</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 209<break/>Patients: 113<break/>Caregivers: 96</td> <td align="left" valign="top">35.5</td> <td align="left" valign="top">Qualitative interviews (n=10)<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_005"><sup>**</sup></xref> + Pilot (n=6)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">2–4</td> <td align="left" valign="top">Sanofi Genzyme</td> <td align="left" valign="top">Web-based survey</td> </tr> <tr> <td align="left" valign="top">Park et al. <sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_036">36</xref>]</sup></td> <td align="left" valign="top">2021</td> <td align="left" valign="top">Patients and caregivers</td> <td align="left" valign="top">South Korea</td> <td align="left" valign="top">Treatment products in haemophilia A</td> <td align="left" valign="top">Total: 505<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_006"><sup>***</sup></xref></td> <td align="left" valign="top">31.5</td> <td align="left" valign="top">N/R</td> <td align="left" valign="top">5</td> <td align="left" valign="top">3</td> <td align="left" valign="top">Pfizer Pharmaceuticals Korea Ltd</td> <td align="left" valign="top">Hospital-based survey</td> </tr> <tr> <td align="left" valign="top">Witkop et al. <sup>[<xref ref-type="bibr" rid="j_jhp-2022-0006_ref_037">37</xref>]</sup></td> <td align="left" valign="top">2021</td> <td align="left" valign="top">Patients</td> <td align="left" valign="top">US</td> <td align="left" valign="top">Haemophilia gene therapy</td> <td align="left" valign="top">Total: 183</td> <td align="left" valign="top">38.5</td> <td align="left" valign="top">Qualitative interviews (n=7) <xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_005"><sup>**</sup></xref> + Pilot (n=14)</td> <td align="left" valign="top">6</td> <td align="left" valign="top">3–4</td> <td align="left" valign="top">uniQure Inc.</td> <td align="left" valign="top">Web-based survey</td> </tr> </tbody> </table><h4 class="mb-4 mt-4">Potential future treatments in haemophilia*</h4><table frame="hsides" rules="all"> <thead> <tr> <th align="left" valign="bottom"><bold>PRODUCT NAME</bold></th> <th align="left" valign="bottom"><bold>THERAPEUTIC FOCUS</bold></th> <th align="left" valign="bottom"><bold>PRODUCT TYPE</bold></th> <th align="left" valign="bottom"><bold>PHASE OF CLINICAL DEVELOPMENT</bold></th> </tr> </thead> <tbody> <tr> <td align="left" valign="top">Serpin PC</td> <td align="left" valign="top">Haemophilia A and Haemophilia B</td> <td align="left" valign="top">Specific inhibitor of activated protein C (APC)</td> <td align="left" valign="top">Phase II</td> </tr> <tr> <td align="left" valign="top">Mim8</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">Next generation FVIII mimetic antibody</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Etranacogene dezaparvovec</td> <td align="left" valign="top">Haemophilia B</td> <td align="left" valign="top">AAV5 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Fidanacogene elaparvovec</td> <td align="left" valign="top">Haemophilia B</td> <td align="left" valign="top">AAV8 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Valoctocogene roxaparvovec</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">AAV5 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Concizumab</td> <td align="left" valign="top">Haemophilia A and Haemophilia B</td> <td align="left" valign="top">Monoclonal antibody directed against tissue factor pathway inhibitor (TFPI)</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Fitusiran</td> <td align="left" valign="top">Haemophilia A and Haemophilia B</td> <td align="left" valign="top">RNAi therapeutic targeting antithrombin</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Efanesoctocog alfa</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">Fully recombinant factor VIII therapy independent of von Willebrand factor</td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Eptacog beta activated</td> <td align="left" valign="top">Haemophilia A and Haemophilia B (in patients with inhibitory antibodies to factor VIII or IX)</td> <td align="left" valign="top">Transgenically produced recombinant human factor VIIa</td> <td align="left" valign="top">Phase III<xref ref-type="table-fn" rid="j_jhp-2022-0006_table-fn_002"><sup>**</sup></xref></td> </tr> <tr> <td align="left" valign="top">Giroctocogene fitelparvovec</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">AAV2/6 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> <tr> <td align="left" valign="top">Dirloctocogene samoparvovec</td> <td align="left" valign="top">Haemophilia A</td> <td align="left" valign="top">AAV-LK03 gene therapy <italic>(in vivo)</italic></td> <td align="left" valign="top">Phase III</td> </tr> </tbody> </table></div></div></div><div id="reference" class="SeriesTab_card__26XnC SeriesTab_tab-pane__3pc7y card tab-pane" 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Practice\u003c/em\u003e is covered by the following services: \u003c/p\u003e \u003cul\u003e \u003cli\u003e Baidu Scholar \u003c/li\u003e \u003cli\u003e Cabell's Whitelist \u003c/li\u003e \u003cli\u003e CNKI Scholar (China National Knowledge Infrastructure) \u003c/li\u003e \u003cli\u003e CNPIEC - cnpLINKer \u003c/li\u003e \u003cli\u003e Dimensions \u003c/li\u003e \u003cli\u003e EBSCO \u003c/li\u003e \u003cli\u003e ExLibris \u003c/li\u003e \u003cli\u003e Google Scholar \u003c/li\u003e \u003cli\u003e J-Gate \u003c/li\u003e \u003cli\u003e JournalTOCs \u003c/li\u003e \u003cli\u003e KESLI-NDSL (Korean National Discovery for Science Leaders) \u003c/li\u003e \u003cli\u003e MyScienceWork \u003c/li\u003e \u003cli\u003e Naver Academic \u003c/li\u003e \u003cli\u003e Naviga (Softweco) \u003c/li\u003e \u003cli\u003e QOAM (Quality Open Access Market) \u003c/li\u003e \u003cli\u003e ReadCube \u003c/li\u003e \u003cli\u003e Semantic Scholar \u003c/li\u003e \u003cli\u003e TDNet \u003c/li\u003e \u003cli\u003e WorldCat (OCLC) \u003c/li\u003e \u003cli\u003e X-MOL \u003c/li\u003e \u003c/ul\u003e"},{"type":"abstracting-and-indexing","language":"German","textformat":null,"content":"\u003cp\u003e \u003cem\u003eThe Journal of Haemophilia Practice\u003c/em\u003e ist in den folgenden Services indiziert: \u003c/p\u003e \u003cul\u003e \u003cli\u003e Baidu Scholar \u003c/li\u003e \u003cli\u003e Cabell's Whitelist \u003c/li\u003e \u003cli\u003e CNKI Scholar (China National Knowledge Infrastructure) \u003c/li\u003e \u003cli\u003e CNPIEC - cnpLINKer \u003c/li\u003e \u003cli\u003e Dimensions \u003c/li\u003e \u003cli\u003e EBSCO \u003c/li\u003e \u003cli\u003e ExLibris \u003c/li\u003e \u003cli\u003e Google Scholar \u003c/li\u003e \u003cli\u003e J-Gate \u003c/li\u003e \u003cli\u003e JournalTOCs \u003c/li\u003e \u003cli\u003e KESLI-NDSL (Korean National Discovery for Science Leaders) \u003c/li\u003e \u003cli\u003e MyScienceWork \u003c/li\u003e \u003cli\u003e Naver Academic \u003c/li\u003e \u003cli\u003e Naviga (Softweco) \u003c/li\u003e \u003cli\u003e QOAM (Quality Open Access Market) \u003c/li\u003e \u003cli\u003e ReadCube \u003c/li\u003e \u003cli\u003e Semantic Scholar \u003c/li\u003e \u003cli\u003e TDNet \u003c/li\u003e \u003cli\u003e WorldCat (OCLC) \u003c/li\u003e \u003cli\u003e X-MOL \u003c/li\u003e \u003c/ul\u003e"},{"type":"advantages","language":"English","textformat":null,"content":"\u003cP\u003e\u003cSTRONG\u003eThe Journal of Haemophilia Practice\u003c/STRONG\u003e is an international, open-access, peer-reviewed journal that accepts papers from all members of the multidisciplinary bleeding disorders care team.\u003cBR\u003eAny manuscript that is relevant to current haemophilia practice will be considered for publication by our editorial review team. To this end, we positively welcome: \u003c/P\u003e \u003cUL\u003e \u003cUL\u003e \u003cP\u003e\u003c/P\u003e\u003c/UL\u003e \u003cLI\u003eOriginal research \u003c/LI\u003e \u003cLI\u003eCase reports \u003c/LI\u003e \u003cLI\u003eCase Series \u003c/LI\u003e \u003cLI\u003eReviews in any field of bleeding disorders that are of relevance to current clinical practice \u003c/LI\u003e \u003cLI\u003eClinical Updates that describe current advances in any clinical field related to bleeding disorders \u003c/LI\u003e \u003cLI\u003eEditorials that address a particular topic of current interest. \u003c/LI\u003e \u003cUL\u003e \u003cP\u003e\u003c/P\u003e\u003c/UL\u003e\u003c/UL\u003e \u003cP\u003eAll manuscripts submitted to The Journal of Haemophilia Practice will first be subject to a technical review, including a quality check of all the files submitted, including tables, figures and references. This will include a plagiarism check (see below). Manuscripts will then be reviewed by the Editor-in-Chief, who will decide whether or not to proceed to peer-review by members of our editorial board and invited experts. The identity of the reviewers will not be disclosed to the authors. The review procedure will result in one of three decisions: \u003c/P\u003e \u003cUL\u003e \u003cUL\u003e \u003cP\u003e\u003c/P\u003e\u003c/UL\u003e \u003cLI\u003eAccept \u003c/LI\u003e \u003cLI\u003eAccept subject to revision \u003c/LI\u003e \u003cLI\u003eReject \u003c/LI\u003e \u003cUL\u003e \u003cP\u003e\u003c/P\u003e\u003c/UL\u003e\u003c/UL\u003e \u003cP\u003eThe editorial decision will be communicated to the authors as soon as the review process has been finalized. In case of revisions, the revised article will be sent to the reviewers who will decide on a new recommendation for revision, acceptance or rejection. \u003c/P\u003e \u003cP\u003eThe estimated time from the submission to first decision is approximately 4-6 weeks, and from the final revision to acceptance is approximately 2 weeks. Prior to publication, the corresponding author will receive a proof of their article in order to confirm the accuracy of the text or suggest modifications. \u003c/P\u003e \u003cP\u003e“The Journal of Haemophilia Practice” is a double-blind peer reviewed journal. The journal has no article processing charges (APCs) nor article submission charges. \u003c/P\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eArchiving\u003c/STRONG\u003e \u003c/P\u003e \u003cP\u003eSciendo archives the contents of this journal in \u003cA href=\"https://www.portico.org/\"\u003ePortico\u003c/A\u003e - digital long-term preservation service of scholarly books, journals and collections. \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003ePlagiarism Policy\u003c/STRONG\u003e \u003c/P\u003e \u003cP\u003eThe editorial board is participating in a growing community of \u003cA href=\"https://www.crossref.org/services/similarity-check/\"\u003eSimilarity Check System's\u003c/A\u003e users in order to ensure that the content published is original and trustworthy. Similarity Check is a medium that allows for comprehensive manuscripts screening, aimed to eliminate plagiarism and provide a high standard and quality peer-review process. \u003c/P\u003e"},{"type":"editorial","language":"English","textformat":null,"content":"\u003cP\u003e\u003cSTRONG\u003eEditor-in-Chief\u003c/STRONG\u003e\u003cBR\u003eDr Kate Khair, Great Ormond Street Hospital, London, UK \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eManaging Editor\u003cBR\u003e\u003c/STRONG\u003eMike Holland, Haemnet, UK \u003cBR\u003e\u003cA href=\"mailto:mike@haemnet.com\"\u003emike@haemnet.com\u003c/A\u003e \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eEditorial Advisory Board\u003cBR\u003e\u003c/STRONG\u003eTami Barazani-Brutman, Sheba Medical Center at Sheba, Tel Hashomer Hospital, Israel\u003cBR\u003ePaul Batty, Queen Mary University of London, UK, and Queen’s University, Ontario, Canada\u003cBR\u003eGreig Blamey, Health Sciences Center, University of Alberta, Canada\u003cBR\u003eErica Crilly, British Columbia Children's Hospital, Vancouver, Canada\u003cBR\u003eJanine Furmedge, The Royal Children’s Hospital Haemophilia Centre, Melbourne, Australia\u003cBR\u003eRich Gorman, Brighton and Sussex Medical School, UK\u003cBR\u003eChris Guelcher, Children’s National Hospital, Washington, USA\u003cBR\u003eChris Harrington, Royal Free Hospital, London, UK\u003cBR\u003eCathy Harrison, Sheffield Haemophilia \u0026amp; Thrombosis Centre, Royal Hallamshire Hospital, Sheffield, UK\u003cBR\u003eDan Hart, The Royal London Hospital and Barts Health NHS Trust, London, UK\u003cBR\u003eAdam Jones, University of Sunderland, UK\u003cBR\u003eRadek Kaczmarek, Indiana University School of Medicine, USA\u003cBR\u003eSebastien Lobet, Cliniques Universitaires Saint-Luc, Brussels, Belgium\u003cBR\u003ePaul McLaughlin, Royal Free Hospital, London, UK\u003cBR\u003eGreta Mulders, Erasmus University Medical Center, Rotterdam, The Netherlands\u003cBR\u003eJames Munn, University of Michigan Hemophilia and Coagulation Disorders Program, Michigan, USA\u003cBR\u003eDeclan Noone, European Haemophilia Association, Brussels\u003cBR\u003eJamie O'Hara, University of Chester, Cheshire, UK\u003cBR\u003eDebra Pollard, Royal Free Hospital, London, UK\u003cBR\u003eBJ Ramsay, Wellington Haemophilia Centre, Wellington, New Zealand\u003cBR\u003eDakota J. Rosenfelt, PharmD, Missouri, US\u003cBR\u003eSylvia von Mackensen, Institute of Medical Psychology at the University Medical Centre, Hamburg, Germany\u003cBR\u003eLinda Myrin Westesson, Sahlgrenska University Hospital, Gothenburg, Sweden\u003cBR\u003eMichelle Witkop, National Hemophilia Foundation, USA \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eTechnical Editor\u003cBR\u003e\u003c/STRONG\u003eKathryn Jenner, Haemnet, UK \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003ePublisher\u003c/STRONG\u003e\u003cBR\u003eDe Gruyter Poland\u003cBR\u003eBogumiła Zuga 32A Str.\u003cBR\u003e01-811 Warsaw, Poland\u003cBR\u003eT: +48 22 701 50 15 \u003c/P\u003e"},{"type":"submission","language":"English","textformat":null,"content":"\u003cDIV align=left\u003e \u003cP\u003eManuscripts should be submitted via the journal’s Scholar One Manuscripts site: \u003cA href=\"http://mc04.manuscriptcentral.com/jhaempract\"\u003emc04.manuscriptcentral.com/jhaempract\u003c/A\u003e. \u003c/P\u003e \u003cP\u003eYou will need to log in. If you are using Scholar One for the first time, use the ‘Create an Account’ link. \u003c/P\u003e \u003cP\u003eOne you have logged in, select the ‘Author’ tab. You should then follow the on-screen instructions – the system will guide you through the process of manuscript submission. Alongside the manuscript, accompanying figures and tables, and general information about the manuscript, you will be asked to provide: \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eThe names of suggested reviewers (optional) \u003c/LI\u003e \u003cLI\u003eA cover letter signed on behalf of all co-authors by the corresponding author \u003c/LI\u003e \u003cLI\u003eAn Open Access License agreement, completed by the corresponding author on behalf of all co-authors (available via the ‘Instructions and Forms’ tab on the journal’s Scholar One Manuscripts site: \u003cA href=\"http://mc04.manuscriptcentral.com/jhaempract\"\u003emc04.manuscriptcentral.com/jhaempract\u003c/A\u003e) \u003c/LI\u003e \u003cLI\u003eDetails of any conflicts of interest \u003c/LI\u003e \u003cLI\u003eConfirmation that informed consent has been obtained where appropriate. \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003eAfter submitting your manuscript, you will receive a confirmation email. \u003c/P\u003e \u003cP\u003eYou can check on the status of your manuscript at any time by logging in to your Scholar One account. \u003c/P\u003e \u003cP\u003eThe journal will notify you by email when a decision has been made on your manuscript. \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eManuscript types\u003c/STRONG\u003e \u003c/P\u003e \u003cP\u003eThe Journal of Haemophilia Practice accepts \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eOriginal research \u003c/LI\u003e \u003cLI\u003eCase reports \u003c/LI\u003e \u003cLI\u003eCase series \u003c/LI\u003e \u003cLI\u003eReviews in any field of bleeding disorders of relevance to current clinical practice \u003c/LI\u003e \u003cLI\u003eClinical Updates that describe current advances in any clinical field related to bleeding disorders \u003c/LI\u003e \u003cLI\u003eEditorials that address a particular topic of current interest. \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003ePlease note that case reports should meet the standards outlined in the \u003cA href=\"https://www.care-statement.org/\"\u003eCARE Case Report Guidelines\u003c/A\u003e. Authors submitting case reports are advised to consult the \u003cA href=\"https://www.care-statement.org/checklist\"\u003eCARE Checklist\u003c/A\u003e. \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eManuscript format\u003c/STRONG\u003e \u003c/P\u003e \u003cP\u003e\u003cU\u003eWordcount\u003c/U\u003e \u003c/P\u003e \u003cP\u003eAlthough we do not specify wordcounts for articles, we recommend the following: \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eFor shorter papers, e.g. case reports and small cohort studies: 1,000–3,000 words \u003c/LI\u003e \u003cLI\u003eFor reviews, larger studies and qualitative studies, which require explanation of methodologies: 3,000–7,000 words. \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003e\u003cU\u003eTitle\u003c/U\u003e \u003c/P\u003e \u003cP\u003eThe title should be short and informative. For case reports, the words ‘case report’ should be included in the title. \u003c/P\u003e \u003cP\u003e\u003cU\u003eAuthors\u003c/U\u003e \u003c/P\u003e \u003cP\u003eFull author names and affiliation should be given. An ORCID iD for each author should be provided where available. \u003c/P\u003e \u003cP\u003e\u003cU\u003eAbstract\u003c/U\u003e \u003c/P\u003e \u003cP\u003eFor original articles, a short abstract should be provided using the following headings: Background or Introduction, Aims, Methods, Results, Conclusion. \u003c/P\u003e \u003cP\u003e\u003cU\u003eKeywords\u003c/U\u003e \u003c/P\u003e \u003cP\u003eSubmissions should include 3–6 keywords for indexing purposes. \u003c/P\u003e \u003cP\u003e\u003cU\u003eReference style\u003c/U\u003e \u003c/P\u003e \u003cP\u003eJournal titles should be abbreviated according to the style of Index Medicus and spelled out in full if not listed in Index Medicus. These can be looked up in the \u003cA href=\"https://www.ncbi.nlm.nih.gov/nlmcatalog/journals\"\u003eNLM Catalog\u003c/A\u003e. \u003c/P\u003e \u003cP\u003eThe DOI (digital object identifier) associated with journal articles should be provided where available. \u003c/P\u003e \u003cP\u003eWhere there are six or more authors associated with a reference, the first three should be listed, followed by ‘et al.’ \u003c/P\u003e \u003cP\u003eDatasets should be cited using the format outlined by the \u003cA href=\"https://www.force11.org/datacitationprinciples\"\u003eJoint Declaration of Data Citation Principles\u003c/A\u003e: Authors; Year; Dataset title; Data repository/archive; Version (if relevant); Persistent identifier, e.g. DOI (if available). \u003c/P\u003e \u003cP\u003e\u003cEM\u003eReference style examples:\u003c/EM\u003e \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eReference to an article: \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003eKhair K, Klukowska A, Myrin Westesson L, et al. The burden of bleeds and other clinical determinants on caregivers of children with haemophilia (the BBC Study). Haemophilia 2019; 25(3): 416-423. doi: 10.1111/hae.13736. \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eReference to a book: \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003eJones P. Living with Haemophilia. 2002. Oxford: Oxford University Press. \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eReference to a chapter in a book: \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003eEscobar MA, Roberts HR. Less common congenital disorders of hemostasis. In: Kitchens CS, ed. Consultative Hemostasis and Thrombosis, 3rd edn. 2013. Philadelphia, PA: W.B. Saunders Company. \u003c/P\u003e \u003cUL\u003e \u003cLI\u003eReference to a webpage/online resource: \u003c/LI\u003e\u003c/UL\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003eEuropean Medicines Agency (EMA). European Public Assessment Report (EPAR) NovoSeven Summary for the Public. 2009. Available from http://www.ema.europa.eu/ema/index.jsp?curl=pages/medicines/human/medicines/000074/human_med_000936.jsp\u0026amp;mid=WC0b01ac058001d124 (accessed 9 September 2013). \u003c/P\u003e \u003cP\u003e\u003c/P\u003e \u003cP\u003e\u003cU\u003eFigures and tables\u003c/U\u003e \u003c/P\u003e \u003cP\u003ePlease provide tables and figures at the end of the manuscript or as a separate file. \u003c/P\u003e \u003cP\u003e\u003cSTRONG\u003eInformed consent\u003c/STRONG\u003e \u003c/P\u003e \u003cP\u003ePeople who are reported in studies have a right to privacy that should not be violated without informed consent. Identifying information, including names, initials, dates of birth or hospital numbers, should not be published in written descriptions, photographs or pedigrees unless the information is essential for scientific purposes and the person (or parent or guardian) gives written informed consent for publication. \u003c/P\u003e \u003cP\u003eInformed consent for this purpose requires that an identifiable person be shown the manuscript to be submitted for publication. Authors should disclose to these participants whether any potential identifiable material might be available via the internet or in print after publication. \u003c/P\u003e \u003cP\u003eEach implicated individual’s consent should be written and archived with the authors. If the patient is a minor or lacks capacity to consent, the written informed consent of a parent or guardian must be given. If the person in the case study is deceased, written consent must be sought from a relative. Without consent, a case report may be considered for publication if the patient is sufficiently anonymised according to \u003cA href=\"http://www.icmje.org/recommendations/\"\u003eICMJE guidelines\u003c/A\u003e. Nonessential identifying details should always be omitted. Informed consent should be obtained if there is any doubt that anonymity can be maintained. For example, masking the eye region in photographs of patients is inadequate protection of anonymity. \u003c/P\u003e \u003cP\u003eConsent must be mentioned in the manuscript. We reserve the right to request copies of consent documentation. 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The relative effects of determinants on Chinese adults’ decision for influenza vaccination choice: What is the effect of priming? Vaccine 2019; 37(30):4124–4132. doi: 10.1016/j.vaccine.2019.05.072.","doi":"10.1016/j.vaccine.2019.05.072","mixed-citation":"\u003cref id=\"j_jhp-2022-0006_ref_049\"\u003e\u003clabel\u003e49\u003c/label\u003e\n\u003cmixed-citation\u003eLiao Q, Lam WWT, Wong CKH, Lam C, Chen J, Fielding R. The relative effects of determinants on Chinese adults’ decision for influenza vaccination choice: What is the effect of priming? \u003citalic\u003eVaccine\u003c/italic\u003e 2019; 37(30):4124–4132. doi: \u003cpub-id pub-id-type=\"doi\"\u003e10.1016/j.vaccine.2019.05.072\u003c/pub-id\u003e.\u003c/mixed-citation\u003e\n\u003celement-citation publication-type=\"journal\" publication-format=\"print\"\u003e\n\u003cname\u003e\u003csurname\u003eLiao\u003c/surname\u003e\u003cgiven-names\u003eQ\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eLam\u003c/surname\u003e\u003cgiven-names\u003eWWT\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eWong\u003c/surname\u003e\u003cgiven-names\u003eCKH\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eLam\u003c/surname\u003e\u003cgiven-names\u003eC\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eChen\u003c/surname\u003e\u003cgiven-names\u003eJ\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eFielding\u003c/surname\u003e\u003cgiven-names\u003eR\u003c/given-names\u003e\u003c/name\u003e\n\u003carticle-title\u003eThe relative effects of determinants on Chinese adults’ decision for influenza vaccination choice: What is the effect of priming?\u003c/article-title\u003e\n\u003csource\u003eVaccine\u003c/source\u003e\n\u003cyear\u003e2019\u003c/year\u003e\n\u003cvolume\u003e37\u003c/volume\u003e\n\u003cissue\u003e30\u003c/issue\u003e\n\u003cfpage\u003e4124\u003c/fpage\u003e\n\u003clpage\u003e4132\u003c/lpage\u003e\n\u003cpub-id pub-id-type=\"doi\"\u003e10.1016/j.vaccine.2019.05.072\u003c/pub-id\u003e\n\u003cdgpm:pub-id xmlns:dgpm=\"http://degruyter.com/resources/fetched-pubmed-id\" pub-id-type=\"pmid\"\u003e31186189\u003c/dgpm:pub-id\u003e\u003c/element-citation\u003e\n\u003c/ref\u003e"},{"refId":"j_jhp-2022-0006_ref_050","citeString":"Infected Blood Inquiry [Internet]. 2021. Available from: https://www.infectedbloodinquiry.org.uk/ (accessed 8 September 2021).","doi":null,"mixed-citation":"\u003cref id=\"j_jhp-2022-0006_ref_050\"\u003e\u003clabel\u003e50\u003c/label\u003e\n\u003cmixed-citation\u003eInfected Blood Inquiry [Internet]. 2021. Available from: \u003citalic\u003e\u003curi\u003ehttps://www.infectedbloodinquiry.org.uk/\u003c/uri\u003e\u003c/italic\u003e (accessed 8 September 2021).\u003c/mixed-citation\u003e\n\u003celement-citation publication-type=\"web\"\u003e\n\u003csource\u003eInfected Blood Inquiry [Internet]\u003c/source\u003e\n\u003cyear\u003e2021\u003c/year\u003e\n\u003ccomment\u003eAvailable from: \u003citalic\u003e\u003curi\u003ehttps://www.infectedbloodinquiry.org.uk/\u003c/uri\u003e\u003c/italic\u003e (accessed 8 September 2021).\u003c/comment\u003e\n\u003c/element-citation\u003e\n\u003c/ref\u003e"},{"refId":"j_jhp-2022-0006_ref_051","citeString":"Smith LE, Sim J, Amlôt R, et al. Side-effect expectations from COVID-19 vaccination: Findings from a nationally representative cross-sectional survey (CoVAccS – wave 2). J Psychosom Res 2021;152: 110679. doi: 10.1016/j.jpsychores.2021.110679.","doi":"10.1016/j.jpsychores.2021.110679","mixed-citation":"\u003cref id=\"j_jhp-2022-0006_ref_051\"\u003e\u003clabel\u003e51\u003c/label\u003e\n\u003cmixed-citation\u003eSmith LE, Sim J, Amlôt R, et al. Side-effect expectations from COVID-19 vaccination: Findings from a nationally representative cross-sectional survey (CoVAccS – wave 2). \u003citalic\u003eJ Psychosom Res\u003c/italic\u003e 2021;152: 110679. doi: \u003cpub-id pub-id-type=\"doi\"\u003e10.1016/j.jpsychores.2021.110679\u003c/pub-id\u003e.\u003c/mixed-citation\u003e\n\u003celement-citation publication-type=\"journal\" publication-format=\"print\"\u003e\n\u003cname\u003e\u003csurname\u003eSmith\u003c/surname\u003e\u003cgiven-names\u003eLE\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eSim\u003c/surname\u003e\u003cgiven-names\u003eJ\u003c/given-names\u003e\u003c/name\u003e\n\u003cname\u003e\u003csurname\u003eAmlôt\u003c/surname\u003e\u003cgiven-names\u003eR\u003c/given-names\u003e\u003c/name\u003e\n\u003cetal/\u003e\n\u003carticle-title\u003eSide-effect expectations from COVID-19 vaccination: Findings from a nationally representative cross-sectional survey (CoVAccS – wave 2)\u003c/article-title\u003e\n\u003csource\u003eJ Psychosom Res\u003c/source\u003e\n\u003cyear\u003e2021\u003c/year\u003e\n\u003cvolume\u003e152\u003c/volume\u003e\n\u003cfpage\u003e110679\u003c/fpage\u003e\n\u003cpub-id pub-id-type=\"doi\"\u003e10.1016/j.jpsychores.2021.110679\u003c/pub-id\u003e\n\u003cdgpm:pub-id xmlns:dgpm=\"http://degruyter.com/resources/fetched-pubmed-id\" pub-id-type=\"pmcid\"\u003e8595305\u003c/dgpm:pub-id\u003e\u003cdgpm:pub-id xmlns:dgpm=\"http://degruyter.com/resources/fetched-pubmed-id\" pub-id-type=\"pmid\"\u003e34823113\u003c/dgpm:pub-id\u003e\u003c/element-citation\u003e\n\u003c/ref\u003e"}],"pdfUrl":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/10.2478_jhp-2022-0006.pdf?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=6bee966e9d830b29bbeee881c8734911dfcdba21cd365086abc178a229405ba5","authorNotes":null,"publishMonth":"05","publishYear":"2022","receivedDate":null,"acceptedDate":null,"ePubDate":"2022-05-20T00:00:00.000+00:00","ePubDateText":"20 May 2022","pPubDate":null,"pPubDateText":null,"issueDate":"2022-01-01T00:00:00.000+00:00","coverDate":"2022-01-01T00:00:00.000+00:00","tableCount":null,"figureCount":null,"refCount":null,"articleCategories":"{\"subj-group\":{\"subject\":\"Evidence Review\"}}","titleGroup":"{\"article-title\":\"Discrete choice experiments: An overview of experience to date in haemophilia\"}","fundingGroup":null,"abstractContent":[{"title":"Abstract","language":"English","content":"\u003cabstract\u003e\n\u003ctitle style='display:none'\u003eAbstract\u003c/title\u003e\n\u003csec\u003e\u003ctitle style='display:none'\u003eBackground\u003c/title\u003e\u003cp\u003eThe patient voice is an important consideration in the availability and choice of pharmaceuticals – however, how to capture this complex area and apply it formally within regulation, health technology assessment and reimbursement remains subject to ongoing debate. Patient preference studies such as discrete choice experiments (DCEs) are being utilised more frequently in healthcare and it is anticipated that patient preference data will be incorporated more frequently into regulatory submissions moving forward.\u003c/p\u003e\u003c/sec\u003e\n\u003csec\u003e\u003ctitle style='display:none'\u003eAim\u003c/title\u003e\u003cp\u003eThe aim of this review is to provide an overview of DCEs conducted within haemophilia to date and to consider the key issues in response to a rapidly evolving therapeutic pathway.\u003c/p\u003e\u003c/sec\u003e\n\u003csec\u003e\u003ctitle style='display:none'\u003eMethods\u003c/title\u003e\u003cp\u003eA systematic literature search was undertaken via Ovid MEDLINE and EMBASE CLASSIC + EMBASE. Abstracts were uploaded and analysed via Rayyan systematic review software. Results: Of 478 records identified from the database searches, 12 full text journal articles met the inclusion criteria with a date range from 2005–2021. There have been two published studies exploring haemophilia patient preferences in relation to gene therapy: one DCE and one utilising a threshold technique. Surveyed audiences included physicians, patients, pharmacists, healthcare professionals and caregivers. 50% of the included studies (n=6) were exclusively conducted in the US, whilst 3 recruited participants across multiple countries. The sample size varied considerably between studies with the total sample size ranging from 30 participants to 505 participants. For the studies involving patients and their caregivers, the mean patient age range was 8.2–41.4 years. There was diversity in (a) the scale of the qualitative work undertaken to support the DCEs, (b) the undertaking of pilots, and (c) how extensively these elements were reported in the included studies. There is a notable trend towards using an online web-based format, with 3 out of 4 DCEs since 2019 utilising this approach. The number of attributes observed per DCE ranged from 5–12 with a median of 6 attributes from the included studies. The number of levels per attribute was relatively consistent (range 2–5) with 2–3 (n=4) and 2–4 levels (n=4) being utilised most frequently.\u003c/p\u003e\u003c/sec\u003e\n\u003csec\u003e\u003ctitle style='display:none'\u003eConclusion\u003c/title\u003e\u003cp\u003ePatient preferences and the methods for capturing these are likely to be subject to ongoing debate as the haemophilia care pathway evolves to offer more therapeutic options with a range of risks and benefits. Whilst techniques such as DCE are effective at quantifying patient preferences, they tell us little about the reasons driving these decisions and the likelihood that they will change in response to temporal or external factors. DCEs could be particularly useful for estimating the uptake of new products and assessing potential budget impact. Accelerated and reformed regulatory processes are likely to increase demand for patient preference studies. There is therefore an increased requirement to ensure that patient advocacy groups (PAGs) are resourced and have the expertise to support these studies alongside other research commitments, and that manufacturers consider collaborative approaches when formally capturing patient preferences.\u003c/p\u003e\n\u003cp\u003e\u003cfig id=\"j_jhp-2022-0006_fig_007\" position=\"float\" fig-type=\"figure\"\u003e\u003ccaption\u003e\u003cp\u003eAs more therapeutic options become available in haemophilia care, discrete choice experiment may be a useful means of gauging patient preference\u003c/p\u003e\u003cp\u003e© Shutterstock\u003c/p\u003e\u003c/caption\u003e\u003cgraphic xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_007.jpg\"/\u003e\u003c/fig\u003e\u003c/p\u003e\u003c/sec\u003e\n\u003c/abstract\u003e"}],"figures":[{"label":null,"caption":"As more therapeutic options become available in haemophilia care, discrete choice experiment may be a useful means of gauging patient preference© Shutterstock","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_007.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=79af89e716693e30c6e318b59e340f745e3eed815e332204d05fde70d8f6438d"},{"label":"Figure 1","caption":"Impact of regulatory developments on capturing patient preferences across the product lifecycle","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_001.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=8aeedcd05980675b18c6ed3725351c069d257ee7eb6b15bddae28d03ac225851"},{"label":"Figure 2","caption":"Example of a DCE in haemophilia with attributes and levels highlighted [16]","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_002.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=4f306027e74c33449db3d4f6f7618371bfa687d15351d27834073df2d286a9c8"},{"label":"Figure 3","caption":"Search terms for literature search on DCEs in haemophilia care","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_003.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=ceebd12e9ce6351080b3c40d09b0fcb5803a3838ab1bc7a1e3dfa74415f717e7"},{"label":"Figure 4","caption":"PRISMA diagram for literature search on DCEs in haemophilia care","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_004.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=ebd03085a1e2b21d6ec2e1e489b2378f9b2470b5ff343da3b72f2aab35c9f6b6"},{"label":"Figure 5","caption":"Product attributes mapped over time","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_005.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=f69a52dedfdf9f8788faeb89b44866ed259dfddcdf5ba3c5207e7e98aedd9f39"},{"label":"Figure 6","caption":"Comparison of haemophilia gene therapy patient preference studies","imageLink":"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_006.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026X-Amz-Date=20220811T015651Z\u0026X-Amz-SignedHeaders=host\u0026X-Amz-Expires=18000\u0026X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026X-Amz-Signature=138a26df76c3fe0185914e844e14f97ce223707540232da81804efdebd63e529"}],"tableContent":{"Overview of DCEs within haemophilia":"\u003ctable frame=\"hsides\" rules=\"all\"\u003e\n\u003cthead\u003e\n\u003ctr\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eAUTHOR\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eYEAR\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSURVEY AUDIENCE\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eREGION\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSTUDY TOPIC\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSAMPLE SIZE (N)\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eMEAN PATIENT AGE (YEARS)\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSCALE OF QUALITATIVE WORK\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eNO. OF ATTRIBUTES\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eLEVELS PER ATTRIBUTE\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eINDUSTRY FUNDING\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSURVEY METHODS\u003c/bold\u003e\u003c/th\u003e\n\u003c/tr\u003e\n\u003c/thead\u003e\n\u003ctbody\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMantovani et al.\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_027\"\u003e27\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2005\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians, patients and pharmacists\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eItaly\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 305\u003cbreak/\u003ePhysicians: 69\u003cbreak/\u003ePatients: 178\u003cbreak/\u003ePharmacists: 58\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e35.9\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFocus group (n=N/R) + Pilot (n=15)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBayer Italia S.p.A.\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eLee et al. \u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_028\"\u003e28\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2008\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eCoagulation factor concentrates\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/xref\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 30\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysician engagement (n \u0026amp; format N/R) + testing (N/R)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e12\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNovo Nordisk Inc\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eScalone et al.\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_029\"\u003e29\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2009\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians, patients and pharmacists\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eItaly\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eCoagulation factor concentrates\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/xref\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 101\u003cbreak/\u003ePhysicians: 39\u003cbreak/\u003ePatients: 37\u003cbreak/\u003ePharmacists: 25\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAdults = 41.4\u003cbreak/\u003ePaedatric = 8.2\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFocus group (n=N/R) + Pilot (n=35)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e8\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNovo Nordisk Denmark\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/R\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBrown et al.\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_016\"\u003e16\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2011\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/xref\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 53\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_004\"\u003e\u003csup\u003e*\u003c/sup\u003e\u003c/xref\u003e\u003cbreak/\u003ePatients: 23\u003cbreak/\u003eCaregivers: 30\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e20.7\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUsed same as Lee et al. (2008)\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_028\"\u003e28\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e12\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNovo Nordisk Inc\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMohamed et al.\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_030\"\u003e30\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2011\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 147\u003cbreak/\u003ePatients: 77\u003cbreak/\u003eCaregivers: 70\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAdults = 38.7\u003cbreak/\u003eJuvenile = 12.4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFace-to-face interviews with adult patients and caregivers (n=8)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBaxter Biosciences\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eGelhorn et al.\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_031\"\u003e31\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2013\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS and EU\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/xref\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 36\u003cbreak/\u003ePhysicians EU: 17\u003cbreak/\u003ePhysicians US: 19\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=4) + Pilot (n=5)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBaxter Biosciences\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eChaugule et al.\u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_032\"\u003e32\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2015\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWillingness to pay for treatment products in haemophilia\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 79\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e40.0\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAssistance from Haem author + Pilot (n=5)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNone\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eiPad at conference\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eLock et al. \u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_033\"\u003e33\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2016\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients, caregivers and HCPs\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMulti-country\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePK-guided dosing of prophylaxis\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 224\u003cbreak/\u003eHCPs: 91\u003cbreak/\u003ePatients: 114\u003cbreak/\u003eCaregivers: 19\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e38.0\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews + Pilot (n=10)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePfizer\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFifer et al. \u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_034\"\u003e34\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2019\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMulti-country\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 54\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_007\"\u003e\u003csup\u003e^\u003c/sup\u003e\u003c/xref\u003e\u003cbreak/\u003ePatients: 24\u003cbreak/\u003eCaregivers: 30\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/R\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=10)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e11\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eRoche\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSu et al. \u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_035\"\u003e35\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2020\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 209\u003cbreak/\u003ePatients: 113\u003cbreak/\u003eCaregivers: 96\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e35.5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=10)\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_005\"\u003e\u003csup\u003e**\u003c/sup\u003e\u003c/xref\u003e + Pilot (n=6)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSanofi Genzyme\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePark et al. \u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_036\"\u003e36\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2021\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSouth Korea\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 505\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_006\"\u003e\u003csup\u003e***\u003c/sup\u003e\u003c/xref\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e31.5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/R\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePfizer Pharmaceuticals Korea Ltd\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHospital-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWitkop et al. \u003csup\u003e[\u003cxref ref-type=\"bibr\" rid=\"j_jhp-2022-0006_ref_037\"\u003e37\u003c/xref\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2021\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia gene therapy\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 183\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e38.5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=7) \u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_005\"\u003e\u003csup\u003e**\u003c/sup\u003e\u003c/xref\u003e + Pilot (n=14)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e3–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003euniQure Inc.\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003c/tbody\u003e\n\u003c/table\u003e","Potential future treatments in haemophilia*":"\u003ctable frame=\"hsides\" rules=\"all\"\u003e\n\u003cthead\u003e\n\u003ctr\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003ePRODUCT NAME\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eTHERAPEUTIC FOCUS\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003ePRODUCT TYPE\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003ePHASE OF CLINICAL DEVELOPMENT\u003c/bold\u003e\u003c/th\u003e\n\u003c/tr\u003e\n\u003c/thead\u003e\n\u003ctbody\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSerpin PC\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSpecific inhibitor of activated protein C (APC)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase II\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMim8\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNext generation FVIII mimetic antibody\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eEtranacogene dezaparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV5 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFidanacogene elaparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV8 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eValoctocogene roxaparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV5 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eConcizumab\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMonoclonal antibody directed against tissue factor pathway inhibitor (TFPI)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFitusiran\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eRNAi therapeutic targeting antithrombin\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eEfanesoctocog alfa\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFully recombinant factor VIII therapy independent of von Willebrand factor\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eEptacog beta activated\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B (in patients with inhibitory antibodies to factor VIII or IX)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTransgenically produced recombinant human factor VIIa\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003cxref ref-type=\"table-fn\" rid=\"j_jhp-2022-0006_table-fn_002\"\u003e\u003csup\u003e**\u003c/sup\u003e\u003c/xref\u003e\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eGiroctocogene fitelparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV2/6 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eDirloctocogene samoparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV-LK03 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003c/tbody\u003e\n\u003c/table\u003e"},"tables":null,"articleContent":"\n\u003cdiv\u003e\n\u003csec id=\"j_jhp-2022-0006_s_001\"\u003e\u003ctitle/\u003e\n\u003cp\u003eThe patient voice is an important consideration in the availability and choice of pharmaceuticals – however, how to capture this complex area and apply it formally within regulation, health technology assessment and reimbursement remains subject to ongoing debate \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_001\"\u003e1\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_002\"\u003e2\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_003\"\u003e3\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cp\u003eThe 21st Century Cures Act in the USA highlights the importance of considering the patient experience during the drug development process \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_004\"\u003e4\u003c/a\u003e]\u003c/sup\u003e. The Act facilitates the submission of patient experience information and ‘real world evidence’ to enable more rapid drug and device approval \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_005\"\u003e5\u003c/a\u003e]\u003c/sup\u003e. However, there remains ambiguity surrounding what constitutes real evidence and concerns that overreliance on this data may potentially mislead clinicians and expose patients to unsafe/ineffective treatments \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_005\"\u003e5\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_006\"\u003e6\u003c/a\u003e]\u003c/sup\u003e. Despite these evidential challenges, it is likely that a rise in patient preference data being incorporated in regulatory submissions will be observed moving forward \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_007\"\u003e7\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cp\u003ePharmaceutical regulators such as the Food and Drug Administration (FDA), European Medicines Agency (EMA), and Medicines \u0026amp; Healthcare products Regulatory Agency (MHRA) are accelerating the availability of medicines which target an unmet need \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_008\"\u003e8\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_009\"\u003e9\u003c/a\u003e]\u003c/sup\u003e. Recent examples of this include Project Orbis \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_010\"\u003e10\u003c/a\u003e]\u003c/sup\u003e and the Innovative Licensing and Access Pathway (ILAP) \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_011\"\u003e11\u003c/a\u003e]\u003c/sup\u003e. As health technology assessment (HTA) evolves to meet this scenario \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_012\"\u003e12\u003c/a\u003e]\u003c/sup\u003e, accelerated access pathways can also exacerbate the challenge of how to capture patient preferences within HTA and the role patient advocacy groups (PAGs) play within this process. \u003ca ref-type=\"fig\" href=\"#j_jhp-2022-0006_fig_001\"\u003eFigure 1\u003c/a\u003e highlights the role played by patients and PAGs in product availability and choice across the lifecycle of a pharmaceutical product. As accelerated initiatives and revised regulatory frameworks will require the formal capture of patient input at an earlier stage, PAGs will have increasing opportunities to campaign and provide support over a wider timeframe.\u003c/p\u003e\n\u003cfigure id=\"j_jhp-2022-0006_fig_001\" position=\"float\" fig-type=\"figure\"\u003e\u003ch2\u003eFigure 1\u003c/h2\u003e\u003cfigCaption\u003e\u003cp\u003eImpact of regulatory developments on capturing patient preferences across the product lifecycle\u003c/p\u003e\u003c/figCaption\u003e\u003cimg xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_001.jpg\" src=\"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_001.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026amp;X-Amz-Date=20220811T015651Z\u0026amp;X-Amz-SignedHeaders=host\u0026amp;X-Amz-Expires=18000\u0026amp;X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026amp;X-Amz-Signature=8aeedcd05980675b18c6ed3725351c069d257ee7eb6b15bddae28d03ac225851\" class=\"mw-100\"\u003e\u003c/img\u003e\u003c/figure\u003e\n\u003csec id=\"j_jhp-2022-0006_s_001_s_001\"\u003e\u003cdiv\u003ePatient preference studies\u003c/div\u003e\n\u003cp\u003ePatient preference studies can be either qualitative or quantitative and seek to capture the desirability of particular characteristics which are associated with a product in a given healthcare scenario \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_003\"\u003e3\u003c/a\u003e]\u003c/sup\u003e. The Medical Device Innovation Consortium (MDIC) have developed a framework for incorporating information on patient preferences regarding benefit and risk into regulatory assessments of new medical technologies \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_013\"\u003e13\u003c/a\u003e]\u003c/sup\u003e. There is no algorithmic approach to determine which patient preference method to use; method selection is a complex issue which depends on the research question being addressed, the population being studied, and time/budgetary constraints \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_014\"\u003e14\u003c/a\u003e]\u003c/sup\u003e. The MDIC report helpfully provides a catalogue of patient preference methods and groups them by the type of information provided, namely, structured weighting, health-state utility, stated preference and revealed preference \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_013\"\u003e13\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003c/sec\u003e\n\u003csec id=\"j_jhp-2022-0006_s_001_s_002\"\u003e\u003cdiv\u003eDiscrete choice experiments\u003c/div\u003e\n\u003cp\u003eDiscrete choice experiments (DCEs) are a stated preference technique which allows researchers to uncover how individuals value selected attributes of a programme, product or service by asking them to state their choice over different hypothetical alternatives \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_015\"\u003e15\u003c/a\u003e]\u003c/sup\u003e. Stated preference techniques such as DCEs utilise hypothetical examples, typically in the form of a questionnaire, and rely on respondents making choices based on these; revealed preferences analyse patient choices and behaviours in the real world, with examples including patient preference trials or direct questions within clinical trials \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_013\"\u003e13\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cp\u003eA DCE is a quantitative technique for eliciting individual preferences. It is grounded in random utility theory and relies on the assumptions of economic rationality and utility maximisation \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_015\"\u003e15\u003c/a\u003e]\u003c/sup\u003e. This means there is a core underlying assumption that participants can rationally select the choice which gives them the most benefit. The outputs from DCEs show the strength of relative preferences of the characteristics under evaluation and the rate at which they are traded off. The results are often expressed in terms of utilities or marginal rates of substitution. For example, a DCE could investigate the strength of preference for a treatment that is considered more effective than an existing treatment but requires more frequent administration, and the balance between the two that is considered optimal by recipients.\u003c/p\u003e\n\u003cp\u003eEstablishing the attributes and their associated levels is one of the most important and challenging steps in designing a DCE. Attributes represent a technology's key characteristic (e.g. frequency of infusion) whereas levels refer to potential options for a specific attribute (e.g. daily, once a week, once a month). The underlying validity of the study rests on specifying these correctly. Once the attributes are established, the associated levels need to be assigned. These should reflect the range of situations that respondents might be likely to experience \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_015\"\u003e15\u003c/a\u003e]\u003c/sup\u003e. Once the attribute levels are established the next step is to generate a set of hypothetical choice sets \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_015\"\u003e15\u003c/a\u003e]\u003c/sup\u003e. An example from a DCE in haemophilia, with the attributes and levels highlighted, is given in \u003ca ref-type=\"fig\" href=\"#j_jhp-2022-0006_fig_002\"\u003eFigure 2\u003c/a\u003e \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_016\"\u003e16\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cfigure id=\"j_jhp-2022-0006_fig_002\" position=\"float\" fig-type=\"figure\"\u003e\u003ch2\u003eFigure 2\u003c/h2\u003e\u003cfigCaption\u003e\u003cp\u003eExample of a DCE in haemophilia with attributes and levels highlighted \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_016\"\u003e16\u003c/a\u003e]\u003c/sup\u003e\u003c/p\u003e\u003c/figCaption\u003e\u003cimg xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_002.jpg\" src=\"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_002.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026amp;X-Amz-Date=20220811T015651Z\u0026amp;X-Amz-SignedHeaders=host\u0026amp;X-Amz-Expires=18000\u0026amp;X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026amp;X-Amz-Signature=4f306027e74c33449db3d4f6f7618371bfa687d15351d27834073df2d286a9c8\" class=\"mw-100\"\u003e\u003c/img\u003e\u003c/figure\u003e\n\u003cp\u003eDCEs are being utilised widely in healthcare and health economics and the method is being used with increasing sophistication in design alongside enhanced analytical techniques which are contributing to a higher quality of output \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_017\"\u003e17\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_018\"\u003e18\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_019\"\u003e19\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003c/sec\u003e\n\u003csec id=\"j_jhp-2022-0006_s_001_s_003\"\u003e\u003cdiv\u003eDCEs in an evolving haemophilia pathway\u003c/div\u003e\n\u003cp\u003eTreatment with haemophilia in the mainstream consists of replacement therapy with coagulation factor VIII or factor IX, with a complication being the development of inhibitory antibodies against the infused factor VIII or IX \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_020\"\u003e20\u003c/a\u003e]\u003c/sup\u003e. The journey to the safe and routine infusion of factor VIII and IX therapies has been a challenging one and the freeze-dried powdered concentrates emerging in the 1970s were found to be contaminated by the transmission of HIV and hepatitis C in blood products \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_021\"\u003e21\u003c/a\u003e]\u003c/sup\u003e. As a result of this tragic phenomenon, tighter screening methods were implemented and recombinant (non-plasma derived) technologies were developed \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_021\"\u003e21\u003c/a\u003e]\u003c/sup\u003e. Treatments have continued to evolve in haemophilia with bypassing agents, long-acting coagulator factors, biological therapies and the emergence of gene therapy. Horizon scanning from the Specialist Pharmacy Service (SPS) \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_022\"\u003e22\u003c/a\u003e]\u003c/sup\u003e has recently identified a number of forthcoming technologies in haemophilia (\u003ca ref-type=\"table\" href=\"#j_jhp-2022-0006_tab_001\"\u003eTable 1\u003c/a\u003e).\u003c/p\u003e\n\u003ctable-wrap id=\"j_jhp-2022-0006_tab_001\" position=\"float\"\u003e\u003clabel\u003eTable 1\u003c/label\u003e\u003ccaption\u003e\u003cp\u003ePotential future treatments in haemophilia\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_001\"\u003e\u003csup\u003e*\u003c/sup\u003e\u003c/a\u003e\u003c/p\u003e\u003c/caption\u003e\n\u003ctable frame=\"hsides\" rules=\"all\"\u003e\n\u003cthead\u003e\n\u003ctr\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003ePRODUCT NAME\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eTHERAPEUTIC FOCUS\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003ePRODUCT TYPE\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003ePHASE OF CLINICAL DEVELOPMENT\u003c/bold\u003e\u003c/th\u003e\n\u003c/tr\u003e\n\u003c/thead\u003e\n\u003ctbody\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSerpin PC\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSpecific inhibitor of activated protein C (APC)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase II\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMim8\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNext generation FVIII mimetic antibody\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eEtranacogene dezaparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV5 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFidanacogene elaparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV8 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eValoctocogene roxaparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV5 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eConcizumab\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMonoclonal antibody directed against tissue factor pathway inhibitor (TFPI)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFitusiran\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eRNAi therapeutic targeting antithrombin\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eEfanesoctocog alfa\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFully recombinant factor VIII therapy independent of von Willebrand factor\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eEptacog beta activated\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A and Haemophilia B (in patients with inhibitory antibodies to factor VIII or IX)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTransgenically produced recombinant human factor VIIa\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_002\"\u003e\u003csup\u003e**\u003c/sup\u003e\u003c/a\u003e\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eGiroctocogene fitelparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV2/6 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eDirloctocogene samoparvovec\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAAV-LK03 gene therapy \u003citalic\u003e(in vivo)\u003c/italic\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhase III\u003c/td\u003e\n\u003c/tr\u003e\n\u003c/tbody\u003e\n\u003c/table\u003e\n\u003ctable-wrap-foot\u003e\n\u003cfn-group\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_001\"\u003e\u003clabel\u003e*\u003c/label\u003e\u003cp\u003eHorizon scanning data as of April 2022\u003c/p\u003e\u003c/fn\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_002\"\u003e\u003clabel\u003e**\u003c/label\u003e\u003cp\u003eMarketed in the US\u003c/p\u003e\u003c/fn\u003e\n\u003c/fn-group\u003e\n\u003c/table-wrap-foot\u003e\n\u003c/table-wrap\u003e\n\u003cp\u003eAs care pathways such as those in haemophilia become increasingly dynamic, there is a key challenge that patient preferences for advanced therapy medicinal products (ATMPs) in relation to current or future therapeutic options are unknown \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_012\"\u003e12\u003c/a\u003e]\u003c/sup\u003e. Understanding this complex interplay between therapeutic interventions will therefore be vital to inform future healthcare investment decisions. Understanding patient preferences and the impact on uptake of current and future interventions is regarded as a core challenge in budget impact analysis (BIA) and there is an acknowledgement that little data may exist to support assumptions which might be highly sensitive in the assessment \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_023\"\u003e23\u003c/a\u003e]\u003c/sup\u003e. ISPOR guidelines recommend that the mix of interventions over time should be based on past changes, market research, or clinical expert opinion \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_023\"\u003e23\u003c/a\u003e]\u003c/sup\u003e. Whilst sensitivity analysis and a commitment to transparency can assist with the interpretation of BIAs \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_023\"\u003e23\u003c/a\u003e]\u003c/sup\u003e, models which rely on historical data, observed uptake of comparable interventions, or expert opinion, fail to account for the dynamic and heterogeneous manner in which individuals make decisions \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_024\"\u003e24\u003c/a\u003e]\u003c/sup\u003e. DCEs could be particularly useful for predicting the uptake of new products where observational data from trials or pilot projects are not available \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_025\"\u003e25\u003c/a\u003e]\u003c/sup\u003e, and can potentially improve models that parameterise uptake solely based on expert opinion \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_024\"\u003e24\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003c/sec\u003e\n\u003c/sec\u003e\n\u003csec id=\"j_jhp-2022-0006_s_002\"\u003e\u003cdiv\u003eMETHODS\u003c/div\u003e\n\u003cp\u003eA systematic literature search was undertaken via Ovid MEDLINE and EMBASE CLASSIC + EMBASE with the full search terms set out in \u003ca ref-type=\"fig\" href=\"#j_jhp-2022-0006_fig_003\"\u003eFigure 3\u003c/a\u003e. Abstracts were uploaded and analysed via Rayyan systematic review software \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_026\"\u003e26\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cfigure id=\"j_jhp-2022-0006_fig_003\" position=\"float\" fig-type=\"figure\"\u003e\u003ch2\u003eFigure 3\u003c/h2\u003e\u003cfigCaption\u003e\u003cp\u003eSearch terms for literature search on DCEs in haemophilia care\u003c/p\u003e\u003c/figCaption\u003e\u003cimg xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_003.jpg\" src=\"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_003.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026amp;X-Amz-Date=20220811T015651Z\u0026amp;X-Amz-SignedHeaders=host\u0026amp;X-Amz-Expires=18000\u0026amp;X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026amp;X-Amz-Signature=ceebd12e9ce6351080b3c40d09b0fcb5803a3838ab1bc7a1e3dfa74415f717e7\" class=\"mw-100\"\u003e\u003c/img\u003e\u003c/figure\u003e\n\u003cp\u003eArticles were included if they concerned a DCE in haemophilia care, if they were a full article, and if they had a pharmacological focus. Articles were excluded for being background information, non-haemophilia specific, biological/other or non-English language.\u003c/p\u003e\n\u003cp\u003eDCEs in relevant articles were analysed in respect of the audience surveyed, sample size, geography, mean age of patients, format (e.g. paper or online), approach (including extent to which qualitative work supported the DCE, piloting and how these elements were reported), and the attributes and levels per attribute included in the DCE. The study topic and funding source were also documented. Product attributes were mapped over time to investigate temporal patterns.\u003c/p\u003e\n\u003c/sec\u003e\n\u003csec id=\"j_jhp-2022-0006_s_003\"\u003e\u003cdiv\u003eRESULTS\u003c/div\u003e\n\u003cp\u003eAs set out in the PRISMA diagram in \u003ca ref-type=\"fig\" href=\"#j_jhp-2022-0006_fig_004\"\u003eFigure 4\u003c/a\u003e, 478 records were identified with 320 records available once duplicates had been removed. A further 200 articles were excluded for being background information (n=146), non-haemophilia specific (n=26), biological/other (n=21) or non-English language (n=7). Of the 120 full-text articles assessed for eligibility, 108 were deemed ineligible due to not being a DCE (n=89), not being a full-journal article (n=18) or not having a pharmacological focus (n=1). This led to 12 full text articles being included, with a date range from 2005–2021 \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_016\"\u003e16\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_027\"\u003e27\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_028\"\u003e28\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_029\"\u003e29\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_030\"\u003e30\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_031\"\u003e31\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_032\"\u003e32\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_033\"\u003e33\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_034\"\u003e34\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_035\"\u003e35\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_036\"\u003e36\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_037\"\u003e37\u003c/a\u003e]\u003c/sup\u003e. The results are summarised in \u003ca ref-type=\"table\" href=\"#j_jhp-2022-0006_tab_002\"\u003eTable 2\u003c/a\u003e. This table has been cross-referenced to update previous work conducted on this topic \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_038\"\u003e38\u003c/a\u003e]\u003c/sup\u003e. The review also identified 3 literature reviews \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_039\"\u003e39\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_040\"\u003e40\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_041\"\u003e41\u003c/a\u003e]\u003c/sup\u003e assessing patient preferences in haemophilia, all of which were not full journal articles. There have been two published studies exploring haemophilia patient preferences in relation to gene therapy: one DCE \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_037\"\u003e37\u003c/a\u003e]\u003c/sup\u003e and one utilising a threshold technique \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_042\"\u003e42\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cfigure id=\"j_jhp-2022-0006_fig_004\" position=\"float\" fig-type=\"figure\"\u003e\u003ch2\u003eFigure 4\u003c/h2\u003e\u003cfigCaption\u003e\u003cp\u003ePRISMA diagram for literature search on DCEs in haemophilia care\u003c/p\u003e\u003c/figCaption\u003e\u003cimg xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_004.jpg\" src=\"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_004.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026amp;X-Amz-Date=20220811T015651Z\u0026amp;X-Amz-SignedHeaders=host\u0026amp;X-Amz-Expires=18000\u0026amp;X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026amp;X-Amz-Signature=ebd03085a1e2b21d6ec2e1e489b2378f9b2470b5ff343da3b72f2aab35c9f6b6\" class=\"mw-100\"\u003e\u003c/img\u003e\u003c/figure\u003e\n\u003ctable-wrap id=\"j_jhp-2022-0006_tab_002\" position=\"float\"\u003e\u003clabel\u003eTable 2\u003c/label\u003e\u003ccaption\u003e\u003cp\u003eOverview of DCEs within haemophilia\u003c/p\u003e\u003c/caption\u003e\n\u003ctable frame=\"hsides\" rules=\"all\"\u003e\n\u003cthead\u003e\n\u003ctr\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eAUTHOR\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eYEAR\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSURVEY AUDIENCE\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eREGION\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSTUDY TOPIC\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSAMPLE SIZE (N)\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eMEAN PATIENT AGE (YEARS)\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSCALE OF QUALITATIVE WORK\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eNO. OF ATTRIBUTES\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eLEVELS PER ATTRIBUTE\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eINDUSTRY FUNDING\u003c/bold\u003e\u003c/th\u003e\n\u003cth align=\"left\" valign=\"bottom\"\u003e\u003cbold\u003eSURVEY METHODS\u003c/bold\u003e\u003c/th\u003e\n\u003c/tr\u003e\n\u003c/thead\u003e\n\u003ctbody\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMantovani et al.\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_027\"\u003e27\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2005\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians, patients and pharmacists\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eItaly\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 305\u003cbreak/\u003ePhysicians: 69\u003cbreak/\u003ePatients: 178\u003cbreak/\u003ePharmacists: 58\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e35.9\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFocus group (n=N/R) + Pilot (n=15)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBayer Italia S.p.A.\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eLee et al. \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_028\"\u003e28\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2008\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eCoagulation factor concentrates\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/a\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 30\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysician engagement (n \u0026amp; format N/R) + testing (N/R)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e12\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNovo Nordisk Inc\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eScalone et al.\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_029\"\u003e29\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2009\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians, patients and pharmacists\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eItaly\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eCoagulation factor concentrates\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/a\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 101\u003cbreak/\u003ePhysicians: 39\u003cbreak/\u003ePatients: 37\u003cbreak/\u003ePharmacists: 25\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAdults = 41.4\u003cbreak/\u003ePaedatric = 8.2\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFocus group (n=N/R) + Pilot (n=35)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e8\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNovo Nordisk Denmark\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/R\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBrown et al.\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_016\"\u003e16\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2011\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/a\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 53\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_004\"\u003e\u003csup\u003e*\u003c/sup\u003e\u003c/a\u003e\u003cbreak/\u003ePatients: 23\u003cbreak/\u003eCaregivers: 30\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e20.7\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUsed same as Lee et al. (2008)\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_028\"\u003e28\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e12\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNovo Nordisk Inc\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMohamed et al.\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_030\"\u003e30\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2011\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 147\u003cbreak/\u003ePatients: 77\u003cbreak/\u003eCaregivers: 70\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAdults = 38.7\u003cbreak/\u003eJuvenile = 12.4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFace-to-face interviews with adult patients and caregivers (n=8)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBaxter Biosciences\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eGelhorn et al.\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_031\"\u003e31\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2013\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePhysicians\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS and EU\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_003\"\u003e\u003csup\u003e†\u003c/sup\u003e\u003c/a\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 36\u003cbreak/\u003ePhysicians EU: 17\u003cbreak/\u003ePhysicians US: 19\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=4) + Pilot (n=5)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eBaxter Biosciences\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eChaugule et al.\u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_032\"\u003e32\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2015\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWillingness to pay for treatment products in haemophilia\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 79\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e40.0\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eAssistance from Haem author + Pilot (n=5)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eNone\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eiPad at conference\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eLock et al. \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_033\"\u003e33\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2016\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients, caregivers and HCPs\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMulti-country\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePK-guided dosing of prophylaxis\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 224\u003cbreak/\u003eHCPs: 91\u003cbreak/\u003ePatients: 114\u003cbreak/\u003eCaregivers: 19\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e38.0\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews + Pilot (n=10)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePfizer\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePaper-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eFifer et al. \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_034\"\u003e34\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2019\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eMulti-country\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 54\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_007\"\u003e\u003csup\u003e^\u003c/sup\u003e\u003c/a\u003e\u003cbreak/\u003ePatients: 24\u003cbreak/\u003eCaregivers: 30\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/R\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=10)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e11\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eRoche\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSu et al. \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_035\"\u003e35\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2020\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 209\u003cbreak/\u003ePatients: 113\u003cbreak/\u003eCaregivers: 96\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e35.5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=10)\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_005\"\u003e\u003csup\u003e**\u003c/sup\u003e\u003c/a\u003e + Pilot (n=6)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSanofi Genzyme\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePark et al. \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_036\"\u003e36\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2021\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients and caregivers\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eSouth Korea\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTreatment products in haemophilia A\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 505\u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_006\"\u003e\u003csup\u003e***\u003c/sup\u003e\u003c/a\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e31.5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eN/R\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e3\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePfizer Pharmaceuticals Korea Ltd\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHospital-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003ctr\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWitkop et al. \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_037\"\u003e37\u003c/a\u003e]\u003c/sup\u003e\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e2021\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003ePatients\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eUS\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eHaemophilia gene therapy\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eTotal: 183\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e38.5\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eQualitative interviews (n=7) \u003ca ref-type=\"table-fn\" href=\"#j_jhp-2022-0006_table-fn_005\"\u003e\u003csup\u003e**\u003c/sup\u003e\u003c/a\u003e + Pilot (n=14)\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e6\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003e3–4\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003euniQure Inc.\u003c/td\u003e\n\u003ctd align=\"left\" valign=\"top\"\u003eWeb-based survey\u003c/td\u003e\n\u003c/tr\u003e\n\u003c/tbody\u003e\n\u003c/table\u003e\n\u003ctable-wrap-foot\u003e\n\u003cfn-group\u003e\n\u003cfn\u003e\u003cp\u003eHCPs: Healthcare professionals\u003c/p\u003e\u003c/fn\u003e\n\u003cfn\u003e\u003cp\u003eN/A: Not applicable\u003c/p\u003e\u003c/fn\u003e\n\u003cfn\u003e\u003cp\u003eN/R: Not reported\u003c/p\u003e\u003c/fn\u003e\n\u003cfn\u003e\u003cp\u003ePK: Pharmacokinetic\u003c/p\u003e\u003c/fn\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_003\"\u003e\u003clabel\u003e†\u003c/label\u003e\u003cp\u003eInhibitor-specific study\u003c/p\u003e\u003c/fn\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_004\"\u003e\u003clabel\u003e*\u003c/label\u003e\u003cp\u003eThe sample size per question varied from 51 to 53, as one or two patients did not respond to certain questions\u003c/p\u003e\u003c/fn\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_005\"\u003e\u003clabel\u003e**\u003c/label\u003e\u003cp\u003eNoting more extensive work was carried out but not fully reported\u003c/p\u003e\u003c/fn\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_006\"\u003e\u003clabel\u003e***\u003c/label\u003e\u003cp\u003eNot possible to obtain number breakdown of caregivers or patients\u003c/p\u003e\u003c/fn\u003e\n\u003cfn id=\"j_jhp-2022-0006_table-fn_007\"\u003e\u003clabel\u003e^\u003c/label\u003e\u003cp\u003eThree respondents were removed as they reported they could not understand the experiment – but not reported if caregiver or patient\u003c/p\u003e\u003c/fn\u003e\n\u003c/fn-group\u003e\n\u003c/table-wrap-foot\u003e\n\u003c/table-wrap\u003e\n\u003cp\u003eSurveyed audiences included physicians, patients, pharmacists, healthcare professionals and caregivers (either alone or in combination). Two studies focused exclusively on physicians, whilst 50% of the included studies (n=6) surveyed patients and their caregivers. Half of the studies (n=6) were conducted in the US, whilst 3 studies recruited participants across multiple countries. Eleven (92%) studies reported funding by manufacturers with a commercial interest in haemophilia. The sample size between studies varied considerably, with the total sample size ranging from 30 participants to 505 participants. For studies involving patients and their caregivers, the mean patient age range was 8.2–41.4 years; removing the juvenile/paediatric patient population figures narrows this range to 20.7–40.0 years. There was diversity in (a) the scale of the qualitative work undertaken to support the DCEs, (b) the undertaking of pilots, and (c) how extensively this was reported in the included studies. One study did not report undertaking qualitative work \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_036\"\u003e36\u003c/a\u003e]\u003c/sup\u003e and in a number of studies the extent of qualitative work or piloting was difficult to establish. It has been previously reported that inadequate information about methodological detail is hindering assessment of quality \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_018\"\u003e18\u003c/a\u003e]\u003c/sup\u003e. Given the crucial role that qualitative work and piloting plays in establishing the validity of the DCE \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_015\"\u003e15\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_017\"\u003e17\u003c/a\u003e]\u003c/sup\u003e, it is vital that future DCEs report the full details of qualitative preparatory work and piloting. Three main methods were employed: web-based, paper-based and in-person survey methods. Whilst paper-based surveys made up the earliest DCEs in this area, there is a clear trend to switching to an online web-based approach, with 3 out of 4 DCEs since 2019 utilising this format. The number of attributes observed per DCE ranged from 5–12, with a median of 6 attributes from the included studies. The levels per attribute was relatively consistent (range 2–5) with 2–3 (n=4) and 2–4 levels (n=4) being utilised most frequently. The development of recombinant products over plasma products was seen as a major therapeutic advance in haemophilia; as shown in \u003ca ref-type=\"fig\" href=\"#j_jhp-2022-0006_fig_005\"\u003eFigure 5\u003c/a\u003e, a shift of attribute focus from safety towards reduction of bleeding risk has been observed in more recent DCE studies \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_038\"\u003e38\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cfigure id=\"j_jhp-2022-0006_fig_005\" position=\"float\" fig-type=\"figure\"\u003e\u003ch2\u003eFigure 5\u003c/h2\u003e\u003cfigCaption\u003e\u003cp\u003eProduct attributes mapped over time\u003c/p\u003e\u003c/figCaption\u003e\u003cimg xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_005.jpg\" src=\"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_005.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026amp;X-Amz-Date=20220811T015651Z\u0026amp;X-Amz-SignedHeaders=host\u0026amp;X-Amz-Expires=18000\u0026amp;X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026amp;X-Amz-Signature=f69a52dedfdf9f8788faeb89b44866ed259dfddcdf5ba3c5207e7e98aedd9f39\" class=\"mw-100\"\u003e\u003c/img\u003e\u003c/figure\u003e\n\u003csec id=\"j_jhp-2022-0006_s_003_s_001\"\u003e\u003cdiv\u003eHead-to-head comparison of gene therapy studies\u003c/div\u003e\n\u003cp\u003eAs shown in \u003ca ref-type=\"table\" href=\"#j_jhp-2022-0006_tab_001\"\u003eTable 1\u003c/a\u003e, a number of gene therapies are being developed for both haemophilia A and B. Two published studies to date have explored haemophilia patient preferences in relation to gene therapy: one DCE \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_037\"\u003e37\u003c/a\u003e]\u003c/sup\u003e and one utilising a threshold technique \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_042\"\u003e42\u003c/a\u003e]\u003c/sup\u003e. Whilst DCEs have been explored extensively in this paper, the threshold technique is a method that determines the maximal change in one attribute respondents are willing to accept to achieve a given change in another attribute \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_043\"\u003e43\u003c/a\u003e]\u003c/sup\u003e. Van Overbeeke and colleagues ruled out the utilisation of DCE methodology in the development of the study protocol as they estimated that it would be challenging to recruit over 100 participants, which are generally required for DCEs \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_044\"\u003e44\u003c/a\u003e]\u003c/sup\u003e. It is widely acknowledged that method selection is a complex issue \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_014\"\u003e14\u003c/a\u003e]\u003c/sup\u003e and that both methods are stated preference studies that can be utilised to quantify patient preferences \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_013\"\u003e13\u003c/a\u003e]\u003c/sup\u003e. \u003ca ref-type=\"fig\" href=\"#j_jhp-2022-0006_fig_006\"\u003eFigure 6\u003c/a\u003e sets out a comparison of the two studies: sample sizes for both were within the range observed with other DCEs to date and, particularly impressive, both studies had to deal with disruption associated with Covid-19. Both studies reported literature reviews, qualitative interviews and piloting to develop and test study attributes and levels. Patients with both haemophilia A and B were included across the studies; sampling in the threshold technique study was aligned to the Belgian haemophilia patient population, whilst over a third of the sampled population in the DCE were haemophilia B patients. Sample severity differed considerably between the studies. The DCE study provided a relatively even split between moderate and severe patients, whilst the threshold technique study had sampling which heavily favoured severe patients. Attributes were consistent between studies with annual bleed rate (ABR), dose frequency, safety issues and quality of life (QoL) being examined, following general trends observed in this area \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_038\"\u003e38\u003c/a\u003e]\u003c/sup\u003e. The DCE also explored the impact on mental health and post-treatment effects. It has been shown previously that training materials result in more choice consistency and facilitate more complex designs \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_045\"\u003e45\u003c/a\u003e]\u003c/sup\u003e and the threshold technique study included an educational tool to assist with understanding of gene therapy. The educational tool was shown to have a significant impact on both the ABR and QoL threshold. The threshold technique study excluded non-factor therapies such as emicizumab, despite 15% (n=17) of the sample being treated with the product.\u003c/p\u003e\n\u003cfigure id=\"j_jhp-2022-0006_fig_006\" position=\"float\" fig-type=\"figure\"\u003e\u003ch2\u003eFigure 6\u003c/h2\u003e\u003cfigCaption\u003e\u003cp\u003eComparison of haemophilia gene therapy patient preference studies\u003c/p\u003e\u003c/figCaption\u003e\u003cimg xmlns:xlink=\"http://www.w3.org/1999/xlink\" xlink:href=\"graphic/j_jhp-2022-0006_fig_006.jpg\" src=\"https://sciendo-parsed-data-feed.s3.eu-central-1.amazonaws.com/6228ac980d198124537d1d3b/j_jhp-2022-0006_fig_006.jpg?X-Amz-Algorithm=AWS4-HMAC-SHA256\u0026amp;X-Amz-Date=20220811T015651Z\u0026amp;X-Amz-SignedHeaders=host\u0026amp;X-Amz-Expires=18000\u0026amp;X-Amz-Credential=AKIA6AP2G7AKP25APDM2%2F20220811%2Feu-central-1%2Fs3%2Faws4_request\u0026amp;X-Amz-Signature=138a26df76c3fe0185914e844e14f97ce223707540232da81804efdebd63e529\" class=\"mw-100\"\u003e\u003c/img\u003e\u003c/figure\u003e\n\u003c/sec\u003e\n\u003c/sec\u003e\n\u003csec id=\"j_jhp-2022-0006_s_004\"\u003e\u003cdiv\u003eDISCUSSION\u003c/div\u003e\n\u003cp\u003eThe haemophilia care pathway is constantly evolving, and this phenomenon must be considered carefully when undertaking a DCE to establish patient preferences for product attributes and levels. One of the included studies in the review encountered this issue directly: they were only able to include patients with FVIII administered intravenously, as the subcutaneous emicizumab had not been launched in Korea at the time of patient enrolment \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_036\"\u003e36\u003c/a\u003e]\u003c/sup\u003e. Whilst horizon scanning information is dynamic, and may not be available to all researchers, qualitative engagement with clinicians to future proof study design against pathway changes should be considered as part of the experimental design.\u003c/p\u003e\n\u003cp\u003eThe sample sizes in the included studies varied considerably and recruitment challenges should be actively considered when choosing the study design to capture stakeholder preferences. Methods such as the threshold technique may be more appropriate than a DCE if recruitment to the study will be challenging or the target population is small \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_044\"\u003e44\u003c/a\u003e]\u003c/sup\u003e. One study in the review had a recruitment period of 21 months \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_034\"\u003e34\u003c/a\u003e]\u003c/sup\u003e; as acknowledged in the paper, this length of recruitment may also lead to potential bias.\u003c/p\u003e\n\u003cp\u003eA recent review highlighted the vital requirement to undertake qualitative research to determine attributes and levels within DCEs, but echoed concerns that inadequate information about methodological detail was hindering assessment of quality \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_018\"\u003e18\u003c/a\u003e]\u003c/sup\u003e. Training materials remain a key but under-developed component of DCEs and the development of interactive tools can potentially improve the quality of choice data if participants are better engaged \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_045\"\u003e45\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cp\u003eThere is an increasing trend for DCEs to present attribute descriptions and content using online survey-based methods \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_045\"\u003e45\u003c/a\u003e]\u003c/sup\u003e and videos \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_046\"\u003e46\u003c/a\u003e]\u003c/sup\u003e. One study compared respondents’ understanding of attribute information based on text or video and found that although there was no systematic difference between video or text arms in the study, the information provided by video may better engage survey participants and improve their retention of content \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_046\"\u003e46\u003c/a\u003e]\u003c/sup\u003e. Another study looked at animation training materials, and whilst providing these did not change the preferences of respondents, they did result in more choice consistency which may facilitate more complex experiment designs \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_045\"\u003e45\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cp\u003eA key challenge with DCEs is that they are cross-sectional surveys, which provide a snapshot at a single point of time. This approach has advantages including being inexpensive, simple to conduct and able to reach large audiences rapidly \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_047\"\u003e47\u003c/a\u003e]\u003c/sup\u003e. However, a key limitation is that they are unable to investigate temporal effects \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_047\"\u003e47\u003c/a\u003e]\u003c/sup\u003e. There are a range of temporal and external influences on patient choice including treatment history, family and friends, evolving life situation and clinical opinion, and these factors can change over time, yet the understanding of behavioural impacts on DCEs is currently limited \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_025\"\u003e25\u003c/a\u003e,\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_048\"\u003e48\u003c/a\u003e–\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_049\"\u003e49\u003c/a\u003e]\u003c/sup\u003e. Consistency of attributes is likely to be a key issue when considering standardisation of outputs and the routine acceptance of DCE-derived patient data by HTA bodies. Key external events may also heavily influence preferences, for example, after the forthcoming UK report on the infected blood inquiry \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_050\"\u003e50\u003c/a\u003e]\u003c/sup\u003e or a major side effect with an emerging technology, as seen with Covid-19 vaccinations \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_051\"\u003e51\u003c/a\u003e]\u003c/sup\u003e.\u003c/p\u003e\n\u003cp\u003eA key statistic from the review is that all but one of the studies was either directly or indirectly funded by pharmaceutical manufacturers with a commercial interest in haemophilia. If patient preference studies are going to become a formal part of regulatory and pricing and reimbursement submissions, then it is likely that manufacturers will be expected to fund these as part of an evidential package to place before the relevant authorities. Consideration needs to be given on how to standardise the study design/outputs and evaluate quality. The alternative would have to be that PAGs, or independent institutions would have to fill this gap, which may be financially challenging and/or practically unfeasible. Consideration should be given towards a collaborative approach to undertaking DCEs involving multiple pharmaceutical manufacturers to (a) optimise PAG resource and (b) avoid potential suggestions that attributes, and levels, are selected to support individual product benefits.\u003c/p\u003e\n\u003c/sec\u003e\n\u003csec id=\"j_jhp-2022-0006_s_005\"\u003e\u003cdiv\u003eCONCLUSION\u003c/div\u003e\n\u003cp\u003ePatient preferences and the methods for capturing these are likely to be subject to ongoing debate as the haemophilia care pathway evolves to offer more therapeutic options with a range of risks and benefits. In addition to challenges with sampling, bias, methodological choice/rigour and manufacturer influence, the question of how best to use the information from patient preference studies remains unclear. Whilst techniques such as DCEs are effective at quantifying patient preferences, they tell us very little about the reasons driving these decisions and the likelihood that they will change in response to temporal or external factors. Given the evolution of the care pathway in haemophilia and the emergence of gene therapy, DCEs could be particularly useful for estimating the uptake of new products and assessing potential budget impact \u003csup\u003e[\u003ca ref-type=\"bibr\" href=\"#j_jhp-2022-0006_ref_025\"\u003e25\u003c/a\u003e]\u003c/sup\u003e. Several DCE studies in the literature review reported here highlighted the challenge of recruiting participants in rare diseases such as haemophilia, and engaging with PAGs to assist with recruitment is therefore likely to be crucial. Accelerated and reformed regulatory processes are likely to increase demand for patient preference studies and therefore there is an increased requirement to ensure that PAGs are resourced and have the expertise to support these studies alongside other research commitments, and that manufacturers consider collaborative approaches when formally capturing patient preferences.\u003c/p\u003e\n\u003c/sec\u003e\n\u003c/div\u003e","keywords":[{"title":"Keywords","language":null,"keywords":["Discrete choice experiment","Gene therapy","Haemophilia","Patient preference","Pharmaceutical","Review"]}],"recentIssues":{"10.2478/jhp-2022-0006":"\u003carticle-title\u003eDiscrete choice experiments: An overview of experience to date in haemophilia\u003c/article-title\u003e","10.2478/jhp-2022-0005":"\u003carticle-title\u003eOsseous bilateral pseudotumour of the thumb in severe haemophilia A – A case report\u003c/article-title\u003e","10.2478/jhp-2022-0008":"\u003carticle-title\u003ePoint of care ultrasonography in patients with haemophilia and acute haemarthrosis: a physiotherapist and sonographer inter-professional agreement pilot study\u003c/article-title\u003e","10.2478/jhp-2022-0007":"\u003carticle-title\u003eDevelopment of decision-making considerations to support equitable patient selection in paediatric haemophilia trials\u003c/article-title\u003e","10.2478/jhp-2022-0002":"\u003carticle-title\u003eKey challenges for patient registries – A report from the 1\u003csup\u003est\u003c/sup\u003e workshop of the EHC Think Tank Workstream on Registries\u003c/article-title\u003e","10.2478/jhp-2022-0001":"\u003carticle-title\u003eNew challenges for an expanding generation of older persons with haemophilia\u003c/article-title\u003e","10.2478/jhp-2022-0004":"\u003carticle-title\u003ePatient agency: key questions and challenges – A report from the 1st workshop of the EHC Think Tank Workstream on Patient Agency\u003c/article-title\u003e","10.2478/jhp-2022-0003":"\u003carticle-title\u003eKey challenges for hub and spoke models of care – A report from the 1st workshop of the EHC Think Tank on Hub and Spoke Treatment Models\u003c/article-title\u003e","10.2478/jhp-2022-0011":"\u003carticle-title\u003e“I didn’t know women could have haemophilia”: A qualitative case study\u003c/article-title\u003e","10.2478/jhp-2022-0010":"\u003carticle-title\u003eTreatment of a patient with severe haemophilia A presenting with left extra pleural haematoma and diagnosed with inhibitors – case report\u003c/article-title\u003e","10.2478/jhp-2022-0009":"\u003carticle-title\u003eProcesses and experiences of satellite haemophilia clinic set-ups in Uganda – a short report\u003c/article-title\u003e"},"supplement":[],"apaString":null,"mlaString":null,"harvardString":null,"chicagoString":null,"vancouverString":null,"citBIBUrl":null,"citRISUrl":null,"citENDNOTEUrl":null},"seriesKey":null,"chapters":[],"chapterData":null,"bookList":{},"bookListForBirkha":{},"bookCategories":null,"bookTitleGroup":null,"bookVolumes":null,"flyerUrl":null,"pressReleaseUrl":null,"citBIBUrl":"/article/download/cite/BIBTEXT?doi=10.2478/jhp-2022-0006","citRISUrl":"/article/download/cite/RIS?doi=10.2478/jhp-2022-0006","citENDNOTEUrl":"/article/download/cite/ENDNOTE?doi=10.2478/jhp-2022-0006","trendMDCode":"65477\r","interview":null,"lookInsideLink":null,"isNew":false,"isConference":false,"ppubDate":null,"epubDate":"2022-01-01T00:00:00.000+00:00","eissn":"2055-3390","pissn":null,"eisbn":null,"pisbn":null,"aicontent":null,"epubDateText":"01 January 2022","ppubDateText":"01 January 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