Telemedicine in haemophilia during COVID-19 and beyond: a comprehensive review

A literature search of articles published in peer-reviewed journals up to 24 October 2020 was conducted by a reviewer (CL) through MEDLINE, Embase, and PubMed databases, using the search terms “telemedicine”, “hemophilia”, and their synonyms. The search strategies are included in the Appendix. Titles and abstracts were screened for relevance. If the reviewer was unsure whether an article met the inclusion criteria, a second reviewer was consulted. The full texts of all relevant abstracts were then screened for inclusion. All full text articles that addressed telemedicine used directly between consumer/patients and their providers in the context of haemophilia were included. The reference lists of included articles were hand searched to identify any additional articles that were not captured by the database search.

All included articles involved telemedicine interventions to facilitate clinical services directly between patients with haemophilia and providers without the use of a third party (e.g. another HCP). Articles were excluded if a third party was used. Studies involving virtual interventions that were not used for the purposes of consultation were excluded. Review articles, abstracts, and conference proceedings were excluded, although those identified as relevant were also hand searched for additional articles. All other study designs were included, such as observational studies, retrospective and prospective cohort studies, case-control studies, case reports, and cross-sectional studies. There were no date or language restrictions in order to capture all available papers. The eligible dates from the databases are included in the Appendix (Search strategies).

Data was extracted by CL. Data collection was based on study-specific, patient-specific, intervention-specific, and outcome-specific data. Study-specific data included the study authors, article title, journal, and country. Patient-specific data included the severity of haemophilia, ages of the patients, and virtual medium used. Intervention-specific data included the virtual medium used and the healthcare team's disciplines. Any outcome-specific data reported was collected, including patient satisfaction, provider satisfaction, patient recovery, prevention of hospital visits, patient attendance rates, clinical outcomes, and economic considerations.

A flow diagram illustrating the article search and selection process is shown in Figure 1.

Figure 1

Various technologies were used during the telemedicine consultation process, with some studies using more than one. Five described the use of telephone calls ^{[13,14,15,16,17]}, two involved the use of text/SMS messaging ^[16,17], two used email ^[13,18], and five used videoconferencing ^{[13,14,15,17,18]}. Five articles described the use of photographs through text, email, or a mobile application as part of the consultation process ^{[13,15,16,17,18]}.

All six articles described the use of telemedicine involving a multidisciplinary team including different healthcare professionals ^{[13,14,15,16,17,18]}. However, it was unclear in two articles which healthcare professionals were involved in the “hemophilia treatment center” ^[16] or who the “healthcare providers” were ^[13].

Some articles mentioned the assessments conducted, including joint range of motion assessment ^[13,14,18], muscle strength ^[18], and gait assessment ^[14]. However, none of the articles described how the assessments were performed or provided details on instruments used, questions asked, and instructions provided to patients to perform these assessments accurately.

Patient satisfaction was described in three studies, each of which measured this using study-specific surveys with different questions ^[13,14,15]. None of the studies used surveys that were validated by sources external to the study. Participants of all three studies described both positive and negative experiences with telemedicine ^[19]. Patient experiences using teleconsultation were largely positive ^[13,14,15]. The most commonly reported benefits of telemedicine contributing to patient satisfaction included ease of use ^[13,14,15], helpfulness in patient-provider communication ^[14,15], and reduction in travel time and cost ^[13,14]. Adolescents in one study commented that telemedicine improved feelings of independence and agency in caring for themselves ^[13]. However, all three studies reported equipment, internet, and connection failures as a source of frustration for patients ^[13,14,15]. Limitations with available devices for teleconsultation ^[15] and accessibility issues such as language and visual impairments ^[14] affected the patient experience.

Provider satisfaction was described in four studies ^{[13,14,15,18]}. Three used study-specific surveys but provided little detail on their content, and one (a case report) did not clarify how provider satisfaction was ascertained. In one study with 21 respondents, 90% and 86% of HCPs felt confident using telephone and video consultation, respectively ^[14]. Here, 79% of HCPs responded that they would likely continue teleconsultation as routine care, and 88% would recommend telemedicine to their colleagues ^[14]. Among physiotherapists, 94% of survey respondents were satisfied or very satisfied with their experience of videoconferencing consultations ^[14]. Gait assessment was satisfactory but joint range of motion assessment, despite being possible, may require evaluation in-person ^[14]. HCPs in three studies believed that using teleconsultation helped prevent unnecessary patient visits to treatment centres ^[13,14,15]. Interestingly, it was identified in a study with four respondents that the use of videoconferencing was not always necessary, as some providers felt that similar information could be conveyed with the use of telephones ^[13]. A case report described that providers in Haematology, Orthopaedics, and Rehabilitation Departments found telemedicine to be useful for a patient with severe haemophilia A after undergoing orthopaedic surgery ^[18]. One study reported limitations of telemedicine described by HCPs. Technical difficulties were found to occur in 38% and 71% of telephone and video consultations, respectively; 67% of HCPs reported not feeling adequately prepared in providing video consultations; and 71% of HCPs were interested in receiving further education in telemedicine ^[14].

Cost-effectiveness of telemedicine was described in four studies ^{[14,15,16,17]}, and three studies demonstrated that using telemedicine decreased the need for hospital visits ^[14,15,17]. One study reported that the introduction of teleconsultation reduced clinic non-attendance rates by 60% ^[14]. In one study the HCP was able to assess bleeding severity adequately in eight out of nine video consultations, which prevented the need for a hospital visit ^[15]. This was also demonstrated in a study conducted in Ireland, in which 49% of patients who utilised teleconferencing lived a significant distance from their haemophilia centre and would have had to travel up to three hours to attend it ^[14]. In another study bleeding frequency was reduced after the usage of an electronic diary with videoconferencing capabilities ^[16].

It is important to mention that, at the time of writing, the COVID-19 pandemic continues to affect healthcare delivery globally. Our search yielded three articles on telemedicine's specific application to haemophilia in the context of COVID-19 ^[14,17,18], with one demonstrating its implementation in Wuhan, the epicentre of the pandemic, in addition to Tianjin ^[17]. In both cities, teleconsultations were implemented in most hospitals from early February 2020, when in-person assessments were not available. This enabled HCPs to guide factor administration, adjust dosages, handle mild bleeding and prescribe coagulation factors, while reducing visits to local hospitals during the lockdown. For those without access to technology or the ability to conduct teleconsultations, home visit services were used as an alternative to in-person hospital visits when possible.

The use of telemedicine in COVID-19 was also described in a case report from Spain, where the pandemic accelerated the implementation of telemedicine services for haemophilia ^[18]. A patient with severe haemophilia A underwent a major orthopaedic surgery and was followed up through telemedicine. This minimised the risk of COVID-19 infection from hospital visits, and enabled HCPs to monitor the patient throughout their postoperative management and rehabilitation.

In another cross-sectional survey conducted in Ireland, COVID-19 resulted in a drop in clinical consultations from 252 during March 2019 to 94 in March 2020 ^[14]. The implementation of telemedicine in March 2020 enabled the clinic to recover to 230 consultations in April 2020 ^[14]. A 52% increase in medical consultations (n=407) was seen in May 2020 compared to the same month in the year prior to the COVID-19 pandemic (n=268), exceeding the clinic's original capacity. Furthermore, a 60% reduction in non-attendance was seen with the use of telehealth. Overall, telemedicine has been demonstrated as an effective intervention to mitigate effects of COVID-19 on clinical operations.

There are limitations to this review. The telemedicine interventions and technologies used in the different countries and hospital centres included in the studies we identified were not clearly outlined and may not have been the same. Due to potential discrepancies in intervention methods, the outcomes of each study may not be comparable to other studies. Additionally, the included studies did not use standardised outcome measurements, meaning that while they may have measured similar concepts, they may not have measured the same outcome. For example, where questionnaires were used, the questions and methodology of assessment varied. Finally, telemedicine is rapidly evolving and advancing due to the COVID-19 pandemic, and any outcomes described in published literature may not represent the most recent technological advances.