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Bleomycin electrosclerotherapy (BEST) for the treatment of vascular malformations. An International Network for Sharing Practices on Electrochemotherapy (InspECT) study group report

Tobian Muir
Tobian Muir
, 
Giulia Bertino
Giulia Bertino
, 
Ales Groselj
Ales Groselj
, 
Lakshmi Ratnam
Lakshmi Ratnam
, 
Erika Kis
Erika Kis
, 
Joy Odili
Joy Odili
, 
Ian McCafferty
Ian McCafferty
, 
Walter A Wohlgemuth
Walter A Wohlgemuth
, 
Maja Cemazar
Maja Cemazar
, 
Aljosa Krt
Aljosa Krt
, 
Masa Bosnjak
Masa Bosnjak
, 
Alessandro Zanasi
Alessandro Zanasi
, 
Michela Battista
Michela Battista
, 
Francesca de Terlizzi
Francesca de Terlizzi
, 
Luca G Campana
Luca G Campana
 and 
Gregor Sersa
Gregor Sersa
   | Jun 21, 2023
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Article Category: Review
Published Online: Jun 21, 2023
Page range: 141 - 149
Received: May 26, 2023
Accepted: Jun 03, 2023
DOI: https://doi.org/10.2478/raon-2023-0029
Keywords
© 2023 Tobian Muir et al., published by Sciendo
This work is licensed under the Creative Commons Attribution 4.0 International License.

FIGURE 1.

Treatment of vascularized melanoma metastasis by electrochemotherapy. (A) Highly vascularized tumor before treatment. (B) Bleeding due to electrode insertion after application of electric pulses to the tumor. (C) Bleeding stopped immediately after electric pulse application.
Treatment of vascularized melanoma metastasis by electrochemotherapy. (A) Highly vascularized tumor before treatment. (B) Bleeding due to electrode insertion after application of electric pulses to the tumor. (C) Bleeding stopped immediately after electric pulse application.

FIGURE 2.

Patient treated by BEST. Axial T2-weighted, fat-saturated MRI with hyperintense (arrow) gluteal venous malformation before treatment (A). Axial T2-weighted, fat-saturated MRI of the same region 4 months after treatment. The main part of the venous malformation is occluded (B). Photography before (C) and after the treatment (D).
Patient treated by BEST. Axial T2-weighted, fat-saturated MRI with hyperintense (arrow) gluteal venous malformation before treatment (A). Axial T2-weighted, fat-saturated MRI of the same region 4 months after treatment. The main part of the venous malformation is occluded (B). Photography before (C) and after the treatment (D).

Clinical studies and case reports using bleomycin electrosclerotherapy40,41,42,43,44,45

Reference Type of malformation N of pts Bleomycin dose and concentration Electrodes used N of pulse applications Response Comment
McMorrow et al., Br J Oral and Maxillofacial Surg 201744 Venous malformation 1 Reduced dose: 1/3 of the standard dose Not reported Not reported Considerable improvement after 6 unsuccessful sessions with bleomycin Case report with poor respiratory function
Horbach et al., Dermatologic Surgery 202045 Hypertrophic capillary malformations 5 pts. (out of 20 planned) 0.25 mg or units/cm3 Plate & needle Not reported 7–8 weeksDEROI (changes in colorimetry)Flux in ROI (in Perfusion Units) Randomized controlled pilot trial
Dalmady et al., Pediatrics 202043 Lymphatic malformation 1 0.5 mg/kg (5.4 mg) Needle 1st session: 68 applications2nd session: 74 applications 63% growth-corrected volume decrease.No recurrence at 18 months Follow up Case report
Wohlgemuth et al., Journal of Vascular Surgery 202140 Venous malformations 17 pts. (20 lesions) Calculated based on the size of the lesion.C = 0.25 mg/mL Intralesional injection (25% concentration of standard bleomycin sclerotherapy) Needle & finger Not reported 3-month post-therapy Changes in volume MRI: Volume reduction,%:
> 90% 9 lesions Retrospective observational case study
> 70% in < 90% 6 lesions
> 50% in < 70% 2 lesions
< 35% 2 lesions
No response 1 lesion
Kostusiak et al., Dermatologic Surgery 202242 Various vascular malformations 30 pts.VM: 17AVM: 3CVMLM: 2Mixed: 2 Calculated based on the size of the lesion.Bleomycin mixed with 1 mL plain 1% lidocaineDose not reported Needle & finger Not reported 17 Complete Response7: significant improvement1: moderate improvement1: minor response1: no response3: active follow up Prospective observational case studyElectrosclerotherapy offered to non-responding patients to standard bleomycin
Krt et al., Front Oncol 202241 Arteriovenous malformation 1 750 IU BLM intralesional Plate 15 CR 18 months after BEST Case report

International Society for the Study of Vascular Anomalies (ISSVA) classification for vascular anomalies 2018

VASCULAR TUMORS VASCULAR MALFORMATIONS
Benign Locally aggressive Malignant Simple Combined
Infantile hemangioma Kaposiform hemangioendothelioma Capillary malformation (CM) CVM, CLM
Congenital hemangioma Retiform hemangioendothelioma Lymphatic malformation (LM) LVM. CLVM
Tufted hemangioma PILA. Dabska tumor Epithelioid hemangioendothelioma Venous malformation (VM) CAVM
Spindle-cell hemangioma Composite hemangioendothelioma Angiosarcoma Arteriovenous malformation (AVM)
Epithelioid hemangioma Kaposi sarcoma Arteriovenous fistula CLAVM
Pyogenic granuloma
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