Electrosclerotherapy for capillary malformations: study protocol for a randomised within-patient controlled pilot trial

Abstract

IntroductionThe current state-of-the-art treatment modality for hypertrophic capillary malformations (CMs), laser therapy, has a considerable rate of non-responders and recurrence. Intralesional bleomycin injections (or ‘sclerotherapy’) are commonly used to treat venous and lymphatic malformations with an excellent effect, but these intravascular injections are not possible in CMs due to the small diameter of the vessels. Electroporation—an electric field applied to the tissue—could increase the permeability of endothelial cells, which could theoretically facilitate targeted localised bleomycin delivery. We therefore hypothesise that bleomycin injections in combination with electroporation—‘electrosclerotherapy’ (EST), also known as ‘electrochemotherapy’—could potentially be a novel alternative treatment option for CMs.Methods and analysisIn this randomised within-patient controlled pilot trial, 20 patients with hypertrophic CMs will be enrolled. Three regions of interest (ROIs) within the CM will be randomly allocated for treatment with (A) EST, (B) bleomycin sclerotherapy without electroporation and (C) no treatment. Patients and outcome assessors are blinded for the treatment allocation. Treatment outcome for each ROI will be measured approximately 7 weeks after the treatment procedure, using patient-reported and physician-reported global assessment scores, colorimetry, laser speckle imaging and reporting of adverse events.Ethics and disseminationThe study protocol is approved by the ethics review committee of the Academic Medical Center, Amsterdam. Results will be published in peer-reviewed medical journals and will be presented at international conferences and scientific meetings. Study results will be fed back to the patient population through website and social media notifications.Trial registration numberNCT02883023;Pre-results. NTR6169.

New Treatment Option for Capillary Lymphangioma: Bleomycin-Based Electrochemotherapy of an Infant

“…18 In case of benign vascular lesions, the aim of electrochemotherapy is to enhance the sclerosing effect. 20 In our case, the growth-corrected target volume decrease was 63% after one session of electrochemotherapy. This response was considered to be good, taking into account that the lesion was benign, and, accordingly, complete eradication was not necessary.…”

Electrochemotherapy of superficial tumors – Current status:

“…In this disease, EP may promote BLM uptake. A Dutch trial is currently investigating "BLM electrosclerotherapy" in patients with hypertrophic capillary malformations [245] .…”

Bleomycin sclerotherapy for large diffuse microcystic lymphatic malformations

“…LMs are more commonly seen in lymphatic-rich areas, such as the head and neck (45-52%), axilla, mediastinum, groin, and retroperitoneum (2). Histologically, the cysts are lined with a single layer of lymphatic endothelial cells and abnormal smooth and skeletal muscular structures, and filled with eosinophilic, technique, and combinations of these treatments (5)(6)(7)(8). Macrocystic LMs respond well to sclerotherapy and surgical resection (9,10).…”