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

Abstract

The treatment of microcystic and combined lymphangiomas, especially in the head and neck region, is still a challenge because the lymphangiomas do not respond to conventional therapies and their recurrence rate is high, regardless of the treatment choice. Complete surgical resection is the main treatment of lymphangiomas, but because of localization perioperative complications, such as bleeding, neural damage and airway obstruction are common disadvantages of this method. Bleomycin-based sclerotherapy is another common therapeutic approach, in which the lymphocysts are aspirated, and 25% to 50% of their volumes are replaced with a sclerotisant drug. This is an effective treatment in cases in which the vessels are large enough for an intravascular or intracystic injection, but because of the small size of vessels and cysts, the microcystic and combined lymphangiomas are not suitable for sclerotherapy. Delivery of drugs for treating sclerosis to endothelial cells can be achieved by electroporation (electrochemotherapy), even for capillary malformations. A congenital, rapidly growing combined lymphangioma of the left cervicofacial region was treated with one session of bleomycin-based electrochemotherapy. Seven months after treatment, the growth-corrected target volume decrease was 63% and the dislocation of the trachea and blood vessels previously observed had ceased. We suggest that bleomycin-based electrochemotherapy is a feasible alternative treatment option for capillary malformations.

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

“…Table 2 summarizes the clinical studies and case reports published thus far. 40 , 41 , 42 , 43 , 44 , 45 In summary, different types of vascular malformations have been treated, with favorable clinical outcomes. Most of the studies used intralesional bleomycin, either diluted or mixed with lidocaine.…”

Oral lymphangioma: A review on its treatment aspects

“…Bleomycin based electrochemotherapy was described by McMorrow et al [37] in a case report in which a venous malformation was successfully treated with intratumoral delivery of bleomycin using electrochemotherapy and referred to the procedure as bleomycin-based electrosclerotherapy. 38…”