Aqueous intralesional bleomycin sclerotherapy in lymphatic malformation: Our experience with children and adult

Abstract

Objectives:Lymphatic malformations (LMs) are aberrant proliferation of sequestrated lymphatic vessels during early embryogenesis and do not communicate directly with the general lymphatic system. The absence of vascular flow is the hallmark of LMs and is usually symptomless apart from painless disfiguring mass with concerns regarding cosmesis.Design:Sclerotherapy has gained prominence as a preferred treatment modality for macrocystic lesions. Here, we present our experience with use of aqueous bleomycin as intralesional sclerosing agent, an economical first-line treatment for macrocystic variant of LMs in children and adults. While bleomycin microsphere in oil has been commonly used in many previous studies, we have used aqueous bleomycin solution as the sclerosing modality which is easily available and economical.Materials and Methods:Twenty-seven patients of macrocystic LM including adults and children underwent bleomycin sclerotherapy under ultrasonography guidance. Number of sessions, dose administered, and the response to therapy along with all side effects were noted.Results:Sixteen patients received 3 or less sessions while rest needed 4–6 sessions of sclerotherapy for desired response. The response was excellent in 22 patients while 5 patients showed good response. Eleven patients developed minor side effects in form of fever, local infection, intracystic bleed, and local skin discoloration. Postsclerotherapy, surgery was performed in two patients.Conclusion:The better response in the present study can be attributed to targeting of individual cysts in multiloculated lesion, ultrasound-guided aspiration of the cysts content before drug delivery, and postprocedure compression which increases the contact time between cyst wall and bleomycin reducing the chances of postprocedure seroma formation. Since the drug acts on the endothelial lining of the cyst, volume of the cyst is the major determinant in response. Aqueous bleomycin had comparable results with oil-based microsphere establishing it as an economical alternative treatment modality.

Vitamin C increases DNA breaks and suppresses DNA damage-independent activation of ATM by bleomycin

“…This dangerous lung injury impairs lung functions and can be lethal. Several recent studies from different countries have found very successful clinical applications of BLM as a frontline treatment for low flow lymphatic malformations in children [8][9][10][11]. The use of BLM has now largely replaced complications-associated surgery for treatment of this disfiguring abnormalities even when presented as large masses.…”

“…The use of BLM has now largely replaced complications-associated surgery for treatment of this disfiguring abnormalities even when presented as large masses. In contrast to cancer patients, the administration of different forms of BLM for treatment of lymphatic abnormalities did not produce any short-or long-term systemic side effects and the BLM therapy is considered to be a safe alternative to surgery [8][9][10][11]. In addition to its clinical applications, BLM is also frequently used in fundamental research on the activation of DNA damage responses by DSBs.…”

Management of macrocystic lymphatic malformation at uncommon site with aqueous bleomycin sclerotherapy

“…Report of the intralesional aqueous solution of bleomycin is limited[ 13 14 ] though at our center we are using aqueous bleomycin with promising results. [ 15 ] The mode of action of belomycin in LM is the loss of secretory power caused by drug-induced inflammation on the endothelial lining rather than the killing of cells. Thus, we believe aqueous bleomycin, which is economical and easily available, can be equally effective in dealing with LM if the principal of adequate contact time post aspiration is strictly adhered to.…”

Intralesional therapies for vascular malformations of the head and neck

“…Pulmonary fibrosis is a dreaded complication of systemic bleomycin which is extremely rare with doses used for percutaneous sclerotherapy; therefore, most practitioners do not obtain pulmonary screening prior to treatment. 4 Dermatologic toxicity includes hyperpigmentation/flagellate dermatitis, which is a painless, nonpruritic, and usually self-limited reaction that often resolves within several weeks to months. 5 Although the exact mechanism of hyperpigmentation is unclear, it has been theorized that bleomycin may be more concentrated in areas of skin affected by scratching, minor trauma, or pressure (likely secondary to capillary leakage) and can be induced by skin adhesives and EKG leads.…”