Outcome and clinical efficacy of bleomycin and doxycycline percutaneous sclerotherapy for treatment of paediatric lymphatic malformations in a limited resource setting
Keywords:Bleomycin, Doxycycline, Lymphatic Malformations, Sclerotherapy
Background: The aim was to study the role of bleomycin and doxycycline as a cheap and readily available sclero-therapeutic agent in the treatment of lymphatic malformations in paediatric populations of poor resource setting.
Methods: It was a longitudinal study. A total of 23 paediatric cases with distinct types of lymphatic malformations were treated with injection sclerotherapy. Bleomycin and doxycycline used for microcystic and macrocystic lesion type respectively. The patient was followed up to complete remission. The level of evidence was Level II and type of evidence was prognosis study.
Results: Commonest site of lesion was neck (78.3%), followed by cheeck (8.7%), chest, shoulder and suprapubic region. Only 21.7% of patients had good reduction (50-89%) in their lesion volume on first follow up. Overall 43.4% of patients showed a reasonable reduction in lesion volume during the follow-up period. Almost 3/4th of patients improved symptomatically on the first follow up visit. Macrocystic lesion showed an excellent response to treatment (50-89% volume reduction) in 33.3% of cases while only 16.7% of microcystic ad 12.5% of the mixed lesion showed a similar response to treatment.
Conclusions: Doxycycline sclerotherapy can be a primary treatment modality in macrocystic and mixed macrocystic lesions. It is inexpensive and widely available and has minimal side effects. In contrast, bleomycine as a sclero-therapeutic agent showed an inadequate response in size reduction of microcystic lesions.
Burrows PE, Mitri RK, Alomari A, Padua HM, Lord DJ, Sylvia MB, et al. Percutaneous sclerotherapy of lymphatic malformations with doxycycline. Lymph Res Biol. 2008;1;6(3-4):209-16.
Cordes BM, Seidel FG, Sulek M, Giannoni CM, Friedman EM. Doxycycline sclerotherapy as the primary treatment for head and neck lymphatic malformations. Otolaryngol Head Neck Surg. 2007;137(6):962-4.
Giguère CM, Bauman NM, Smith RJ. New treatment options for lymphangioma in infants and children. Ann Otol Rhinol Laryngol. 2002;111(12):1066-75.
Shiels WE, Kang DR, Murakami JW, Hogan MJ, Wiet GJ. Percutaneous treatment of lymphatic malformations. Otolaryngol Head Neck Surg. 2009;141(2):219-24.
Molitch HI, Unger EC, Witte CL, Sonnenberg E. Percutaneous sclerotherapy of lymphangiomas. Radiol. 1995;194(2):343-7.
Churchill P, Otal D, Pemberton J, Ali A, Flageole H, Walton JM. Sclerotherapy for lymphatic malformations in children: a scoping review. J Ped Surg. 2011;1;46(5):912-22.
Cheng J. Doxycycline sclerotherapy in children with head and neck lymphatic malformations. J Ped Surg. 2015;50(12):2143-6.
Chaudry G, Burrows PE, Padua HM, Dillon BJ, Fishman SJ, Alomari AI. Sclerotherapy of abdominal lymphatic malformations with doxycycline. J Vascul Interven Radiol. 2011;22(10):1431-5.
Motz KM, Nickley KB, Bedwell JR, Yadav B, Guzzetta PC, Oh AK, et al. OK432 versus doxycycline for treatment of macrocystic lymphatic malformations. Ann Otol Rhinol Laryngol. 2014;123(2):81-8.
Kennedy TL. Cystic hygroma‐lymphangioma: a rare and still unclear entity. Laryngosc. 1989;99(S1):1-0.
Gan RW, Chauhan K, Singh S. Spontaneous resolution of a recurrent axillary cystic hygroma following acute infection. BMJ Case Rep. 2015;9:bcr2015211383.
Lymphatic Malformations. NORD (National Organization for Rare Disorders). Available at: https://rarediseases.org/rare-diseases/lymphatic-malformations/. Accessed 14 December 2018.
Hassanein AH, Mulliken JB, Fishman SJ, Quatrano NA, Zurakowski D, Greene AK. Lymphatic malformation: risk of progression during childhood and adolescence. J Cranio Surg. 2012; 23(1):149-52.
Perkins JA, Manning SC, Tempero RM, Cunningham MJ, Edmonds JL, Hoffer FA, et al. Lymphatic malformations: review of current treatment. Otolaryngol Head Neck Surg. 2010;142(6):795-803.
Bowden DH, Adamson IY. Bronchiolar and alveolar lesions in the pathogenesis of crocidolite‐induced pulmonary fibrosis in mice. J Pathol. 1985;147(4):257-67.
Shergill A, John P, Amaral JG. Doxycycline sclerotherapy in children with lymphatic malformations: outcomes, complications and clinical efficacy. Ped Radiol. 2012;42(9):1080-8.
Poldervaart MT, Breugem CC, Speleman L, Pasmans S. Treatment of lymphatic malformations with OK-432 (Picibanil): review of the literature. J Cranio Surg. 2009;20(4):1159-62.
Cho BC, Kim JB, Lee JW, Choi KY, Yang JD, Lee SJ, et al. Cervicofacial lymphatic malformations: a retrospective review of 40 cases. Arch Plastic Surg. 2016;43(1):10.