Literature DB >> 30133999

A case of microcystic lymphatic malformation successfully treated with topical sirolimus.

Ercan Çalışkan1, Cemile Tuğba Altunel2, Cansel Köse Özkan3, Mustafa Tunca1.   

Abstract

Microcystic lymphatic malformation (MLM), also known as "lymphangioma circumscriptum," is a lymphatic malformation which may involve the skin and subcutaneous tissues. Progressive growth of lesions may cause pressure to the surrounding organs. Lesions frequently reoccur after treatment with surgery, sclerotherapy, radiotherapy, and laser therapy. In the last decades, oral sirolimus has been successfully used in lymphatic malformations. Since systemic treatment with sirolimus is associated with various side effects, topical form of the drug has been used with satisfying results, especially for the angiofibromas. Promisingly, few case studies indicate topical sirolimus as a potential treatment for the lymphatic malformations. Here, we report an 8-year-old girl with MLM on left trunk whose lesions recurred after the surgery that has been performed at 1 year of age. We administered twice daily topical sirolimus at 0.75 mg/ml concentration. After 2 weeks, a local irritation occurred and the dose was decreased to once daily. The treatment was stopped at the end of 3 months as the lesions were almost cleared and the patient did not want to continue to therapy. There were no new lesions after 8 months of follow up.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  lymphangioma; sirolimus; topical

Mesh:

Substances:

Year:  2018        PMID: 30133999     DOI: 10.1111/dth.12673

Source DB:  PubMed          Journal:  Dermatol Ther        ISSN: 1396-0296            Impact factor:   2.851


  3 in total

1.  Topical sirolimus 0.1% for treating cutaneous microcystic lymphatic malformations in children and adults (TOPICAL): protocol for a multicenter phase 2, within-person, randomized, double-blind, vehicle-controlled clinical trial.

Authors:  Sophie Leducq; Agnès Caille; Sébastien Barbarot; Nathalie Bénéton; Didier Bessis; Olivia Boccara; Anne-Claire Bursztejn; Christine Chiaverini; Anne Dompmartin; Catherine Droitcourt; Valérie Gissot; Dominique Goga; Laurent Guibaud; Denis Herbreteau; Anne Le Touze; Christine Léauté-Labrèze; Gérard Lorette; Stéphanie Mallet; Ludovic Martin; Juliette Mazereeuw-Hautier; Alice Phan; Patrice Plantin; Isabelle Quéré; Pierre Vabres; Hélène Bourgoin; Bruno Giraudeau; Annabel Maruani
Journal:  Trials       Date:  2019-12-17       Impact factor: 2.279

2.  Lymphangioma Circumscriptum - A Rare Cause of Gingival Enlargement: A Case Report and Review of Literature.

Authors:  Arun Sadasivan; Roshni Ramesh
Journal:  Clin Cosmet Investig Dent       Date:  2022-07-11

3.  Frog Spawns on the Tongue.

Authors:  Sudip Kumar Ghosh; Sisir Das; Satarupa Mondal; Madhab Dutta
Journal:  Turk Arch Pediatr       Date:  2022-09
  3 in total

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