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Comparative Effects of Topical 0.2% Sirolimus for Angiofibromas in Adults and Pediatric Patients with Tuberous Sclerosis Complex.

Authors
 Lee Y.I.  ;  Lee J.H.  ;  Kim D.Y.  ;  Chung K.Y.  ;  Shin J.U. 
Citation
 Dermatology, Vol.234(1-2) : 13-22, 2018 
Journal Title
 Dermatology 
ISSN
 1018-8665 
Issue Date
2018
Keywords
Angiofibroma ; Sirolimus ; Tuberous sclerosis complex
Abstract
BACKGROUND: Recent reports have suggested that the topical formulation of sirolimus is effective in treating facial angiofibromas in tuberous sclerosis complex (TSC). Here, we determined the safety and efficacy of 0.2% topical sirolimus for the treatment of facial angiofibroma and compared its effects based on age. METHOD: This was a retrospective study which involved 36 TSC patients with facial angiofibromas who were treated with 0.2% sirolimus ointment. Its effect was evaluated using the Facial Angiofibroma Severity Index (FASI). In order to observe its comparative effect based on patient age, a subgroup analysis was performed, between the adult group (> 18 years old) and the pediatric group (≤18 years old). RESULTS: The total FASI as well as its subcategories (erythema, size, and extent) showed statistically significant improvements after the topical treatment with 0.2% sirolimus ointment (FASI before treatment: 7.2 ± 1.1, FASI after treatment: 4.4± 1.4, p < 0.001). Among the subcategories of FASI, the erythema was most significantly reduced with the fastest response to the treatment. In a subgroup analysis, the pediatric group showed significantly greater improvements in FASI (improvement of FASI in the pediatric group = 49.7 ± 12.2%, adult group = 27.9 ± 15.6%, p < 0.001). The serial improvement analysis also showed that the pediatric group achieved a consistently greater improvement in FASI at all visits. Its 1-year application in 3 patients demonstrated a continuous maintenance effect. No significant adverse effects were observed. CONCLUSION: 0.2% sirolimus ointment is safe and effective for facial angiofibromas. Considering its higher efficacy in younger patients, an early initiation of the treatment is recommended.
Full Text
https://www.karger.com/Article/FullText/489089
DOI
10.1159/000489089
Appears in Collections:
1. College of Medicine (의과대학) > Dept. of Dermatology (피부과학교실) > 1. Journal Papers
Yonsei Authors
Kim, Do Young(김도영) ORCID logo https://orcid.org/0000-0002-0194-9854
Shin, Jung U(신정우) ORCID logo https://orcid.org/0000-0001-5259-6879
Lee, Ju Hee(이주희) ORCID logo https://orcid.org/0000-0002-1739-5956
Chung, Kee Yang(정기양) ORCID logo https://orcid.org/0000-0003-3257-0297
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URI
https://ir.ymlib.yonsei.ac.kr/handle/22282913/165537
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