{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T21:40:39Z","timestamp":1790458839298,"version":"4.1.0"},"reference-count":22,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2020,1,13]],"date-time":"2020-01-13T00:00:00Z","timestamp":1578873600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp; Cancer"],"published-print":{"date-parts":[[2020,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Vascular anomalies (VA), characterized by the abnormal development or growth of blood and\/or lymphatic vessels, encompasses a spectrum of conditions with a range of symptoms and complications. VA are frequently associated with cutaneous complications that can cause significant morbidity. Systemic sirolimus has previously been shown to be effective in the treatment of complicated VA. There are limited studies to date on the use of topical sirolimus for the treatment of cutaneous manifestations of VA.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Retrospective review of medical records of pediatric patients with VA treated with topical sirolimus at a single quaternary pediatric institution. Response was determined by clinical subjective and objective measures of improvement.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Twenty\u2010three patients with cutaneous VA manifestations were treated with topical sirolimus. Median age was 14 (range 4\u201027 years). The main indication for treatment was complication of lymphatic blebbing (82%, n\u00a0=\u00a019) including lymphatic fluid leakage, bleeding, pain, pruritus, swelling, or recurrent infection. Treatment course ranged from 109 to 1424 days with median of 622 days. No major side effects were reported. Eighty\u2010six percent of patients (n\u00a0=\u00a020) had subjective or objective improvement of cutaneous lesions. Lymphatic blebbing complications improved in 90% (n\u00a0=\u00a017) of individuals. Eighty\u2010two percent (n\u00a0=\u00a014) of patients not receiving concurrent systemic sirolimus demonstrated improvement with topical therapy. One patient electively stopped treatment due to pruritus and burning sensation.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Topical sirolimus appears to be a beneficial therapy for lymphatic blebbing associated with lymphatic malformations or mixed malformations with a lymphatic component, although benefit in other VA remains unclear. Topical sirolimus was well\u2010tolerated with minimal side effects.<\/jats:p><\/jats:sec>","DOI":"10.1002\/pbc.28088","type":"journal-article","created":{"date-parts":[[2020,1,13]],"date-time":"2020-01-13T09:35:48Z","timestamp":1578908148000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":23,"title":["Topical sirolimus for the treatment of cutaneous manifestations of vascular anomalies: A case series"],"prefix":"10.1002","volume":"67","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7916-9022","authenticated-orcid":false,"given":"Priscila","family":"Badia","sequence":"first","affiliation":[{"name":"Division of Hematology Hemangioma and Vascular Malformation Center Cincinnati Children's Hospital Medical Center  Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7253-5632","authenticated-orcid":false,"given":"Kiersten","family":"Ricci","sequence":"additional","affiliation":[{"name":"Division of Hematology Hemangioma and Vascular Malformation Center Cincinnati Children's Hospital Medical Center  Cincinnati Ohio"},{"name":"Department of Pediatrics University of Cincinnati College of Medicine  Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juan P.","family":"Gurria","sequence":"additional","affiliation":[{"name":"Division of Pediatric Surgery Cincinnati Children's Hospital Medical Center  Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roshni","family":"Dasgupta","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Cincinnati College of Medicine  Cincinnati Ohio"},{"name":"Division of Pediatric Surgery Cincinnati Children's Hospital Medical Center  Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manish","family":"Patel","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Cincinnati College of Medicine  Cincinnati Ohio"},{"name":"Division of Interventional Radiology Cincinnati Children's Hospital Medical Center  Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0820-4924","authenticated-orcid":false,"given":"Adrienne","family":"Hammill","sequence":"additional","affiliation":[{"name":"Division of Hematology Hemangioma and Vascular Malformation Center Cincinnati Children's Hospital Medical Center  Cincinnati Ohio"},{"name":"Department of Pediatrics University of Cincinnati College of Medicine  Cincinnati Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,1,13]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.23124"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1525-1470.1999.00069.x"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pcl.2008.01.007"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1346-8138.1997.tb02522.x"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1093\/annonc\/mdi113"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2014.12.069"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.26780"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2015.10.031"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.21927"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mayocp.2014.05.020"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2015-3257"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/ijd.13419"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40264-019-00810-9"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.2165\/11634580-000000000-00000"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1001\/archdermatol.2010.125"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2017.04.005"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2014.10.011"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2015.03.062"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2016-2105"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamadermatol.2016.3697"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/pde.13547"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamadermatol.2016.3545"}],"container-title":["Pediatric Blood &amp; 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