{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,30]],"date-time":"2026-10-30T21:12:41Z","timestamp":1793394761243,"version":"4.5.0"},"reference-count":43,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2025,9,10]],"date-time":"2025-09-10T00:00:00Z","timestamp":1757462400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Lymphatic Research and Biology"],"published-print":{"date-parts":[[2025,12,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>Venous malformations can cause substantial morbidity and long-term complications. There are no Food and Drug Administration (FDA)-approved therapies for the treatment of venous malformations. However, off-label use of sirolimus has demonstrated clinical benefit in these patients. This systematic review evaluates the efficacy and safety of sirolimus in the management of venous malformations.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods and Results:<\/jats:title>\n                    <jats:p>A systematic literature search identified 26 studies comprising 98 patients treated with sirolimus for venous malformations. Most studies were case reports or case series (77%), with 15% prospective trials and 8% retrospective analyses. Sirolimus, primarily administered orally, yielded some level of clinical improvement in 72% of reported patients, with consistent symptomatic relief observed in coagulopathy, bleeding, anemia, pain, and improved function. However, reduction in the size of the malformation varied and treatment-limiting side effects were observed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>Oral sirolimus is a promising treatment option for venous malformations, especially for those with deep venous malformations. A direct, targeted topical treatment could provide clinical benefit for the cutaneous manifestation without the risk of off-target effects as occur with oral sirolimus. Further prospective, controlled studies are warranted to elucidate sirolimus\u2019s role in managing venous malformations.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/15578585251377562","type":"journal-article","created":{"date-parts":[[2025,10,7]],"date-time":"2025-10-07T21:03:03Z","timestamp":1759870983000},"page":"311-319","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":6,"title":["Sirolimus for Venous Malformations: A Systematic Review of Efficacy and Safety"],"prefix":"10.1177","volume":"23","author":[{"given":"Joyce","family":"Teng","sequence":"first","affiliation":[{"name":"Lucile Packard Children\u2019s Hospital, Stanford University, Stanford, California, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2230-2670","authenticated-orcid":false,"given":"Jeff","family":"Martini","sequence":"additional","affiliation":[{"name":"Palvella Therapeutics, Wayne, Pennsylvania, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Kelly","sequence":"additional","affiliation":[{"name":"LGDA Alliance, Saint Petersburg, Florida, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megha","family":"Tollefson","sequence":"additional","affiliation":[{"name":"Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexander","family":"Greer","sequence":"additional","affiliation":[{"name":"Massachusetts Institute of Technology, Cambridge, Massachusetts, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2025,9,10]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1097\/JOVA.0000000000000068"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.21037\/cdt.2016.11.10"},{"key":"e_1_3_3_4_2","unstructured":"Podda S Caputy G. Venous malformations. Medscape; 2023. 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