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Sirolimus, a mammalian target of rapamycin (mTOR) inhibitor, is an oral antitumor and immunosuppressive therapy used to treat various neoplastic disorders, including histiocytic disorders. We report two pediatric cases of JXG successfully treated with oral sirolimus monotherapy, and postulate that sirolimus may induce rapid disease resolution and long\u2010term remission for patients with both skin\u2010limited and multisystemic JXG. Our findings warrant further investigation of the relationship between the mTOR pathway and JXG.<\/jats:p>","DOI":"10.1111\/pde.15579","type":"journal-article","created":{"date-parts":[[2024,3,6]],"date-time":"2024-03-06T00:00:47Z","timestamp":1709683247000},"page":"849-852","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Oral sirolimus for the treatment of juvenile xanthogranuloma: Report of two pediatric cases"],"prefix":"10.1111","volume":"41","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6513-9033","authenticated-orcid":false,"given":"Michelle","family":"Toker","sequence":"first","affiliation":[{"name":"Albert Einstein College of Medicine  Bronx New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fatema Esaa","family":"Hassonjee","sequence":"additional","affiliation":[{"name":"Department of Dermatology Long Island Jewish Medical Center  New Hyde Park New York USA"},{"name":"Donald 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