{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T02:00:43Z","timestamp":1785117643575,"version":"3.55.0"},"reference-count":10,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T00:00:00Z","timestamp":1785110400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T00:00:00Z","timestamp":1785110400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Oral Maxillofac Surg"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Lymphangiomas are uncommon congenital benign tumors of the lymphatic system. They are typically diagnosed at birth and develop during the first years of life. The tongue is the most commonly affected structure in the oral cavity. Lymphangioma circumscriptum of the tongue is a common cause of macroglossia in children, and this macroglossia can lead to complications such as exclusive nasal breathing, airway obstructions, impaired oral feeding, esthetic disfigurement, and difficulties in mastication, swallowing, and speech. This report discusses the successful treatment of lymphangioma circumscriptum of the tongue with sirolimus, an immunosuppressant drug. The extent of the lesion made traditional surgical management impossible without excessive morbidity, so the patient was managed medically. This case report was unique due to its entirely nonsurgical treatment approach. The patient completed a 17-month course of sirolimus monotherapy with response to treatment monitored using T2 MRI. There was excellent response to treatment, full resolution of symptoms, and no treatment-related toxicity.<\/jats:p>","DOI":"10.1007\/s10006-026-01604-x","type":"journal-article","created":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T01:49:19Z","timestamp":1785116959000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Successful management of lymphangioma circumscriptum of the tongue with sirolimus monotherapy: a case report"],"prefix":"10.1007","volume":"30","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-2324-3850","authenticated-orcid":false,"given":"Allison","family":"Salmon","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paige","family":"Reilly","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ashleigh","family":"Weyh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nick","family":"Callahan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,7,27]]},"reference":[{"key":"1604_CR1","doi-asserted-by":"publisher","unstructured":"De Queiroz AM, Silv RAB, Margato CL, Nelson-Filho P (2006) Dental care management of a young patient with extensive lymphangioma of the tongue: A Case Report. 26:20\u201324. https:\/\/doi.org\/10.1111\/j.1754-4505.2006.tb01505.x","DOI":"10.1111\/j.1754-4505.2006.tb01505.x"},{"key":"1604_CR2","doi-asserted-by":"publisher","unstructured":"Jian Xchun (2005) Surgical management of lymphangiomatous or lymphangiohemangiomatous macroglossia. 63:15\u201319. https:\/\/doi.org\/10.1016\/j.joms.2004.04.024","DOI":"10.1016\/j.joms.2004.04.024"},{"key":"1604_CR3","doi-asserted-by":"publisher","unstructured":"Wiegand S, Dietz A, Wichmann G (2022) Efficacy of sirolimus in children with lymphatic malformations of the head and neck. 279:3801\u20133810. https:\/\/doi.org\/10.1007\/s00405-022-07378-8","DOI":"10.1007\/s00405-022-07378-8"},{"key":"1604_CR4","doi-asserted-by":"publisher","unstructured":"Leboulanger N, Roger G, Caze A, Enjolras O, Denoyelle F, Garabedian EN (2008) Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma. 72:953\u2013958. https:\/\/doi.org\/10.1016\/j.ijporl.2008.03.013","DOI":"10.1016\/j.ijporl.2008.03.013"},{"key":"1604_CR5","doi-asserted-by":"publisher","unstructured":"Nelson BL, Bischoff EL, Nathan A, Ma L (2020) Lymphangioma of the dorsal tongue. 14:512\u2013515. https:\/\/doi.org\/10.1007\/s12105-019-01108-z","DOI":"10.1007\/s12105-019-01108-z"},{"key":"1604_CR6","doi-asserted-by":"publisher","first-page":"1920","DOI":"10.1097\/SCS.0b013e31826cf6e3","volume":"23","author":"L Catalfamo","year":"2012","unstructured":"Catalfamo L, Nava C, Lombardo G, Iudicello V, Siniscalchi EN, Saverio DPF (2012) Tongue lymphangioma adult 23:1920\u20131922. https:\/\/doi.org\/10.1097\/SCS.0b013e31826cf6e3","journal-title":"Tongue Lymphangioma Adult"},{"key":"1604_CR7","doi-asserted-by":"publisher","first-page":"86","DOI":"10.4103\/0975-5950.85862","volume":"2","author":"M Goswami","year":"2011","unstructured":"Goswami M, Singh S, Gokkulakrishnan S, Singh A (2011) Lymphangioma tongue 2:86. https:\/\/doi.org\/10.4103\/0975-5950.85862","journal-title":"Lymphangioma tongue"},{"key":"1604_CR8","doi-asserted-by":"publisher","unstructured":"Wiegand S, Wichmann G, Dietz A (2018) Treatment of lymphatic malformations with the mTOR inhibitor sirolimus: A systematic review. 16:330\u2013339. https:\/\/doi.org\/10.1089\/lrb.2017.0062","DOI":"10.1089\/lrb.2017.0062"},{"key":"1604_CR9","doi-asserted-by":"publisher","unstructured":"Durand R, Reid JR, Belasco JB et al (2021) MRI for response assessment of extensive lymphatic malformations in children treated with sirolimus. 217:741\u2013752. https:\/\/doi.org\/10.2214\/AJR.20.24378","DOI":"10.2214\/AJR.20.24378"},{"key":"1604_CR10","doi-asserted-by":"publisher","unstructured":"Boon L, Hammer J, Seront E et al Rapamycin as novel treatment for refractory-to-standard-care slow-flow vascular malformations. https:\/\/doi.org\/10.1097\/01.prs.0000472322.66501.f6","DOI":"10.1097\/01.prs.0000472322.66501.f6"}],"container-title":["Oral and 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May 2025","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"13 July 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"27 July 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Obtained and submitted.","order":1,"name":"Ethics","label":"Ethical exemption","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"All appropriate patient photo consent was obtained. The authors affirm that the guardians of the patient in this report provided informed consent for publication of the images in Figs.\u00a0\n                      \n                      and\n                      \n                      . Clinical trial number not applicable.","order":2,"name":"Ethics","label":"Consent","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"All appropriate patient photo consent was obtained. The authors affirm that the guardians of the patient in this report provided informed consent for publication of the images in Figs.\n                      \n                      and\n                      \n                      .","order":3,"name":"Ethics","label":"Patient consent","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors declare no competing interests.","order":4,"name":"Ethics","label":"Competing interests","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":5,"name":"Ethics","label":"Clinical trial number","group":{"name":"EthicsHeading","label":"Declarations"}}],"article-number":"120"}}