{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T00:05:27Z","timestamp":1756339527649,"version":"3.44.0"},"reference-count":19,"publisher":"BMJ","issue":"6","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2025,6,9]]},"published-print":{"date-parts":[[2025,6]]},"abstract":"<jats:p>Langerhans cell histiocytosis (LCH) is a neoplastic disease characterised by the deposition of granulomatous lesions in various tissues in the body. LCH of the lateral skull base commonly presents with scalp lesions, post-auricular lesions, otalgia and persistent ear infections. A toddler was brought to the ENT clinic with persistent left ear canal granulation tissue and bloody and malodorous discharge. After failed medical treatment with antibiotic and steroid topical drops. The ear was examined under general anaesthesia. Large friable granulation tissue was seen obstructing the external auditory canal extending to the tympanic membrane. Histological examination of the surgical specimen revealed CDa-1-positive cells, diagnostic of LCH. There were widespread enhancing tissues throughout the skull base bilaterally on CT. She was diagnosed with multiple bone LCH, enrolled in the LCH-IV trial and started on chemotherapy. This case highlights the importance of histological and radiological diagnosis of granulation tissue after failed medical treatment.<\/jats:p>","DOI":"10.1136\/bcr-2024-263728","type":"journal-article","created":{"date-parts":[[2025,6,22]],"date-time":"2025-06-22T20:40:27Z","timestamp":1750624827000},"page":"e263728","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Skull base Langerhans cell histiocytosis presenting as persistent ear discharge with granulation-like tissue in the external ear canal"],"prefix":"10.1136","volume":"18","clinical-trial-number":[{"clinical-trial-number":"nct02205762","registry":"10.18810\/clinical-trials-gov"}],"author":[{"given":"Marwa Mohammed Elrashreed","family":"Saad","sequence":"first","affiliation":[{"name":"Alder Hey Children\u2019s Hospital, Liverpool, Merseyside, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4121-8762","authenticated-orcid":false,"given":"Christopher S G","family":"Thompson","sequence":"additional","affiliation":[{"name":"Alder Hey Children\u2019s Hospital, Liverpool, Merseyside, UK"},{"name":"Anatomy, University of Edinburgh, Division of Health Sciences, Edinburgh, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mohammad","family":"Alsalem","sequence":"additional","affiliation":[{"name":"Alder Hey Children\u2019s Hospital, Liverpool, Merseyside, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2025,6,22]]},"reference":[{"key":"2025082708451551000_18.6.e263728.1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.13247"},{"key":"2025082708451551000_18.6.e263728.2","doi-asserted-by":"publisher","DOI":"10.1016\/j.critrevonc.2004.01.002"},{"key":"2025082708451551000_18.6.e263728.3","unstructured":"Tillotson CV , Reynolds SB , Patel BC . Langerhans cell histiocytosis. 2024."},{"key":"2025082708451551000_18.6.e263728.4","doi-asserted-by":"publisher","DOI":"10.1007\/s00256-006-0200-7"},{"key":"2025082708451551000_18.6.e263728.5","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24367"},{"key":"2025082708451551000_18.6.e263728.6","doi-asserted-by":"publisher","DOI":"10.1016\/j.otohns.2006.03.019"},{"key":"2025082708451551000_18.6.e263728.7","doi-asserted-by":"crossref","first-page":"135","DOI":"10.1016\/j.ijporl.2017.06.011","article-title":"Pediatric Langerhans cell histiocytosis of the lateral skull base","volume":"99","author":"Majumder","year":"2017","journal-title":"Int J Pediatr Otorhinolaryngol"},{"key":"2025082708451551000_18.6.e263728.8","doi-asserted-by":"crossref","first-page":"70","DOI":"10.5152\/iao.2023.22716","article-title":"Cholesteatoma Masquerading as Recurrent Langerhans Cell Histiocytosis","volume":"19","author":"Gendre","year":"2023","journal-title":"J Int Adv Otol"},{"key":"2025082708451551000_18.6.e263728.9","unstructured":"Board PPTE . Childhood rhabdomyosarcoma treatment (PDQ). In: PDQ cancer information summaries. 2024."},{"key":"2025082708451551000_18.6.e263728.10","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2273.2004.00764.x"},{"key":"2025082708451551000_18.6.e263728.11","doi-asserted-by":"publisher","DOI":"10.3174\/ajnr.A4676"},{"key":"2025082708451551000_18.6.e263728.12","doi-asserted-by":"publisher","DOI":"10.2214\/AJR.07.3573"},{"key":"2025082708451551000_18.6.e263728.13","doi-asserted-by":"publisher","DOI":"10.1053\/ajot.2002.123452"},{"key":"2025082708451551000_18.6.e263728.14","doi-asserted-by":"publisher","DOI":"10.1097\/00007611-198301000-00017"},{"key":"2025082708451551000_18.6.e263728.15","doi-asserted-by":"crossref","first-page":"290","DOI":"10.1097\/PEC.0000000000001092","article-title":"Cranium-Penetrating Mass Detected by Ultrasound Expedited Management of Langerhans Cell Histiocytosis","volume":"33","author":"Morgan","year":"2017","journal-title":"Pediatr Emerg Care"},{"key":"2025082708451551000_18.6.e263728.16","doi-asserted-by":"crossref","first-page":"219","DOI":"10.1177\/0145561319869605","article-title":"Langerhans Cell Histiocytosis of the Skull","volume":"100","author":"Ong","year":"2021","journal-title":"Ear Nose Throat J"},{"key":"2025082708451551000_18.6.e263728.17","doi-asserted-by":"crossref","first-page":"611","DOI":"10.1007\/s11845-008-0221-4","article-title":"Multifocal Langerhans\u2019 cell histiocytosis: a case report","volume":"179","author":"Burns","year":"2010","journal-title":"Ir J Med Sci"},{"key":"2025082708451551000_18.6.e263728.18","doi-asserted-by":"publisher","DOI":"10.2214\/ajr.174.1.1740217"},{"key":"2025082708451551000_18.6.e263728.19","doi-asserted-by":"crossref","unstructured":"Clarke R . Pediatric otolaryngology: practical clinical management. 2017.393. Available: https:\/\/www.jlo.co.uk\/book-review\/paediatric-otolaryngology-practical-clinical-management\/","DOI":"10.1055\/b-0037-145044"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2024-263728","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T15:45:25Z","timestamp":1756309525000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2024-263728"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,6]]},"references-count":19,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2025,6,22]]},"published-print":{"date-parts":[[2025,6]]}},"alternative-id":["10.1136\/bcr-2024-263728"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2024-263728","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2025,6]]}}}