{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,30]],"date-time":"2025-09-30T10:27:10Z","timestamp":1759228030045,"version":"3.44.0"},"reference-count":13,"publisher":"BMJ","issue":"12","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2021,11,14]]},"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:p>The skin manifestations of Langerhans cell histiocytosis (LCH) in the neonate have a heterogenous presentation and can mimic other causes of neonatal rashes. We report an uncommon case of LCH in a term female neonate presenting with non-specific papules and vesicles from the first day of life. There was a maternal history of genital herpes simplex virus (HSV) infection in the third trimester. Blood, cerebrospinal fluid, surface swabs and vesicular fluid were negative for HSV by PCR, and a skin biopsy confirmed the diagnosis of LCH. Further investigations for systemic involvement returned negative. Our case emphasises the variable and non-specific presentation of neonatal cutaneous LCH, which can progress to or be part of multisystem disease.<\/jats:p>","DOI":"10.1136\/bcr-2021-247213","type":"journal-article","created":{"date-parts":[[2021,12,8]],"date-time":"2021-12-08T11:50:49Z","timestamp":1638964249000},"page":"e247213","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":3,"title":["Langerhans cell histiocytosis: an unusual cause of vesicular rashes in a neonate"],"prefix":"10.1136","volume":"14","author":[{"given":"Liang Yi Justin","family":"Wee","sequence":"first","affiliation":[{"name":"Paediatric Medicine, KK Women's and Children's Hospital, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1767-4402","authenticated-orcid":false,"given":"Alvin","family":"Ngeow","sequence":"additional","affiliation":[{"name":"Department of Neonatal and Developmental Medicine, Singapore General Hospital, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4136-4105","authenticated-orcid":false,"given":"Mark","family":"Koh","sequence":"additional","affiliation":[{"name":"Department of Dermatology, KK Women\u2019s and Children\u2019s Hospital, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2021,12,8]]},"reference":[{"key":"2025082713260424000_14.12.e247213.1","article-title":"A neonate with Langerhans cell histiocytosis presenting as blueberry muffin rash: case report and review of the literature","volume":"8","author":"Cyr","year":"2020","journal-title":"SAGE Open Med Case Rep"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.2","DOI":"10.1001\/archpedi.155.7.778"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.3","DOI":"10.1002\/pbc.20362"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.4","DOI":"10.1002\/pbc.24367"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.5","DOI":"10.1001\/archderm.1973.01620170071020"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.6","DOI":"10.1136\/archdischild-2017-313970"},{"key":"2025082713260424000_14.12.e247213.7","doi-asserted-by":"crossref","first-page":"e140","DOI":"10.1111\/pde.13416","article-title":"Congenital Langerhans cell histiocytosis presenting in a 27-week-gestation neonate","volume":"35","author":"Dennin","year":"2018","journal-title":"Pediatr Dermatol"},{"key":"2025082713260424000_14.12.e247213.8","article-title":"Congenital self-healing Langerhans cell histiocytosis: clinical and pathological characteristics","volume":"12","author":"Pan","year":"2019","journal-title":"Int J Clin Exp Pathol"},{"key":"2025082713260424000_14.12.e247213.9","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1155\/2016\/4972180","article-title":"A fatal case of congenital Langerhans cell histiocytosis with disseminated cutaneous lesions in a premature neonate","volume":"2016","author":"Inoue","year":"2016","journal-title":"Case Rep Pediatr"},{"key":"2025082713260424000_14.12.e247213.10","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1590\/abd1806-4841.20175308","article-title":"Congenital Langerhans cell histiocytosis: a good prognosis disease?","volume":"92","author":"Frade","year":"2017","journal-title":"An Bras Dermatol"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.11","DOI":"10.1111\/j.1365-4362.1986.tb00867.x"},{"key":"2025082713260424000_14.12.e247213.12","doi-asserted-by":"crossref","first-page":"e155","DOI":"10.1097\/MPH.0000000000001428","article-title":"Langerhans cell histiocytosis presenting as enterocolitis and shock in neonate","volume":"41","author":"Bar\u00f3n Gonz\u00e1lez de Suso","year":"2019","journal-title":"J Pediatr Hematol Oncol"},{"doi-asserted-by":"publisher","key":"2025082713260424000_14.12.e247213.13","DOI":"10.1016\/S0190-9622(94)70258-6"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2021-247213","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T20:26:12Z","timestamp":1756326372000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2021-247213"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,12]]},"references-count":13,"journal-issue":{"issue":"12","published-online":{"date-parts":[[2021,12,8]]},"published-print":{"date-parts":[[2021,12]]}},"alternative-id":["10.1136\/bcr-2021-247213"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2021-247213","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2021,12]]}}}