{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T20:25:46Z","timestamp":1787775946104,"version":"build-2784847793"},"reference-count":13,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"1","content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2018,1]]},"abstract":"<jats:p>Langerhans cell histiocytosis (LCH) is characterized by idiopathic monoclonal infiltration of Langerhans cells in different organs such as the skeleton, skin, pituitary gland, liver, spleen, lungs, and the hematopoietic system. Skin lesions are common in LCH and affect about 40% of cases. It is reported that skin lesions are usually the first manifestation of LCH in 80% of patients. Usually, cutaneous presentations of LCH in adults are generalized or seborrhea-like lesions and it is often the first manifestation of disease. Here, we describe a 45-year old female who was known case of hypothyroidism, systemic lupus erythematosus, and diabetes insipidus. In our patient, cutaneous involvement was unusual. It was single and presented in unusual site (ankle) and before developing such lesion, she had diabetes insipidus for several years due to the involvement of pituitary gland.<\/jats:p>","DOI":"10.4103\/abr.abr_119_17","type":"journal-article","created":{"date-parts":[[2018,6,25]],"date-time":"2018-06-25T06:15:25Z","timestamp":1529907325000},"update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":11,"title":["Langerhans Cell Histiocytosis: A Case Report with Unusual Cutaneous Manifestation"],"prefix":"10.4103","volume":"7","author":[{"given":"Fariba","family":"Iraji","sequence":"first","affiliation":[{"name":"From the Department of Dermatology, Skin Diseases and Leishmaniasis Research Center, Isfahan University of Medical Sciences, Isfahan, Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nazila","family":"Poostiyan","sequence":"additional","affiliation":[{"name":"From the Department of Dermatology, Skin Diseases and Leishmaniasis Research Center, Isfahan University of Medical Sciences, Isfahan, Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Parvin Rajabi","family":"Dehnavi","sequence":"additional","affiliation":[{"name":"Department of Pathology, Isfahan University of Medical Sciences, Isfahan, Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mojgan","family":"Soghrati","sequence":"additional","affiliation":[{"name":"Alzahra Research Institute, Isfahan, Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2018,6,25]]},"reference":[{"key":"R1-20251008","doi-asserted-by":"crossref","first-page":"225","DOI":"10.5114\/pdia.2014.40970","article-title":"Adult onset of multisystem Langerhans cell histiocytosis with skin and lymph node involvement","volume":"32","author":"Blazewicz","year":"2015","journal-title":"Postepy Dermatol Alergol"},{"key":"R2-20251008","doi-asserted-by":"crossref","first-page":"25","DOI":"10.5826\/dpc.0201a04","article-title":"Langerhans cell histiocytosis - A case report","volume":"2","author":"Jeunon","year":"2012","journal-title":"Dermatol Pract Concept"},{"key":"R3-20251008","doi-asserted-by":"crossref","first-page":"560","DOI":"10.4103\/0378-6323.144206","article-title":"Adult onset Langerhans cell histiocytosis: Report of two patients","volume":"80","author":"Reddy","year":"2014","journal-title":"Indian J Dermatol Venereol Leprol"},{"key":"R4-20251008","doi-asserted-by":"crossref","first-page":"354","DOI":"10.1016\/j.ctrv.2010.02.012","article-title":"Langerhans cell histiocytosis: Current concepts and treatments","volume":"36","author":"Abla","year":"2010","journal-title":"Cancer Treat Rev"},{"key":"R5-20251008","first-page":"95","article-title":"Langerhans Cell Histiocytosis in an Adult - a rare, life-threatening and not to be missed","volume":"69","author":"Lai","year":"2014","journal-title":"Med J Malaysia"},{"key":"R6-20251008","doi-asserted-by":"crossref","first-page":"219","DOI":"10.1620\/tjem.230.219","article-title":"The adverse prognostic hallmarks in identical twins with Langerhans cell histiocytosis: A clinical report and literature review","volume":"230","author":"Chai","year":"2013","journal-title":"Tohoku J Exp Med"},{"key":"R7-20251008","first-page":"27","article-title":"Langerhans cell histiocytosis mimicking osteomyelitis in an infant","volume":"4","author":"Erdem","year":"2013","journal-title":"APSP J Case Rep"},{"key":"R8-20251008","first-page":"627","article-title":"Langerhans cell histiocytosis of maxillary sinus","volume":"67","author":"Azreen","year":"2012","journal-title":"Med J Malaysia"},{"key":"R9-20251008","doi-asserted-by":"crossref","first-page":"363","DOI":"10.1111\/j.1600-0609.2006.00648.x","article-title":"Adult Langerhans cell histiocytosis","volume":"76","author":"Stockschlaeder","year":"2006","journal-title":"Eur J Haematol"},{"key":"R11-20251008","doi-asserted-by":"crossref","first-page":"728","DOI":"10.1067\/mpd.2001.111331","article-title":"A randomized trial of treatment for multisystem Langerhans' cell histiocytosis","volume":"138","author":"Gadner","year":"2001","journal-title":"J Pediatr"},{"key":"R12-20251008","doi-asserted-by":"crossref","first-page":"262","DOI":"10.5021\/ad.2016.28.2.262","article-title":"A case of adult generalized cutaneous langerhans cell histiocytosis","volume":"28","author":"Sun","year":"2016","journal-title":"Ann Dermatol"},{"key":"R13-20251008","doi-asserted-by":"crossref","first-page":"765","DOI":"10.1111\/j.1365-2230.2007.02467.x","article-title":"Langerhans' cell histiocytosis with multisystem involvement in an adult","volume":"32","author":"Liu","year":"2007","journal-title":"Clin Exp Dermatol"},{"key":"R14-20251008","doi-asserted-by":"crossref","first-page":"424","DOI":"10.4103\/0019-5154.160545","article-title":"An unusual case of neonatal langehrans cell histiocytosis presenting with diffuse hemorrhagic nodules and a cutaneous abdominal mass","volume":"60","author":"Wong","year":"2015","journal-title":"Indian J Dermatol"}],"container-title":["Advanced Biomedical Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/01679891-201807000-00102","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T19:46:07Z","timestamp":1787773567000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/01679891-201807000-00102"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,1]]},"references-count":13,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2018]]}},"URL":"https:\/\/doi.org\/10.4103\/abr.abr_119_17","relation":{},"ISSN":["2277-9175"],"issn-type":[{"value":"2277-9175","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,1]]},"assertion":[{"value":"2017-07-01","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2017-08-01","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}