{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T07:04:48Z","timestamp":1780383888160,"version":"3.54.1"},"reference-count":20,"publisher":"Wiley","license":[{"start":{"date-parts":[[2022,12,5]],"date-time":"2022-12-05T00:00:00Z","timestamp":1670198400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Case Reports in Endocrinology"],"published-print":{"date-parts":[[2022,12,5]]},"abstract":"<jats:p>Background\/Objective. We present a case of Langerhans cell histiocytosis (LCH) with gastrointestinal involvement masquerading as inflammatory bowel disease (IBD) in a patient who initially had features of central diabetes insipidus (CDI). Case Report. A 19-year-old male presented at 14 years of age with central diabetes insipidus. He subsequently developed panhypopituitarism and sellar-suprasellar mass, the biopsy of which was inconclusive. Secondary causes for hypophysitis were ruled out. Five years later, he developed perianal pus discharging sinuses, positive ASCA, and sacroiliitis. Rectal ulcer biopsy showed nonspecific inflammation and necrosis. Hence, he was managed as inflammatory bowel disease (IBD). Due to nonresponsiveness of symptoms, doubt about diagnosis was invoked and rectal ulcer biopsy was repeated, which then showed infiltration by Langerhans cells. Hence, he was diagnosed with LCH and showed resolution of symptoms on initiating steroids and vinblastine. Discussion. Gastrointestinal involvement by LCH is unusual and only rarely has represented a prominent clinical manifestation. In most cases, such involvement suggests widespread multisystem disease. Its distinctive morphologic and immunohistochemical features allow LCH to be distinguished from other inflammatory infiltrations found in mucosal biopsy specimens. Conclusion. Preceding CDI and hypopituitarism may predict LCH in patients with IBD-like diseases.<\/jats:p>","DOI":"10.1155\/2022\/4672473","type":"journal-article","created":{"date-parts":[[2022,12,5]],"date-time":"2022-12-05T21:35:14Z","timestamp":1670276114000},"page":"1-5","source":"Crossref","is-referenced-by-count":1,"title":["Multifocal Multisystem Langerhans Cell Histiocytosis Involving Pituitary Masquerading as Crohn\u2019s Disease: A Case Report and Review of the Literature"],"prefix":"10.1155","volume":"2022","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7649-6749","authenticated-orcid":true,"given":"Mohd. Ashraf","family":"Ganie","sequence":"first","affiliation":[{"name":"Department of Endocrinology, Sher-I-Kasmir Institute of Medical Sciences, Srinagar, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4070-7548","authenticated-orcid":true,"given":"Bhanu","family":"Malhotra","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, PGIMER, Chandigarh 160012, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manpreet","family":"Saini","sequence":"additional","affiliation":[{"name":"Baba Farid University, Faridkot, Punjab, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Arshiya","family":"Dutta","sequence":"additional","affiliation":[{"name":"MMC and Hospital, Solan, Himachal Pradesh, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atul","family":"Sharma","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Sher-I-Kasmir Institute of Medical Sciences, Srinagar, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kim","family":"Vaiphie","sequence":"additional","affiliation":[{"name":"Department of Histopathology, PGIMER, Chandigarh 160012, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5415-1611","authenticated-orcid":true,"given":"Pinaki","family":"Dutta","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, PGIMER, Chandigarh 160012, India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","reference":[{"issue":"17","key":"1","doi-asserted-by":"crossref","first-page":"2601","DOI":"10.1182\/blood.2021014343","article-title":"International expert consensus recommendations for the diagnosis and treatment of Langerhans cell histiocytosis in adults","volume":"139","author":"G. Goyal","year":"2022","journal-title":"Blood"},{"key":"2","doi-asserted-by":"publisher","DOI":"10.1159\/000517040"},{"key":"3","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20760"},{"key":"4","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2003.12.030"},{"key":"5","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.22663"},{"key":"6","doi-asserted-by":"publisher","DOI":"10.3748\/wjg.v13.i16.2312"},{"key":"7","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2020.03.022"},{"key":"8","doi-asserted-by":"publisher","DOI":"10.1111\/apa.15590"},{"key":"9","doi-asserted-by":"publisher","DOI":"10.5152\/balkanmedj.2016.160492"},{"key":"10","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2020.00350"},{"key":"11","article-title":"Unusual Presentation of Central Diabetes Insipidus in a Patient with Crohn\u2019s disease. Case reports in classical and unusual causes of hypopituitarism II","volume-title":"Neuroendocrinology and Pituitary MON-236","author":"C. S. Johns","year":"2020"},{"key":"12","doi-asserted-by":"publisher","DOI":"10.1002\/(sici)1096-911x(199612)27:6<540::aid-mpo6>3.0.co;2-l"},{"key":"13","doi-asserted-by":"crossref","first-page":"279","DOI":"10.1016\/j.anpedi.2015.04.014","article-title":"Histiocitosis de c\u00e9lulas de Langerhans con afectaci\u00f3n gastrointestinal","volume":"83","author":"M. Andi\u00f3n Catal\u00e1n","year":"2015","journal-title":"Anales de Pediat\u00eda"},{"issue":"2","key":"14","doi-asserted-by":"crossref","first-page":"228","DOI":"10.1002\/pbc.20425","article-title":"Risk factors for diabetes insipidus in langerhans cell histiocytosis","volume":"46","author":"N. Grois","year":"2006","journal-title":"Pediatric Blood and Cancer"},{"issue":"4","key":"15","doi-asserted-by":"crossref","first-page":"1370","DOI":"10.1210\/jcem.85.4.6501","article-title":"Hypothalamo-pituitary abnormalities in adult patients with langerhans cell histiocytosis: clinical, endocrinological, and radiological features and response to treatment","volume":"85","author":"G. A. Kaltsas","year":"2000","journal-title":"The Journal of Cinical Endocrinology and Metabolism"},{"key":"16","doi-asserted-by":"crossref","first-page":"248","DOI":"10.1002\/mpo.2950240407","article-title":"Diabetes insipidus in Langerhans cell histiocytosis: results from the DAL-HX 83 study.","volume":"24","author":"N. Grois","year":"1995","journal-title":"Medical and Pediatric Oncology"},{"key":"17","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1016\/j.jpeds.2021.06.016","article-title":"additive prognostic impact of gastrointestinal involvement in severe multisystem langerhans cell histiocytosis","volume":"237","author":"M. Minkov","year":"2021","journal-title":"Jornal de Pediatria"},{"key":"18","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2006-0708"},{"key":"19","doi-asserted-by":"crossref","first-page":"305","DOI":"10.1097\/PAS.0b013e31820654e4","article-title":"Gastrointestinal tract Langerhans cell histiocytosis: a clinicopathologic study of 12 patients","volume":"35","author":"A. D. Singhi","year":"2011","journal-title":"The American Journal of Surgical Pathology"},{"key":"20","doi-asserted-by":"crossref","first-page":"9","DOI":"10.1002\/(SICI)1096-911X(199701)28:1<9::AID-MPO3>3.0.CO;2-P","article-title":"Langerhans'-cell histiocytosis in adults","volume":"28","author":"I. Baumgartner","year":"1997","journal-title":"Medical and Pediatric Oncology"}],"container-title":["Case Reports in Endocrinology"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/downloads.hindawi.com\/journals\/crie\/2022\/4672473.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/downloads.hindawi.com\/journals\/crie\/2022\/4672473.xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/downloads.hindawi.com\/journals\/crie\/2022\/4672473.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,12,5]],"date-time":"2022-12-05T21:35:23Z","timestamp":1670276123000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.hindawi.com\/journals\/crie\/2022\/4672473\/"}},"subtitle":[],"editor":[{"given":"Osamu","family":"Isozaki","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2022,12,5]]},"references-count":20,"alternative-id":["4672473","4672473"],"URL":"https:\/\/doi.org\/10.1155\/2022\/4672473","relation":{},"ISSN":["2090-651X","2090-6501"],"issn-type":[{"value":"2090-651X","type":"electronic"},{"value":"2090-6501","type":"print"}],"subject":[],"published":{"date-parts":[[2022,12,5]]}}}