{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,29]],"date-time":"2026-06-29T13:17:35Z","timestamp":1782739055793,"version":"3.54.5"},"reference-count":17,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,12,1]],"date-time":"2025-12-01T00:00:00Z","timestamp":1764547200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Langerhans cell histiocytosis (LCH) is a rare hematopoietic disorder with diverse clinical presentations and sequential or concurrent multi-organ involvement. The adult onset is particularly uncommon and challenging to diagnose early.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>A 36-year-old female patient initially presented with polydipsia, polyuria, and dry mouth, gradually developing bone pain, facial erythema, and lymph node enlargement. Cranial MRI showed pituitary stalk thickening with the absence of a posterior pituitary signal. Iliac bone biopsy with immunohistochemistry demonstrated S-100(+), CD1a(+), and CD68(+) expression. After Systemic Chemotherapy and DDAVP administration, follow-up showed improvement of polyuria, polydipsia, and polyphagia, remission of osseous pain, regression of cutaneous manifestations, normalized pituitary stalk morphology, and reduced lesion metabolic activity.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>This report documents a complex and rare case to improve early diagnosis, reduce misdiagnosis, and prevent poor outcomes from delayed treatment.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fmed.2025.1643331","type":"journal-article","created":{"date-parts":[[2025,12,1]],"date-time":"2025-12-01T05:17:05Z","timestamp":1764566225000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Adult onset multisystem Langerhans cell histiocytosis initially presenting with arginine vasopressin deficiency: a case report"],"prefix":"10.3389","volume":"12","author":[{"given":"Dan","family":"Zeng","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mingyu","family":"Hao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liming","family":"Zhou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuli","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junying","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shujuan","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Litao","family":"Zheng","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yeyu","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haiyan","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2025,12,1]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"1319","DOI":"10.1182\/blood.2019000934","article-title":"Langerhans cell histiocytosis","volume":"135","author":"Carlos Rodriguez-Galindo","year":"2020","journal-title":"Blood"},{"key":"ref2","doi-asserted-by":"publisher","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":"Goyal","year":"2022","journal-title":"Blood"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"728","DOI":"10.1111\/bjh.18459","article-title":"Incidence, prevalence and survival in patients with Langerhans cell histiocytosis: a national registry study from England, 2013\u20132019","volume":"199","author":"Liu","year":"2022","journal-title":"Br J Haematol"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1002\/hon.2857","article-title":"Langerhans cell histiocytosis: version 2021","volume":"39","author":"Gulati","year":"2021","journal-title":"Hematol Oncol"},{"key":"ref5","volume-title":"Langerhans cell histiocytosis","author":"Tillotson","year":"2025"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"3687","DOI":"10.1111\/cas.15879","article-title":"Clinical and prognostic features of Langerhans cell histiocytosis in adults","volume":"114","author":"Sato","year":"2023","journal-title":"Cancer Sci"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"102","DOI":"10.4103\/abr.abr_119_17","article-title":"Langerhans cell histiocytosis: a case report with unusual cutaneous manifestation","volume":"7","author":"Iraji","year":"2018","journal-title":"Adv Biomed Res"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"18678","DOI":"10.18632\/oncotarget.7892","article-title":"Langerhans cell histiocytosis in adults: a case report and review of the literature","volume":"7","author":"Lian","year":"2016","journal-title":"Oncotarget"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"13","DOI":"10.3389\/fendo.2022.868558","article-title":"Pituitary stalk thickening: causes and consequences. The children\u2019s memorial health institute experience and literature review","volume":"13","author":"Moszczy\u0144ska","year":"2022","journal-title":"Front Endocrinol"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"EDM22-0232","DOI":"10.1530\/EDM-22-0232","article-title":"Adult-onset Langerhans cell histiocytosis changing CNS lesion from pituitary to suprasellar extension","volume":"2022","author":"Kadowaki","year":"2022","journal-title":"Endocrinol Diabetes Metab Case Rep"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"662","DOI":"10.1016\/S2352-4642(21)00088-2","article-title":"Management of children and young people with idiopathic pituitary stalk thickening, central diabetes insipidus, or both: a national clinical practice consensus guideline","volume":"5","author":"Cerbone","year":"2021","journal-title":"Lancet Child Adolesc Health"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"95","DOI":"10.1530\/EJE-20-0058","article-title":"Central diabetes insipidus and pituitary stalk thickening in adults: distinction of neoplastic from non-neoplastic lesions","volume":"183","author":"Devuyst","year":"2020","journal-title":"Eur J Endocrinol"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"1689","DOI":"10.1007\/s00259-023-06122-6","article-title":"Comparison of whole-body DW-MRI with 2-[18F]FDG PET for staging and treatment monitoring of children with Langerhans cell histiocytosis","volume":"50","author":"Baratto","year":"2023","journal-title":"Eur J Nucl Med Mol Imaging"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"3707","DOI":"10.1111\/cas.13817","article-title":"Langerhans cell histiocytosis in adults: advances in pathophysiology and treatment","volume":"109","author":"Kobayashi","year":"2018","journal-title":"Cancer Sci"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"909","DOI":"10.3390\/jcm11040909","article-title":"Adult Langerhans cell histiocytosis and the skeleton","volume":"11","author":"Georgakopoulou","year":"2022","journal-title":"J Clin Med"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"1452003","DOI":"10.3389\/fmed.2024.1452003","article-title":"Clinical features and prognostic factors of pediatric Langerhans cell histiocytosis: a single-center retrospective study","volume":"11","author":"Lu","year":"2024","journal-title":"Front Med (Lausanne)"},{"key":"ref17","doi-asserted-by":"publisher","first-page":"6568","DOI":"10.1182\/bloodadvances.2023010706","article-title":"Long-term outcomes among adults with Langerhans cell histiocytosis","volume":"7","author":"Goyal","year":"2023","journal-title":"Blood Adv"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1643331\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,12,1]],"date-time":"2025-12-01T05:17:06Z","timestamp":1764566226000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1643331\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,1]]},"references-count":17,"alternative-id":["10.3389\/fmed.2025.1643331"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2025.1643331","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,1]]},"article-number":"1643331"}}