---
title: "Adult-Onset Langerhans Cell Histiocytosis: Two Cases with Calvarial and Hypothalamic Involvement"
id: "pubmed-42669941"
canonical_url: "https://medichelpline.com/clinical-feed/pubmed-42669941"
content_type: "clinical_feed_article"
specialty: "Critical Care"
source_name: "PubMed / NCBI"
source_url: "https://pubmed.ncbi.nlm.nih.gov/42669941/"
published_at: "2026-09-20T00:00:00.000Z"
evidence_level: "Case Reports"
license: "CC-BY-NC-4.0 / Informational Use"
---
# Adult-Onset Langerhans Cell Histiocytosis: Two Cases with Calvarial and Hypothalamic Involvement
## Provenance & Clinical Metadata
- **Canonical URL:** https://medichelpline.com/clinical-feed/pubmed-42669941
- **Specialty:** [Critical Care](https://medichelpline.com/clinical-feed/critical-care.md)
- **Primary Source:** PubMed / NCBI
- **Source URL:** [Original Journal Publication](https://pubmed.ncbi.nlm.nih.gov/42669941/)
- **Published At:** 2026-09-20T00:00:00.000Z
- **Evidence Rating:** Case Reports
## Executive GIST (TL;DR)
- Langerhans cell histiocytosis (**LCH**) is a rare clonal proliferation of bone marrow–derived dendritic cells that is uncommon in adults. Clinical features depend on the organs involved. - CNS involvement in adult LCH presents a major therapeutic challenge because it can lead to permanent neurological dysfunction. - Case 1: a 36-year-old woman presented with persistent headache. Neuroimaging showed a solitary osteolytic lesion in the left parietal bone with dural thickening. Surgical resection and histopathology confirmed LCH with positive **S-100** staining. Disease was limited to the calvarium; the patient received **bisphosphonates** and remained stable without systemic disease. - Case 2: a 26-year-old man presented with cognitive impairment, somnolence, polyuria, and endocrine dysfunction. MRI identified an enhancing hypothalamic lesion. Biopsy and immunohistochemistry showed LCH with **CD1a** and **S-100** positivity; **BRAF V600E** mutation was not detected by pyrosequencing. - The second patient received systemic chemotherapy with **cladribine**, which produced partial tumor regression but cognitive dysfunction persisted. - These contrasting cases demonstrate that lesion distribution, especially **CNS** involvement, determines therapeutic strategy and neurological outcomes. - The authors emphasize the importance of early diagnosis and comprehensive systemic evaluation because functional recovery may be limited even after cytoreductive therapy when neurological damage has occurred.
## Clinical Analysis & Structured Key Points
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Affiliations Expand ### Affiliation * 1 Department of Neurosurgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa 259-1193, Japan. masataka@tokai.ac.jp. * PMID: **42669941** Free article Item in Clipboard Case Reports # Adult-Onset Langerhans Cell Histiocytosis With Calvarial and Hypothalamic Involvement: Two Cases Chiaki Shinohara et al. Tokai J Exp Clin Med. 2026. Free article Show details Display options Display options Format Abstract PubMed PMID Tokai J Exp Clin Med Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Tokai+J+Exp+Clin+Med%22%5Bjour%5D&sort=date&sort_order=desc) * [ Search in NLM Catalog ](https://www.ncbi.nlm.nih.gov/nlmcatalog?term=%22Tokai+J+Exp+Clin+Med%22%5BTitle+Abbreviation%5D) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42669941/) . 2026 Sep 20;51(3):113-118. ### Authors [Chiaki Shinohara](https://pubmed.ncbi.nlm.nih.gov/?term=Shinohara+C&cauthor_id=42669941), [Takuya Yonemochi](https://pubmed.ncbi.nlm.nih.gov/?term=Yonemochi+T&cauthor_id=42669941), [Miyu Kikuchi](https://pubmed.ncbi.nlm.nih.gov/?term=Kikuchi+M&cauthor_id=42669941), [Naokazu Hayashi](https://pubmed.ncbi.nlm.nih.gov/?term=Hayashi+N&cauthor_id=42669941), [Kyoko Nozue](https://pubmed.ncbi.nlm.nih.gov/?term=Nozue+K&cauthor_id=42669941), [Chie Inomoto](https://pubmed.ncbi.nlm.nih.gov/?term=Inomoto+C&cauthor_id=42669941), [Masamichi Takahashi](https://pubmed.ncbi.nlm.nih.gov/?term=Takahashi+M&cauthor_id=42669941)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42669941/#short-view-affiliation-1 "Department of Neurosurgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa 259-1193, Japan. masataka@tokai.ac.jp.") ### Affiliation * 1 Department of Neurosurgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa 259-1193, Japan. masataka@tokai.ac.jp. * PMID: **42669941** Item in Clipboard Full text links Cite Display options Display options Format Abstract PubMed PMID ## Abstract Langerhans cell histiocytosis (LCH) is a rare clonal proliferative disorder of bone marrow-derived dendritic cells that is uncommon in adults. Its clinical presentation varies depending on the organ involved. Central nervous system (CNS) involvement poses a critical therapeutic challenge because of permanent neurological dysfunction. Here, we report two contrasting cases of adult-onset LCH with distinct clinical outcomes. Case 1 involved a 36-year-old woman presenting with a persistent headache. Neuroimaging revealed a solitary osteolytic lesion in the left parietal bone with dural thickening. Surgical resection was performed, and histopathological examination revealed characteristic Langerhans cells with positive S-100 immunostaining, confirming the diagnosis of LCH limited to the calvarium. The patient was treated with bisphosphonates and remained stable without systemic involvement. Case 2 involved a 26-year-old man presenting with cognitive impairment, somnolence, polyuria, and endocrine dysfunction. Magnetic resonance imaging revealed an enhancing hypothalamic lesion. Histopathological and immunohistochemical analyses confirmed LCH with CD1a and S-100 positivity. BRAF V600E mutation was not detected via pyrosequencing. Systemic chemotherapy with cladribine resulted in partial tumor regression; however, the cognitive dysfunction persisted. These contrasting cases highlight that disease distribution, particularly CNS involvement, influences therapeutic strategies and neurological outcomes in adult LCH. Our findings emphasize that early diagnosis and proactive systemic evaluation are essential as functional recovery remains limited once neurological damage occurs, even with successful cytoreductive therapy. [PubMed Disclaimer](https://pubmed.ncbi.nlm.nih.gov/disclaimer/) ## Similar articles * [ Successful treatment with cladribine of Erdheim-Chester disease with orbital and central nervous system involvement developing after treatment of Langerhans cell histiocytosis. ](https://pubmed.ncbi.nlm.nih.gov/26964390/) Perić P, Antić B, Knezević-Usaj S, Radić-Tasić O, Radovinović-Tasić S, Vasić-Vilić J, Sekulović L, Tarabar O, Tukić L, Jovandić S, Magić Z.Perić P, et al.Vojnosanit Pregl. 2016 Jan;73(1):83-7. doi: 10.2298/vsp140915037p.Vojnosanit Pregl. 2016.PMID: 26964390 * [ Isolated tumorous Langerhans cell histiocytosis of the brainstem: a diagnostic and therapeutic challenge. ](https://pubmed.ncbi.nlm.nih.gov/23848290/) Savardekar A, Tripathi M, Bansal D, Vaiphei K, Gupta SK.Savardekar A, et al.J Neurosurg Pediatr. 2013 Sep;12(3):258-61. doi: 10.3171/2013.6.PEDS13132. 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