CASE REPORT article
Front. Oncol.
Sec. Cancer Imaging and Image-directed Interventions
Case Report: A Rare Adult Multisystem Langerhans Cell Histiocytosis Involving the Skin, Thyroid, Jaw-Periodontal Region, and Bone: Diagnostic Challenges and Complementary PET/CT–SPECT/CT Findings
Longchuan Zhuang 1
Kexu Chen 2
Xiaokun Qi 2
Jie Li 2
Song Qin 1
Hua Yu 3
Minggang Yuan 2
1. Yantai Institute of Coastal Zone Research, Chinese Academy of Sciences (CAS), Yantai, China
2. Department of Nuclear Medicine, Qingdao Central Hospital of University of Health and Rehabilitation Sciences, Qingdao, China
3. Qingdao University Affiliated Qingdao Third People's Hospital, Qingdao, China
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Abstract
Langerhans cell histiocytosis (LCH) is a rare clonal myeloid neoplasm with prominent inflammatory features and heterogeneous manifestations in adults, often leading to diagnostic delay. We report a 32-year-old man who presented with a two-year history of recurrent ulcerative intertriginous skin lesions refractory to empirical treatment for presumed inflammatory or infectious dermatoses. Biopsy from the ulcer edge showed atypical histiocytoid cells with characteristic nuclear grooves and numerous eosinophils; immunohistochemistry was positive for CD1a, S-100, and Langerin with a Ki-67 index of ~40%, confirming cutaneous LCH. Baseline 18F-FDG PET/CT (August 2022) demonstrated multisystem disease with extensive cutaneous involvement and imaging-suggestive involvement of multiple extracutaneous sites, including multifocal osseous lesions, the thyroid, gastrointestinal tract, lymph nodes, lung (cystic lesions), and an intramuscular nodule. After six cycles of cytarabine-based induction chemotherapy, follow-up PET/CT (April 2023) showed near-complete metabolic remission with only residual low-grade uptake in the perineal skin and maxillomandibular/gingival region. In contrast, 99mTc-MDP whole-body bone scintigraphy with SPECT/CT performed one year later (July 2024) revealed persistent multifocal tracer uptake, most prominent in the craniofacial skeleton and long bones, with corresponding CT changes suggestive of ongoing remodeling. This case highlights the importance of early biopsy of persistent unexplained ulcerative intertriginous lesions in adults to expedite systemic staging, and demonstrates the complementary roles of serial 18F-FDG PET/CT and bone scintigraphy with SPECT/CT in longitudinal assessment of osseous disease in adult multisystem LCH.
Summary
Keywords
18F-FDG PET/CT, adult multisystem disease, bone scintigraphy with SPECT/CT, Cutaneous involvement, langerhans cell histiocytosis
Received
09 January 2026
Accepted
17 February 2026
Copyright
© 2026 Zhuang, Chen, Qi, Li, Qin, Yu and Yuan. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
*Correspondence: Hua Yu; Minggang Yuan
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