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
文献类型：期刊论文
| 作者 | Zhuang, Longchuan2,3,4,5; Chen, Kexu4; Qi, Xiaokun4; Li, Jie4; Qin, Song2,5; Yu, Hua1; Yuan, Minggang4 |
| 刊名 | FRONTIERS IN ONCOLOGY
 |
| 出版日期 | 2026-02-27 |
| 卷号 | 16页码:7 |
| 关键词 | F-18-FDG PET/CT adult multisystem disease bone scintigraphy with SPECT/CT cutaneous involvement Langerhans cell histiocytosis |
| ISSN号 | 2234-943X |
| DOI | 10.3389/fonc.2026.1784088 |
| 通讯作者 | Yu, Hua(qdyhua279@163.com) ; Yuan, Minggang(13314613@163.com) |
| 英文摘要 | 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 similar to 40%, confirming cutaneous LCH. Baseline F-18-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, Tc-99m-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 F-18-FDG PET/CT and bone scintigraphy with SPECT/CT in longitudinal assessment of osseous disease in adult multisystem LCH. |
| WOS研究方向 | Oncology |
| 语种 | 英语 |
| WOS记录号 | WOS:001713431500001 |
| 资助机构 | Qingdao Medical and Health Research Project ; Seed Project of Yantai Institute of Coastal Zone Research, Chinese Academy of Sciences ; Yantai Science and Technology Innovation Development Program - Key Basic Research Project |
| 源URL | [http://ir.yic.ac.cn/handle/133337/42302]  |
| 专题 | 海岸带生物学与生物资源利用重点实验室 |
| 通讯作者 | Yu, Hua; Yuan, Minggang |
| 作者单位 | 1.Qingdao Univ, Affiliated Qingdao Peoples Hosp 3, Qingdao, Shandong, Peoples R China 2.Shandong Key Lab Coastal Zone Environm Proc & Ecol, Yantai, Shandong, Peoples R China 3.Yantai Key Lab Nucl Safety Marine Ecol Environm, Yantai, Shandong, Peoples R China 4.Univ Hlth & Rehabil Sci, Qingdao Cent Hosp, Dept Gen Med, Qingdao, Shandong, Peoples R China 5.Chinese Acad Sci, Yantai Inst Coastal Zone Res, Yantai, Shandong, Peoples R China |
| 推荐引用方式 GB/T 7714 | Zhuang, Longchuan,Chen, Kexu,Qi, Xiaokun,et al. 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[J]. FRONTIERS IN ONCOLOGY,2026,16:7. |
| APA | Zhuang, Longchuan.,Chen, Kexu.,Qi, Xiaokun.,Li, Jie.,Qin, Song.,...&Yuan, Minggang.(2026).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.FRONTIERS IN ONCOLOGY,16,7. |
| MLA | Zhuang, Longchuan,et al."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".FRONTIERS IN ONCOLOGY 16(2026):7. |
入库方式： OAI收割
来源：烟台海岸带研究所
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