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一名3岁患有不可切除的中央巨细胞病变患者的肿瘤性骨软化症

Tumor-induced Osteomalacia in a 3-Year-Old With Unresectable Central Giant Cell Lesions.

作者信息

Crossen Stephanie S, Zambrano Eduardo, Newman Beverley, Bernstein Jonathan A, Messner Anna H, Bachrach Laura K, Twist Clare J

机构信息

Divisions of *Pediatric Endocrinology and Diabetes §Medical Genetics ¶Pediatric Hematology and Oncology Departments of †Pathology ‡Radiology ∥Otolaryngology, Stanford University School of Medicine, Stanford, CA.

出版信息

J Pediatr Hematol Oncol. 2017 Jan;39(1):e21-e24. doi: 10.1097/MPH.0000000000000686.

DOI:10.1097/MPH.0000000000000686
PMID:27820122
Abstract

Tumor-induced osteomalacia (TIO) is a rare cause of hypophosphatemia involving overproduction of fibroblast growth factor 23. TIO has been described largely in adults with small mesenchymal tumors. We report a case of TIO in a child who presented with knee pain and radiographic findings concerning for rickets, and was found to have maxillomandibular giant cell lesions. The patient was treated with oral phosphorus and calcitriol, surgical debulking, and intralesional corticosteroids, which resulted in tumor regression and normalization of serum fibroblast growth factor 23 and phosphorus. This case illustrates the occurrence of this rare paraneoplastic syndrome in children and adds to our knowledge about clinical manifestations and pathologic findings associated with pediatric TIO.

摘要

肿瘤诱导的骨软化症(TIO)是低磷血症的一种罕见病因,涉及成纤维细胞生长因子23的过度产生。TIO主要在患有小间充质肿瘤的成人中被描述。我们报告了一例TIO患儿,该患儿表现为膝关节疼痛及与佝偻病相关的影像学表现,并被发现患有颌骨巨细胞病变。该患者接受了口服磷和骨化三醇、手术减瘤及病灶内注射皮质类固醇治疗,结果肿瘤消退,血清成纤维细胞生长因子23和磷恢复正常。本病例说明了这种罕见的副肿瘤综合征在儿童中的发生情况,并增加了我们对与儿童TIO相关的临床表现和病理发现的认识。

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引用本文的文献

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Front Endocrinol (Lausanne). 2024 Apr 19;15:1383681. doi: 10.3389/fendo.2024.1383681. eCollection 2024.
2
Tumor-induced Osteomalacia: A Case Report and Etiological Analysis with Literature Review.肿瘤相关性骨软化症:病例报告及文献复习并病因分析。
Orthop Surg. 2023 Dec;15(12):3342-3352. doi: 10.1111/os.13901. Epub 2023 Nov 7.
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Global guidance for the recognition, diagnosis, and management of tumor-induced osteomalacia.
肿瘤相关性骨软化症的识别、诊断和管理全球指南。
J Intern Med. 2023 Mar;293(3):309-328. doi: 10.1111/joim.13593. Epub 2022 Dec 13.
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Disorders of phosphate homeostasis in children, part 2: hypophosphatemic and hyperphosphatemic disorders.儿童磷代谢紊乱,第 2 部分:低磷血症和高磷血症性疾病。
Pediatr Radiol. 2022 Nov;52(12):2290-2305. doi: 10.1007/s00247-022-05373-z. Epub 2022 May 10.
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