磷酸盐间充质瘤,结缔组织变异:一种罕见的放射学表现,涉及桡骨模拟纤维发育不良伴致癌性佝偻病。

IF 2.2 3区 医学 Q2 ORTHOPEDICS
Skeletal Radiology Pub Date : 2026-06-01 Epub Date: 2026-02-02 DOI:10.1007/s00256-026-05149-8
Amar Nitin Kanani, Benjamin Jacobs, Thillainayagam Muthukumar, Ramanan Rajakulasingham, Ruhaid Khurram
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引用次数: 0

摘要

磷化间充质肿瘤(pmt)是一种罕见的肿瘤,其分泌成纤维细胞生长因子-23 (FGF-23),引起肿瘤诱导的骨软化症(TIO)。组织学重叠与幼年沙砾样骨化纤维瘤(JPOF)可导致诊断困难,特别是颅面骨骼外。一名四岁女孩表现为前臂肿胀疼痛。x线片显示桡骨有纤维骨性病变,活检最初无法诊断,因此推定为纤维发育不良。在接下来的6年里,病变扩大,在10岁时,患者出现膝外翻,并伴有低磷血症佝偻病的生化证据(低磷酸盐,碱性磷酸酶升高,FGF-23升高)。药物治疗纠正了佝偻病,尽管畸形需要引导生长手术。13岁时,对扩大的病变进行重复活检,发现有梭形间质和沙粒样小骨的纤维骨性肿瘤,GNAS突变阴性,与结缔组织变异的磷化间充质肿瘤一致。本病例表现为罕见的累及长骨的PMT影像学表现,其影像学特征与纤维发育不良非常相似,组织学上与JPOF有重叠。影像学表现的快速进展、不一致的代谢异常和FGF-23水平的显著升高给诊断带来了挑战。因此,低磷血症佝偻病的四肢纤维性发育不良样病变应提示考虑PMT。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Phosphaturic mesenchymal tumour, connective tissue variant: a rare radiological presentation involving the radius mimicking fibrous dysplasia with oncogenic rickets.

Phosphaturic mesenchymal tumours (PMTs) are rare neoplasms that secrete fibroblast growth factor-23 (FGF-23), causing tumour-induced osteomalacia (TIO). Histological overlap with juvenile psammomatoid ossifying fibroma (JPOF) can lead to diagnostic difficulty, particularly outside the craniofacial skeleton. A 4-year-old girl presented with a painful forearm swelling. Radiographs demonstrated a fibro-osseous lesion in the radius and biopsy was initially non-diagnostic, leading to a presumptive diagnosis of fibrous dysplasia. Over the next 6 years, the lesion enlarged, and at age 10, the patient developed genu valgum with biochemical evidence of hypophosphataemic rickets (low phosphate, raised alkaline phosphatase, elevated FGF-23). Medical therapy corrected the rickets, though deformity required guided growth surgery. At age 13, a repeat biopsy of the enlarging lesion revealed a fibro-osseous tumour with spindle stroma and psammomatoid ossicles, negative for GNAS mutation, and consistent with a phosphaturic mesenchymal tumour, connective tissue variant. This case highlights a rare radiologic presentation of PMT involving a long bone, with imaging features closely mimicking fibrous dysplasia and histological overlap with JPOF. The rapid progression of imaging findings, discordant metabolic abnormalities and markedly elevated FGF-23 levels poses a diagnostic challenge. As such, fibrous dysplasia-like lesions of the extremities in the setting of hypophosphataemic rickets should prompt consideration of PMT.

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来源期刊
Skeletal Radiology
Skeletal Radiology 医学-核医学
CiteScore
4.40
自引率
9.50%
发文量
253
审稿时长
3-8 weeks
期刊介绍: Skeletal Radiology provides a forum for the dissemination of current knowledge and information dealing with disorders of the musculoskeletal system including the spine. While emphasizing the radiological aspects of the many varied skeletal abnormalities, the journal also adopts an interdisciplinary approach, reflecting the membership of the International Skeletal Society. Thus, the anatomical, pathological, physiological, clinical, metabolic and epidemiological aspects of the many entities affecting the skeleton receive appropriate consideration. This is the Journal of the International Skeletal Society and the Official Journal of the Society of Skeletal Radiology and the Australasian Musculoskelelal Imaging Group.
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