{"title":"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。","authors":"Amar Nitin Kanani, Benjamin Jacobs, Thillainayagam Muthukumar, Ramanan Rajakulasingham, Ruhaid Khurram","doi":"10.1007/s00256-026-05149-8","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":21783,"journal":{"name":"Skeletal Radiology","volume":" ","pages":"1415-1419"},"PeriodicalIF":2.2000,"publicationDate":"2026-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Phosphaturic mesenchymal tumour, connective tissue variant: a rare radiological presentation involving the radius mimicking fibrous dysplasia with oncogenic rickets.\",\"authors\":\"Amar Nitin Kanani, Benjamin Jacobs, Thillainayagam Muthukumar, Ramanan Rajakulasingham, Ruhaid Khurram\",\"doi\":\"10.1007/s00256-026-05149-8\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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.</p>\",\"PeriodicalId\":21783,\"journal\":{\"name\":\"Skeletal Radiology\",\"volume\":\" \",\"pages\":\"1415-1419\"},\"PeriodicalIF\":2.2000,\"publicationDate\":\"2026-06-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Skeletal Radiology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1007/s00256-026-05149-8\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2026/2/2 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q2\",\"JCRName\":\"ORTHOPEDICS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Skeletal Radiology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00256-026-05149-8","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2026/2/2 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"ORTHOPEDICS","Score":null,"Total":0}
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.
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.