Occult phosphaturic mesenchymal tumor of femur cortex causing oncogenic osteomalacia: Diagnostic challenges and clinical outcomes

Dutta, Deep; Pandey, Raghuvansh Kumar; Gogoi, Rakhee; Solanki, Nagender; Madan, Renu; Mondal, Anupam; Dogra, Shruti; Thapa, Pradeep

doi:10.5603/ep.a2018.0016

Cited by 7 publications

(11 citation statements)

References 18 publications

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“…This patient reported a 2-year history of debilitation with progression to walker use before low-energy pathologic femur fracture, incomplete contralateral femur fracture, and impending humerus fracture. The length-to-diagnosis of this patient is consistent with the literature, which reports 2.5 years to >20 years in some cases [ 10 , 23 , 27 ]. The patient’s prolonged symptoms were inaccurately attributed to diabetic neuropathy.…”

Section: Discussionsupporting

confidence: 91%

“…TIO causes fatigue, bone pain, weakness, or impending fractures secondary to severe hypophosphatemia and osteomalacia [ 5 , 9 ]. Tumor cells stimulate upregulation of fibroblast growth factor 23 (FGF-23), preventing formation of 1, 25-dihydroxyvitamin D3 (calcitriol), which inhibits reabsorption of phosphate in the kidneys [ 5 , 7 , 9 , 10 ]. PMTs have been reported in bone and soft tissue, including the thigh, foot, hip, head, neck, and spine [ 1 , 2 , 3 , 4 , 5 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 ].…”

Section: Introductionmentioning

confidence: 99%

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Phosphaturic Mesenchymal Tumor-induced Osteomalacia Masquerading as an Orthopedic Polytrauma Patient: A Case Report and Review of the Literature

Robles¹,

Rockov²,

Brien³

et al. 2022

JOCR

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Introduction: Phosphaturic mesenchymal tumor (PMT) is a rare benign tumor (500 cases to date) that can present in combination with a paraneoplastic syndrome called tumor-induced osteomalacia (TIO). To the best of our knowledge, it is the first case to date that presented as an orthopedic trauma patient. Case Report: This is a case of a 61-year-old male who initially presented as a polytrauma patient, but further investigation revealed a PMT causing TIO. This report describes his initial diagnosis and management from 2015 to 2021. Conclusion: TIO resultant of PMT may lead to severe bone pain, impending fractures, and delayed or misdiagnosis. This case demonstrates the importance of careful diagnosis and a team-based approach to managing PMT and its sequelae. Keywords: Pathological fracture, phosphaturic mesenchymal tumor, tumor-induced osteomalacia

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Section: Discussionsupporting

confidence: 91%

Section: Introductionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor-induced Osteomalacia Masquerading as an Orthopedic Polytrauma Patient: A Case Report and Review of the Literature

Robles¹,

Rockov²,

Brien³

et al. 2022

JOCR

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“…Fortunately, complete tumoral excision leads to rapid normalization of biochemical parameters and resolution of symptoms (14). Around 300 cases of TIO have been reported worldwide, however reports from the Indian subcontinent are limited (2, 15, 16, 17, 18, 19, 20, 21). Herein, we describe the clinical features, biochemical parameters, imaging modalities and the treatment outcome in 30 patients of TIO diagnosed at three tertiary care hospitals in India.…”

Section: Introductionmentioning

confidence: 99%

Tumor-induced osteomalacia: experience from three tertiary care centers in India

Pal

Bhadada

Singhare

et al. 2019

Endocrine Connections

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Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by recalcitrant hypophosphatemia. Reports from the Indian subcontinent are scarce, with most being single center experiences involving few patients. Herein, we conducted a retrospective analysis of 30 patients of TIO diagnosed at three tertiary care hospitals in India. Patients with persistent hypophosphatemia (despite correction of hypovitaminosis D), normocalcemia, elevated alkaline phosphatase, low TmP/GFR and elevated or ‘inappropriately normal’ FGF23 levels were labeled as having TIO. They were sequentially subjected to functional followed by anatomical imaging. Patients with a well-localized tumor underwent excision; others were put on phosphorous and calcitriol supplementation. The mean age at presentation was 39.6 years with female:male ratio of 3:2. Bone pain (83.3%) and proximal myopathy (70%) were the chief complaints; 40% of cases had fractures. The mean delay in diagnosis was 3.8 years. Tumors were clinically detectable in four patients (13.3%). The mean serum phosphate was 0.50 mmol/L with a median serum FGF23 level of 518 RU/mL. Somatostatin receptor-based scintigraphy was found to be superior to FDG-PET in tumor localization. Lower extremities were the most common site of the tumor (72%). Tumor size was positively correlated with serum FGF23 levels. Twenty-two patients underwent tumor resection and 16 of them had phosphaturic mesenchymal tumors. Surgical excision led to cure in 72.7% of patients whereas disease persistence and disease recurrence were seen in 18.2% and 9.1% of cases, respectively. At the last follow-up, serum phosphate in the surgically treated group was significantly higher than in the medically managed group.

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“…While TIO is most often caused by tumors in soft tissues in adults, some cases of bony lesions are reported, some of which occured in minors [4] , [27] , [28] , [29] , [30] .…”

Section: Discussion and Review Of The Literaturementioning

confidence: 99%

FGF-23 transmitted tumor – induced hypophosphatemic osteomalacia: A rare case of a young woman with recurrent fractures and review of the literature

Frank

Gerber

Cornelius

et al. 2022

Journal of Bone Oncology

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Highlights Tumor-transmitted osteomalacia is a rare FGF 23 transmitted phosphaturic disease. Delay of diagnosis is common due to small and clinically silent underlying tumors. The most sensitive imaging modality is 68 Ga-DOTA-TOC-PET/CT-scan. Complete resection of the tumor mass cures this paraneoplastic disease.

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Occult phosphaturic mesenchymal tumor of femur cortex causing oncogenic osteomalacia: Diagnostic challenges and clinical outcomes

Cited by 7 publications

References 18 publications

Phosphaturic Mesenchymal Tumor-induced Osteomalacia Masquerading as an Orthopedic Polytrauma Patient: A Case Report and Review of the Literature

Phosphaturic Mesenchymal Tumor-induced Osteomalacia Masquerading as an Orthopedic Polytrauma Patient: A Case Report and Review of the Literature

Tumor-induced osteomalacia: experience from three tertiary care centers in India

FGF-23 transmitted tumor – induced hypophosphatemic osteomalacia: A rare case of a young woman with recurrent fractures and review of the literature

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