Oncogenic osteomalacia due to phosphaturic mesenchymal tumour in the upper thoracic spine

Garg, Bhavuk; Mehta, Nishank; Goyal, Archit; Khadgawat, Rajesh

doi:10.1136/bcr-2020-238209

Cited by 4 publications

(5 citation statements)

References 32 publications

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“…Cases of PMT were reviewed and summarized in 2020 by Garg et al. ( 5 ). Since its discovery in 1947, only 21 cases of PMT have been reported involving the spinal column, of which only six were PMT involving the thoracic spine.…”

Section: Discussionmentioning

confidence: 99%

“…In soft tissues, PMT most often involves the extremities, while bone tumors usually involve the appendicular bones, skull, and sinuses (1). Cases of PMT were reviewed and summarized in 2020 by Garg et al (5). Since its discovery in 1947, only 21 cases of PMT have been reported involving the spinal column, of which only six were PMT involving the thoracic spine.…”

Section: Discussionmentioning

confidence: 99%

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Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Zhang,

Hu,

Guo

et al. 2024

Front. Endocrinol.

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Phosphaturic mesenchymal tumors (PMT) are rare and distinctive tumors that typically result in paraneoplastic syndrome known as tumor-induced osteomalacia (TIO). We report a case of bilateral osteoporotic femoral neck fracture caused by PMT. PMT was surgically resected, followed by sequential treatment of bilateral femoral neck fractures with total hip arthroplasty (THA). A 49-year-old perimenopausal woman experienced consistent bone pain with limb weakness persisting for over 2 years. Initially, she was diagnosed with early osteonecrosis of the femoral head and received nonsurgical treatment. However, from 2020 to 2022, her pain extended to the bilateral shoulders and knees with increased intensity. She had no positive family history or any other genetic diseases, and her menstrual cycles were regular. Physical examination revealed tenderness at the midpoints of the bilateral groin and restricted bilateral hip range of motion, with grade 3/5 muscle strength in both lower extremities. Laboratory findings revealed moderate anemia (hemoglobin 66 g/L), leukopenia (2.70 × 109/L), neutropenia (1.28 × 109/L), hypophosphatemia (0.36 mmol/L), high alkaline phosphatase activity (308.00 U/L), and normal serum calcium (2.22 mmol/L). After surgery, additional examinations were performed to explore the cause of hypophosphatemic osteomalacia. After definitive diagnosis, the patient underwent tumor resection via T11 laminectomy on August 6, 2022. Six months after the second THA, the patient regained normal gait with satisfactory hip movement function without recurrence of PMT-associated osteomalacia or prosthesis loosening. By providing detailed clinical data and a diagnostic and treatment approach, we aimed to improve the clinical understanding of femoral neck fractures caused by TIO.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Zhang,

Hu,

Guo

et al. 2024

Front. Endocrinol.

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“…Garg et al in 2020 highlighted that there have been only 21 cases of TIO-associated mesenchymal tumors involving the spinal column reported in the 70 years since these tumors were first described, of which only six were PMTs involving the thoracic spine. All of these were treated surgically; four without recurrence at follow-up, one with recurrence of serum abnormalities at follow-up, and one with no follow-up documented ( 7 ).…”

Section: Discussionmentioning

confidence: 99%

Tumor-induced osteomalacia treated with T12 tumor resection

Mancini

Sabet

Nielsen

et al. 2022

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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Summary Tumor-induced osteomalacia (TIO) is a rare form of osteomalacia caused by fibroblast growth factor-23 (FGF23)-secreting tumors. Most of these tumors are phosphaturic mesenchymal tumors (PMTs) typically involving soft tissue in the extremities and bone of the appendicular skeleton and cranium. We report the case of a 60-year-old woman with about 3 years of persistent bone pain and multiple fractures, initially diagnosed as osteoporosis, who was found to have hypophosphatemia with low 1,25-dihydroxyvitamin D and elevated alkaline phosphatase and inappropriately normal FGF23 consistent with TIO. Her symptoms improved with phosphate supplementation, vitamin D and calcitriol. 68Ga-DOTATATE imaging revealed a T12 vertebral body lesion confirmed on biopsy to be a PMT. She underwent resection of the PMT with resolution of TIO and increased bone density. This rare case of TIO secondary to a PMT of the thoracic spine highlights some of the common features of PMT-associated TIO and draws attention to PMT-associated TIO as a possible cause of unexplained persistent bone pain, a disease entity that often goes undiagnosed and untreated for years. Learning points Tumor-induced osteomalacia (TIO) is typically caused by phosphaturic mesenchymal tumors (PMTs) that are usually found in the soft tissue of the extremities and bone of the appendicular skeleton/cranium and rarely in the spine. TIO may be misdiagnosed as osteoporosis or spondyloarthritis, and the correct diagnosis is often delayed for years. However, osteoporosis, in the absence of fracture, is not associated with bone pain. The hallmark of TIO is hypophosphatemia with inappropriately normal or low 1,25-dihydroxyvitamin D and elevated or inappropriately normal fibroblast growth factor-23 (FGF23) levels. In patients with unexplained persistent bone pain, a serum phosphate should be measured. Consider PMT-associated TIO as a potential cause of unexplained persistent bone pain and hypophosphatemia. PMTs express somatostatin receptors and may be identified with 68Ga-DOTATATE imaging. Complete surgical resection is the preferred treatment for spinal PMTs associated with TIO.

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“…TIO is commonly located in the craniofacial regions and limbs [1]. Only 20 spine tumor cases have been reported in the literature [4]. The refractory rate of TIO overall was reported to be 18.3% but highest in spine tumors (77.8%) [5].…”

Section: Discussionmentioning

confidence: 99%

Spinal Tumor Resection with Intraoperative Computed Tomography Navigation in Tumor-Induced Osteomalacia: A Case Report

Endo¹,

Arima²,

Hasegawa³

et al. 2021

SCR

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This is the first report using intraoperative CT navigation for Tumor-induced Osteomalacia (TIO) lesions of the spine. TIO is a rare paraneoplastic disorder caused by tumors secret Fibroblast growth factor 23 secreted by tumor tissue. Surgical resection of the main tumor is the only definitive treatment. However, the tumor is usually small, and it hard to find it, and it tends to be seen when the recurrence is caused by the difficulty of whole tumor resection. A 57-year-old woman presented with a rare case of TIO located in T10 vertebra body. Using the intraoperative CT navigation, we resected the tumor in the T10 vertebral body very effective for accurate localization of the tumor and helpful for guidance of resection area and confirmation for excision of tumor. As a treatment for TIO, we report a tumor resection with intraoperative computed tomography navigation that made it possible to resect tumor in T10 vertebrae precisely and safely, which is small and difficult and dangerous access.

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Oncogenic osteomalacia due to phosphaturic mesenchymal tumour in the upper thoracic spine

Cited by 4 publications

References 32 publications

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Tumor-induced osteomalacia treated with T12 tumor resection

Spinal Tumor Resection with Intraoperative Computed Tomography Navigation in Tumor-Induced Osteomalacia: A Case Report

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