Oncogenic Osteomalacia Caused by Phosphaturie Mesenchymal Tumours in the Proximal and Shaft of the Tibia: A Case Report

Pithankuakul, Kongkit; Ratanasuwan, Tanaporn; Thanakit, Voranuch; Sukhantanak, Bhasanan; Kiatisevi, Piya

doi:10.1177/230949901402200231

Cited by 6 publications

(3 citation statements)

References 27 publications

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“…OO is a rare clinicopathological type of acquired osteomalacia secondary to PMT. Classically, the hypophosphatemia caused by PMT is corrected after surgical removal [60]. There is a small percentage of tumors within the histological spectrum of PMT that do not demonstrate phosphate diuresis and have been referred to as PMT non-phosphaturic variant [32,37,61,62].…”

Section: Discussionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Qari

Hamao-Sakamoto

Fuselier

et al. 2015

Head and Neck Pathol

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Phosphaturic mesenchymal tumor (PMT) is a rare neoplasm that secretes fibroblast growth factor-23 (FGF-23) and causes oncogenic osteomalacia. It occurs in adults with equal gender distribution and the most common location is the lower extremities, followed by the head and neck. Besides osteomalacia, the clinical presentation includes bone pain and multiple bone fractures. Microscopic features consist of spindle cells, multinucleated giant cells, and calcifications embedded in a chondromyxoid matrix. Laboratory findings indicate normal calcium and parathyroid levels, hypophosphatemia, and increased levels of FGF-23 that usually revert to normal after surgical removal. Due to its rarity, the purpose of the study was to report 2 new oral cases of PMT and to review the literature in the head and neck. The first case occurred in the gingiva and had been present for 6 years. The second case was a recurrence of a previously diagnosed PMT in the right mandible that metastasized to the lung and soft tissue. The literature review included 53 cases in the head and neck. There was a predilection for extra-oral sites (76 %) compared to intra-oral sites (24 %) with paranasal sinuses considered the most common location (38 %) followed by the mandible (15 %). There were 9 recurrences that included 3 malignant cases indicating a potentially aggressive tumor. Due to the indeterminate biological behavior of PMT and its rarity, a comprehensive evaluation of medical, laboratory, radiographic, and histological findings are crucial for a definitive diagnosis and treatment.

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Section: Discussionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Qari

Hamao-Sakamoto

Fuselier

et al. 2015

Head and Neck Pathol

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“…It secretes phosphaturic hormones, among which fibroblast growth factor (FGF)-23 is the main one. [ 2 ] FGF-23 reduces renal phosphate absorption by suppressing the expression of sodium phosphate cotransporter in the brush border membrane of proximal tubules. It also reduces intestinal phosphate absorption by decreasing the production of 1,25 hydroxyvitamin D in the kidneys, which mediates intestinal absorption of phosphate.…”

Section: Discussionmentioning

confidence: 99%

Perioperative management of patients with severe hypophosphataemia secondary to oncogenic osteomalacia: Our experience and review of literature

2017

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Oncogenic osteomalacia (OOM) is a rare paraneoplastic syndrome associated with mesenchymal tumours. It is characterised by phosphaturia, hypophosphataemia, decreased serum Vitamin D3 levels and severe osteomalacia. OOM-inducing tumours are usually benign, arising either from bone or soft tissue, with extremities and craniofacial region being the most common sites. Surgical resection of the tumour remains the mainstay of treatment. Challenges to an anaesthesiologist arise when such patients are planned for surgical resection of the underlying tumour. All the perioperative dilemmas are directly related to the severe hypophosphataemia. We describe three such cases of OOM and their perioperative management.

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“…In our case, biochemical normalization did not happen, despite complete surgical excision of the metacarpal lesion after the second surgery. Although multifocality is extremely rare and has only previously been described in four patients (7)(8)(9)(10), persisting symptoms and failure of FGF-23 levels to normalize after complete excision of the metacarpal lesion prompted a detailed reevaluation of the case and the adoption of more sensitive imaging in the form of Ga-68 DOTANOC PET. It is likely that if the latter had been used as the preferred imaging modality at the time of his referral to our service, then a structured approach as outlined above, combining selective venous sampling and biopsy, would potentially have led to earlier identification of the multifocality, although still requiring two separate surgical procedures.…”

mentioning

confidence: 99%

Needle(s) in the Haystack—Synchronous Multifocal Tumor-Induced Osteomalacia

Annamalai

Sampathkumar

Kane

et al. 2016

The Journal of Clinical Endocrinology & Metabolism

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Oncogenic Osteomalacia Caused by Phosphaturie Mesenchymal Tumours in the Proximal and Shaft of the Tibia: A Case Report

Cited by 6 publications

References 27 publications

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Perioperative management of patients with severe hypophosphataemia secondary to oncogenic osteomalacia: Our experience and review of literature

Needle(s) in the Haystack—Synchronous Multifocal Tumor-Induced Osteomalacia

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