Bilateral Subtrochanteric Fractures in Tumour-Induced Osteomalacia Caused by a Nasal Haemangiopericytoma

Burnand, Henry; Samuels, Abigail; Hagan, Ian; Sawant, Nitin; Mutimer, J

doi:10.5301/hip.2012.9235

Cited by 3 publications

(4 citation statements)

References 12 publications

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“…Here we present a detailed review of published English literature for TIO cases involving head and neck region ( n = 163) (5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30, 31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41, 42, 43, 44, 45, 46, 47, 48, 49, 50, 51, 52, 53, 54, 55, 56, 57, 58, 59, 60, 61, 62, 63, 64, 65, 66, 67, 68, 69, 70, 71, 72, 73, 74, 75, 76, 77, 78, 79, 80, 81, 82, 83, 84, 85, 86, 87, 88, 89, 90, 91, 92, 93, 94, 95, 96, …”

Section: Discussionmentioning

confidence: 99%

Tumor induced osteomalacia in head and neck region: single center experience and systematic review

Shah

Lila

Jadhav

et al. 2019

Endocrine Connections

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Tumor-induced osteomalacia in the head and neck region remains a challenging diagnosis to manage. Literature pertaining to management and outcome details remains sparse. We describe two cohorts: cohort 1 included seven patients from a single center in Western India with tumors located in paranasal sinuses (n = 3), intracranial (n = 2) and maxilla (n = 2). The unique features from our series is the management of persistent disease with radiation therapy (n = 2) and peptide receptor radionuclide therapy (PRRT) (n = 1). Cohort two has 163 patients identified from 109 publications for systematic review. Paranasal sinuses, mandible, intracranial disease, maxilla and oral cavity, in descending order, are reportedly common tumor sites. Within this cohort, mean age was 46 ± 14 years at presentation with 44.1% having local symptoms. Duration of symptoms varied from 1 to 240 months. Pre-surgery mean serum phosphorus was 1.4 ± 0.4 mg/dL and median FGF-23 levels were 3.6 (IQR:1.8–6.8) times of normal upper limit of normal. Majority (97.5%) were managed primarily with surgical excision; however, primary radiotherapy (n = 2) and surgery combined with radiotherapy (n = 2) were also reported. Twenty patients had persistent disease while nine patients had recurrence, more commonly noted with intracranial and oral cavity tumors. Surgery was the most common second mode of treatment employed succeeded by radiotherapy. Four patients had metastatic disease. The most common histopathological diagnosis reported is PMT mixed connective tissue, while the newer terminology ‘PMT mixed epithelial and connective tissue type’ has been described in 15 patients.

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Section: Discussionmentioning

confidence: 99%

Tumor induced osteomalacia in head and neck region: single center experience and systematic review

Shah

Lila

Jadhav

et al. 2019

Endocrine Connections

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“…One case reported severe bleeding, [17] and another an amount of bleeding of approximately 300 mL during resection of the sinonasal hemangiopericytoma. [10]…”

Section: Discussionmentioning

confidence: 99%

“…[24] Bleeding during surgery should be concerned that 1 case reported severe bleeding, [17] and another an amount of bleeding of approximately 300 mL during surgery. [10] For those causative tumors were not located, or complete resection were impossible, medical treatment should be initiated. Phosphorus and active vitamin D supplementation is commonly used for TIO treatment.…”

Section: Discussionmentioning

confidence: 99%

Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia

Huang

Yang³

et al. 2018

Medicine

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Rationale:Tumor-induced osteomalacia (TIO) is a rare, paraneoplastic syndrome featured with fibroblast growth factor 23 (FGF23) secretion primarily by benign mesenchymal tumors and sometimes by malignancies. TIO diagnosis and treatment is often delayed because TIO usually has nonspecific generalized bone pain and weakness, and location of TIO tumor is quite challenging. Very few TIO caused by sinonasal hemangiopericytoma have been reported in the literature.Patient concerns:A 40-year-old Chinese woman presented with diffuse bone pain for more than 1 year. Laboratory examination showed hypophosphatemia, hyperphosphaturia, hypocalcemia, an elevated serum alkaline phosphatase (ALP) level and bone-specific ALP level. Imaging studies revealed low bone mineral density (BMD) and multiple pseudofractures at the ribs. F-18 fluorodeoxyglucose positron emission tomography was negative in searching for tumors. Because no tumor was located, the patient was treated with oral phosphate, calcium, and alfacalcidol, and achieved great relief in her symptoms and improvement in BMD. Six years later, the patient had breast cancer surgery and received chemotherapy, and still had hypophosphatemia. During this time, nasopharyngo-fiberscope showed nasal mass in her left nasal cavity. Then she had her nasal polyps removed and surprisingly the serum phosphate became normal.Diagnoses and interventions:The patient had the nasal mass resected, and pathological diagnosis of the nasal mass was sinonasal hemangiopericytoma. Immunohistochemical analysis was positive for FGF23. Thus the final diagnosis was osteomalacia induced by sinonasal hemangiopericytoma. Phosphate supplementation and alfacalcidol were discontinued.Outcomes:The patient had normal serum phosphate after 6-month follow-up.Lessons:By presenting this case, we hope to remind clinicians that in patients with osteomalacia with undetermined reason and intranasal polypoid mass, sinonasal hemangiopericytoma should be suspected.

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“…Bumard H and co workers presented a case with bilateral insufficiency fractures of the proximal femur often have a pathological basis of hemangiopericytoma. Diagnosis of rare causes of insufficiency fractures can be challenging when the causative factor is a tumour [18].…”

Section: Discussionmentioning

confidence: 99%

Functional and Anatomical Imaging for Early Diagnosis of Tumor Induced Osteomalacia

Malik¹

2017

CTOIJ

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Oncogenic osteomalacia, or tumor-induced osteomalacia (TIO), is an acquired paraneoplastic syndrome being associated with benign or in rare cases malignant mesenchymal. Patients with TIO often present with many years of nonspecific but progressive symptoms commonly with bone pain, muscle weakness, reduced height, and multiple fractures. Due to this nonspecificity, patients are often misdiagnosed with a variety of musculoskeletal, rheumatologic diseases and sometimes even having psychiatric disorders. Not only that these tumors are rare but also finding the tumor can be a major diagnostic challenge and may need total body magnetic resonance imaging, computed tomography and scintigraphy using radio labelled somatostatin analogue and sometimes even venous sampling. Majority of clinicians, radiologists and pathologists are not aware of this rare disease because mostly the cases are described in rheumatology practice. We provide mini review of the current literature by having looked at the different studies done including case reports to conclude the best practice for the diagnosis and treatment of this rare entity.

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Bilateral Subtrochanteric Fractures in Tumour-Induced Osteomalacia Caused by a Nasal Haemangiopericytoma

Cited by 3 publications

References 12 publications

Tumor induced osteomalacia in head and neck region: single center experience and systematic review

Tumor induced osteomalacia in head and neck region: single center experience and systematic review

Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia

Functional and Anatomical Imaging for Early Diagnosis of Tumor Induced Osteomalacia

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