Oncogenic osteomalacia caused by occult nasal mesenchymal tumor: a monster in the cave

Ray, Sayantan; Chakraborty, Partha Pratim; Biswas, Kaushik; Beatrice, Anne M; Ghosh, Sujoy; Mukhopadhyay, Satinath; Chowdhury, Subhankar

doi:10.1093/omcr/omv031

Cited by 4 publications

(2 citation statements)

References 10 publications

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“…In the majority of cases, successful removal of tumors leads to recovery; however, long term follow-up should be performed in case of recurrence (16). Of the phosphate excreted by the kidney, ~80% is reabsorbed by the proximal renal tubule through the natrium-potassium co-transfer protein IIa (NaP IIa) (17).…”

Section: Discussionmentioning

confidence: 99%

Acquired hypophosphatemic osteomalacia is easily misdiagnosed or neglected by rheumatologists: A report of 9 cases

Wang

et al. 2018

Exp Ther Med

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The aim of the present study was to assist rheumatologists in differentiating hypophosphatemic osteomalacia (HO) from mimic rheumatology diseases. Clinical data was obtained from 9 patients with acquired HO, initially misdiagnosed as mimic rheumatologic diseases. The data were retrospectively analyzed and a literature review was performed. The etiology of the cases was as follows: Adefovir dipivoxil-induced Fanconi syndrome was present in 6 of the cases, 2 were tumors and 1 case was chronic nephropathy. The chief complaint was thoracic or back pain and arthralgia, followed by progressive muscle weakness and dramatic movement limitation. All patients were transferred to 3-6 hospitals for extended periods due to misdiagnosis with conditions such as ankylosing spondylitis, chronic arthritis, lumbar disc disease, osteoporosis and somatoform disorder. Hypophosphatemia was observed in the patients and bone scans revealed diffusely decreased tracer uptake, with multiple hot spots of fractured sites and involved joints. Furthermore, patients' bone density was markedly low compared with the normal range for their age and sex. In the present study, 6 of the patients recovered when adefovir dipivoxil was stopped. In 1 case, hypophosphatemia was ameliorated following tumor resection. The remaining patients, 1 with sub-skull tumor and 1 with chronic kidney disease, had poor prognoses due to incurable diseases. In conclusion, diagnosing HO is challenging for rheumatologists and physicians. Basic examinations of electrolyte balance and bone mineral density should be performed, as should tumor screening and a careful collection of patient medical history and drugs in young patients with unexplained thoracic or back pain and muscle weakness. Removing any secondary etiology, such as drugs may dramatically improve the patients clinical manifestations and result in an improved prognosis.

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Section: Discussionmentioning

confidence: 99%

Acquired hypophosphatemic osteomalacia is easily misdiagnosed or neglected by rheumatologists: A report of 9 cases

Wang

et al. 2018

Exp Ther Med

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“…[8–19] To be noticed, sinonasal hemangiopericytoma were unilateral in all cases except one with a mass in right nasal cavity extending into the left one, [8] and 8 cases were in the left side. [9–16] Six cases had regional bony resorption by CT. [8–14] Time for diagnosis was from 2 to 6 years since disease onset. One case reported severe bleeding, [17] and another an amount of bleeding of approximately 300 mL during resection of the sinonasal hemangiopericytoma.…”

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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Successful treatment of tumor-induced osteomalacia causing by phosphaturic mesenchymal tumor of the foot

et al. 2019

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Tumor-induced osteomalacia causing by phosphaturic mesenchymal tumor of the foot is exceedingly rare, thus may bring great challenges to the timely and proper diagnosis and treatment of clinicians. The only definitive management is removal of the phosphaturic mesenchymal tumor completely. The objective of this article is to report 2 unusual cases with tumor-induced osteomalacia causing by phosphaturic mesenchymal tumor of the foot. We describe 2 patients with phosphaturic mesenchymal tumor involving the foot who were successfully treated with tumor resection. On presentation to our institution, the patients both had signs of severe osteomalacia, and the patients’ most outstanding complaints were diffuse bone pain, general weakness, and disabled walking. A 53-year-old female underwent surgical excision of pathogenic tumor on the sole of left foot. A 62-year-old female underwent complete excision of pathogenic tumor of right plantar. The patients showed appropriate destruction of the tumor, adequate pain relief, and the elevated blood phosphorus levels compared with the previous status. Surgical resection is the most effective treatment option for patients with tumor-induced osteomalacia who can undergo appropriate surgical treatment. This represents a safe and reasonable approach to sustainably relieve pain and other symptoms with tumor-induced osteomalacia in the foot.

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Oncogenic osteomalacia caused by occult nasal mesenchymal tumor: a monster in the cave

Cited by 4 publications

References 10 publications

Acquired hypophosphatemic osteomalacia is easily misdiagnosed or neglected by rheumatologists: A report of 9 cases

Acquired hypophosphatemic osteomalacia is easily misdiagnosed or neglected by rheumatologists: A report of 9 cases

Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia

Successful treatment of tumor-induced osteomalacia causing by phosphaturic mesenchymal tumor of the foot

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