Osteomalacia cured by surgery

Furco, A; Røger, M; Mouchet, B; Richard, O; Martinache, X.; Fur, A.

doi:10.1016/s0953-6205(01)00196-0

Cited by 9 publications

(5 citation statements)

References 13 publications

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“…Although McCance [7] described the first case of tumor-induced osteomalacia in 1947, Prader et al [2] first recognized the causal role of a tumor in this syndrome in 1959. The tumor-induced osteomalacia occurs mainly in men and women evenly at approximately 40 years of age, and could rarely occur in children and adolescents [8]. Although the incidence of the phosphaturic mesenchymal tumor is not precisely known, according to reports from the Mayo clinic, 19 patients from 1965 to 2001 received treatment for tumor-induced osteomalacia.…”

Section: Discussionmentioning

confidence: 99%

A 9-month-old phosphaturic mesenchymal tumor mimicking the intractable rickets

Jung

Kim

Cho

et al. 2010

Journal of Pediatric Orthopaedics B

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Phosphaturic mesenchymal tumor is an extremely rare disease and is frequently associated with oncogenic osteomalacia showing paraneoplastic syndrome, which is characterized by phosphaturia, hypophosphatemia, normocalcemia, and decreased levels of 1,25-dihydroxyvitamin D3 associated with a tumor. A 2-year-old boy, who had a soft tissue tumor on his right thigh and previously diagnosed as myositis ossificans at 9-months-old, was presented with rachitic rosary and mildly enlarged tumor. Biochemical investigations showed hypophosphatemia, hyperphosphaturia, and an increased alkaline phosphatase level of 440 U/l (25-100 U/l), suggesting rickets, which was resistant to vitamin D dietary supplementation. We were certain of intractable rickets because of oncogenic hypophosphatemia and thus decided to excise the soft tissue mass. We observed laboratory improvement of rickets after 2 weeks. On the basis of surgical and histopathological examinations, the tumor was finally diagnosed as the phosphaturic mesenchymal tumor.

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Section: Discussionmentioning

confidence: 99%

A 9-month-old phosphaturic mesenchymal tumor mimicking the intractable rickets

Jung

Kim

Cho

et al. 2010

Journal of Pediatric Orthopaedics B

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“…Fibroblast growth factor‐23 (FGF‐23), a phosphatonin, has been identified as a major pathophysiological factor responsible for phosphate wasting in TIO 3, 4. Until this time, there were only 269 cases described in the world's literature over 61 years 5–98…”

Section: Introductionmentioning

confidence: 99%

Tumor-induced osteomalacia: An important cause of adult-onset hypophosphatemic osteomalacia in China: Report of 39 cases and review of the literature

Jiang

Xia

Xing

et al. 2012

Journal of Bone and Mineral Research

196

190

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Tumor-induced osteomalacia (TIO) is an acquired form of hypophosphatemia. Tumor resection leads to cure. We investigated the clinical characteristics of TIO, diagnostic methods, and course after tumor resection in Beijing, China, and compared them with 269 previous published reports of TIO. A total of 94 patients with adult-onset hypophosphatemic osteomalacia were seen over a 6-year period (January, 2004 to May, 2010 in Peking Union Medical College Hospital. After physical examination (PE), all patients underwent technetium-99m octreotide scintigraphy ( 99 Tc m -OCT). Tumors were removed after localization. The results demonstrated that 46 of 94 hypophosphatemic osteomalacia patients had high uptake in 99 Tc m -OCT imaging. Forty of them underwent tumor resection with the TIO diagnosis established in 37 patients. In 2 patients, the tumor was discovered on PE but not by 99 Tc m -OCT. The gender distribution was equal (M/F ¼ 19/20). Average age was 42 AE 14 years. In 35 patients (90%), the serum phosphorus concentration returned to normal in 5.5 AE 3.0 days after tumor resection. Most of the tumors (85%) were classified as phosphaturic mesenchymal tumor (PMT) or mixed connective tissue variant (PMTMCT). Recurrence of disease was suggested in 3 patients (9%). When combined with the 269 cases reported in the literature, the mean age and sex distribution were similar. The tumors were of bone (40%) and soft tissue (55%) origins, with 42% of the tumors being found in the lower extremities. In summary, TIO is an important cause of adult-onset hypophosphatemia in China. 99 Tc m -OCT imaging successfully localized the tumor in the overwhelming majority of patients. Successful removal of tumors leads to cure in most cases, but recurrence should be sought by long-term follow-up. ß

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“…In patients with tumor-induced osteomalacia, resection of the causative tumor is curative and laboratory values including serum FGF-23 levels can be followed to monitor for tumor recurrence. [56891113] While our patient did not have osteomalacia or abnormal laboratory values, her serum FGF-23 level was normal post-operatively and could potentially indicate tumor recurrence if it was elevated in the future. This will be reassessed in follow up if any of her future MRIs demonstrates evidence of recurrence.…”

Section: Discussionmentioning

confidence: 81%

Intracranial phosphaturic mesenchymal tumor, mixed connective tissue variant presenting without oncogenic osteomalacia

Bower¹,

Daugherty²,

Giannini³

et al. 2012

Surg Neurol Int

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Background:Phosphaturic mesenchymal tumor, mixed connective tissue variant (PMTMCT) is a rare tumor typically occurring in soft tissues and bone, causing oncogenic (tumor-induced) osteomalacia (TIO) through secretion of the phosphaturic hormone, fibroblast growth factor-23 (FGF-23). Rare tumors identical to PMTMCT occur without known TIO. Intracranial localization of PMTMCT is extremely rare, with only two cases reported in the literature. We present a very unusual case of a patient with an intracranial PMTMCT that presented with neurologic changes without osteomalacia.Case Description:A 67-year-old woman presented with progressive incontinence, apathy, and abulia after having undergone a total knee replacement 1 month earlier. Imaging disclosed a large left frontal anterior fossa mass. She underwent uncomplicated surgical resection of this tumor. Surprisingly, histopathology suggested PMTMCT. Reverse transcription polymerase chain reaction (RT-PCR) assay demonstrating FGF-23 expression in the tumor confirmed the diagnosis. Serum FGF-23 levels postoperatively were normal and she had no clinical or laboratory evidence of osteomalacia or phosphaturia.Conclusion:This report should serve to alert clinicians to the possibility that PMTMCT can be included in the differential diagnosis of intracranial masses even in the absence of tumor-induced osteomalacia.

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Osteomalacia cured by surgery

Cited by 9 publications

References 13 publications

A 9-month-old phosphaturic mesenchymal tumor mimicking the intractable rickets

A 9-month-old phosphaturic mesenchymal tumor mimicking the intractable rickets

Tumor-induced osteomalacia: An important cause of adult-onset hypophosphatemic osteomalacia in China: Report of 39 cases and review of the literature

Intracranial phosphaturic mesenchymal tumor, mixed connective tissue variant presenting without oncogenic osteomalacia

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