Development of Tumor-induced Osteomalacia in a Subcutaneous Tumor, Defined by Venous Blood Sampling of Fibroblast Growth Factor-23

Ogura, Eriko; Kageyama, Kazunori; Fukumoto, Seiji; Yagihashi, Norito; Fukuda, Yoshiko; Kikuchi, Toru; Masuda, Mitsuo; Suda, Toshio

doi:10.2169/internalmedicine.47.0761

Cited by 24 publications

(10 citation statements)

References 15 publications

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“…FGF23 regulates urinary phosphate excretion, as well as production of vitamin D and parathyroid hormone. FGF23 was shown to be positively associated with TmP/GFR in healthy subjects [34], and, in addition, phosphaturic activities of FGF23 have also been convincingly demonstrated in the genetically engineered animal models [35, 36] and in a patient with high FGF23 levels due to tumor-induced osteomalacia [37]. …”

Section: Discussionmentioning

confidence: 99%

Association of serum levels of FGF23 and α-Klotho with glomerular filtration rate and proteinuria among cardiac patients

et al. 2014

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BackgroundExpression and/or excretion of fibroblast growth factor-23 (FGF23) and its co-receptor Klotho are altered in patients with end-stage renal disease. The possibility that the FGF23/α-Klotho system mediates the aggravated cardiovascular outcome among patients with chronic kidney disease (CKD) has been suggested. We determined whether FGF23 and α-Klotho concentrations are altered among patients with reduced renal function and proteinuria.MethodsSerum FGF23 and α-Klotho were measured in cardiology patients who were not undergoing chronic hemodialysis. Estimated glomerular filtration rate (eGFR) was correlated negatively with FGF23 and positively with α-Klotho.ResultsThe correlation between FGF23 and the renal tubular maximum reabsorption rate of phosphate to the GFR (TmP/GFR) was not significant, but that between FGF23 and serum calcium or inorganic phosphate was significant among patients with an estimated GFR of less than 60 mL/min/m2. By stepwise multivariate regression analysis, eGFR was selected as significant predictor for FGF23 or α-Klotho among patients with an estimated GFR of less than 60 mL/min/m2; however, urine albumin/creatinine ratio was not selected as a predictor for FGF23 or α-Klotho irrespective of the eGFR levels. In patients with eGFR of <60 mL/min/1.73 m2, UACR was significantly associated with log(FGF23); but, this association did not remain statistically significant in a multivariate model.ConclusionsAmong cardiology patients with various stages of CKD, serum concentrations of FGF23 and α-Klotho were associated with renal function, but not with the extent of proteinuria.

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Section: Discussionmentioning

confidence: 99%

Association of serum levels of FGF23 and α-Klotho with glomerular filtration rate and proteinuria among cardiac patients

et al. 2014

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“…Interestingly, Takeuchi et al [6] located a TIO before surgery by testing venous blood sampling for FGF23, indicating that the serum FGF23 level was higher around the tumor than in other parts of the body. This method, combined with other modalities like MRI, was also reported by other authors as an approach to locate the small tumor responsible for TIO[5],[23].…”

Section: Discussionmentioning

confidence: 69%

Tumor-induced osteomalacia with elevated fibroblast growth factor 23: a case of phosphaturic mesenchymal tumor mixed with connective tissue variants and review of the literature

Yuan²,

Jiang³

et al. 2011

Chin J Cancer

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Tumor-induced osteomalacia (TIO), or oncogenic osteomalacia (OOM), is a rare acquired Paraneoplastic disease characterized by renal phosphate wasting and hypophosphatemia. Recent evidence shows that tumor-overexpressed fibroblast growth factor 23 (FGF23) is responsible for the hypophosphatemia and osteomalacia. The tumors associated with TIO are usually phosphaturic mesenchymal tumor mixed connective tissue variants (PMTMCT). Surgical removal of the responsible tumors is clinically essential for the treatment of TIO. However, identifying the responsible tumors is often difficult. Here, we report a case of a TIO patient with elevated serum FGF23 levels suffering from bone pain and hypophosphatemia for more than three years. A tumor was finally located in first metacarpal bone by octreotide scintigraphy and she was cured by surgery. After complete excision of the tumor, serum FGF23 levels rapidly decreased, dropping to 54.7% of the preoperative level one hour after surgery and eventually to a little below normal. The patient's serum phosphate level rapidly improved and returned to normal level in four days. Accordingly, her clinical symptoms were greatly improved within one month after surgery. There was no sign of tumor recurrence during an 18-month period of follow-up. According to pathology, the tumor was originally diagnosed as “glomangioma” based upon a biopsy sample, “proliferative giant cell tumor of tendon sheath” based upon sections of tumor, and finally diagnosed as PMTMCT by consultation one year after surgery. In conclusion, although an extremely rare disease, clinicians and pathologists should be aware of the existence of TIO and PMTMCT, respectively.

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“…Although some reports referred to the usefulness of SVS (10, 11), there was only one case of the tumor responsible for TIO being localized by SVS alone (12). Ito et al .…”

Section: Discussionmentioning

confidence: 99%

Effective localization in tumor-induced osteomalacia using 68Ga-DOTATOC-PET/CT, venous sampling and 3T-MRI

Shintaro

Hiroyuki

Furukawa

et al. 2017

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryTumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by renal phosphate wasting leading to hypophosphatemia due to excessive actions of fibroblast growth factor 23 (FGF23) produced by the tumors. Although the best way of curing TIO is complete resection, it is usually difficult to detect the culprit tumors by general radiological modalities owing to the size and location of the tumors. We report a case of TIO in which the identification of the tumor by conventional imaging studies was difficult. Nonetheless, a diagnosis was made possible by effective use of multiple modalities. We initially suspected that the tumor existed in the right dorsal aspect of the scapula by 68Ga-DOTATOC positron emission tomography/computed tomography (68Ga-DOTATOC-PET/CT) and supported the result by systemic venous sampling (SVS). The tumor could also be visualized by 3T-magnetic resonance imaging (MRI), although it was not detected by 1.5T-MRI, and eventually be resected completely. In cases of TIO, a stepwise approach of 68Ga-DOTATOC-PET/CT, SVS and 3T-MRI can be effective for confirmation of diagnosis.Learning points:TIO shows impaired bone metabolism due to excessive actions of FGF23 produced by the tumor. The causative tumors are seldom detected by physical examinations and conventional radiological modalities.In TIO cases, in which the localization of the culprit tumors is difficult, 68Ga-DOTATOC-PET/CT should be performed as a screening of localization and thereafter SVS should be conducted to support the result of the somatostatin receptor (SSTR) imaging leading to increased diagnosability.When the culprit tumors cannot be visualized by conventional imaging studies, using high-field MRI at 3T and comparing it to the opposite side are useful after the tumor site was determined.

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Development of Tumor-induced Osteomalacia in a Subcutaneous Tumor, Defined by Venous Blood Sampling of Fibroblast Growth Factor-23

Abstract: A 25-year-old

Cited by 24 publications

References 15 publications

Association of serum levels of FGF23 and α-Klotho with glomerular filtration rate and proteinuria among cardiac patients

Association of serum levels of FGF23 and α-Klotho with glomerular filtration rate and proteinuria among cardiac patients

Tumor-induced osteomalacia with elevated fibroblast growth factor 23: a case of phosphaturic mesenchymal tumor mixed with connective tissue variants and review of the literature

Effective localization in tumor-induced osteomalacia using 68Ga-DOTATOC-PET/CT, venous sampling and 3T-MRI

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