Oncogenic osteomalacia: Diagnostic importance of fibroblast growth factor 23 and F-18 fluorodeoxyglucose PET/CT SCAN for the diagnosis and follow-up in one case

Bone ◽  
2005 ◽  
Vol 36 (3) ◽  
pp. 375-378 ◽  
Author(s):  
J.L. Dupond ◽  
H. Mahammedi ◽  
D. Prié ◽  
F. Collin ◽  
H. Gil ◽  
...  
Medicina ◽  
2020 ◽  
Vol 56 (1) ◽  
pp. 34
Author(s):  
Cornelia Then ◽  
Evelyn Asbach ◽  
Harald Bartsch ◽  
Niklas Thon ◽  
Christian Betz ◽  
...  

A possible cause of hypophosphatemia is paraneoplastic secretion of fibroblast growth factor 23 (FGF-23). Tumors secreting FGF-23 are rare, mostly of mesenchymal origin, usually benign, and may be located anywhere in the body, including hands and feet, which are often not represented in conventional imaging. A 50-year-old woman presented with diffuse musculoskeletal pain and several fractures. Secondary causes of osteoporosis were excluded. Laboratory analysis revealed hypophosphatemia and elevated alkaline phosphatase, parathyroid hormone, and FGF-23. Thus, oncogenic osteomalacia due to neoplastic FGF-23 secretion was suspected. FDG-PET-CT and DOTATATE-PET-CT imaging demonstrated no tumor. Cranial MRI revealed a tumorous mass in the left cellulae ethmoidales. The tumor was resected and histopathological examination showed a cell-rich tumor with round to ovoid nuclei, sparse cytoplasm, and sparse matrix, resembling an olfactory neuroblastoma. Immunohistochemical analysis first led to diagnosis of olfactory neuroblastoma, which was later revised to phosphaturic mesenchymal tumor. Following the resection, FGF-23 and phosphate levels normalized. In conclusion, we here describe a patient with an FGF-23-secreting phosphaturic mesenchymal tumor with an unusual morphology. Furthermore, we emphasize diagnostic pitfalls when dealing with FGF-23-induced hypophosphatemia.


Circulation ◽  
2014 ◽  
Vol 129 (suppl_1) ◽  
Author(s):  
Amber L Fyfe-Johnson ◽  
Alvaro Alonso ◽  
Elizabeth Selvin ◽  
Sunil K Agarwal ◽  
James S Pankow ◽  
...  

Background: Elevated serum fibroblast growth factor-23 (FGF23), an endogenous hormone, is associated with endothelial dysfunction, chronic kidney disease, arterial wall stiffness, and inflammation. These factors may contribute to an increased risk of hypertension. To date, the association of FGF23 with incident hypertension has not been examined. Hypothesis: Elevated serum FGF23 will be positively associated with risk of incident hypertension. Methods: The ARIC study measured intact FGF23 in stored serum from 7,948 middle-aged men and women without hypertension at baseline (1990-92). Participants were examined during two follow-up visits, in 1993-95 and 1996-98. Incident hypertension was determined by measured blood pressure (DBP 90 mm Hg, or SBP140 mm Hg) and/or hypertension medication use during the follow-up exams. Multivariate Cox proportional hazards regression models and complementary log-log models were used to adjust for potential confounding variables. Results: During a median follow-up of 5.9 years, 27% (2,152/7,948) participants developed hypertension. A nonlinear association between serum FGF23 and incident hypertension was observed; only the highest decile of serum FGF23 was positively associated with incident hypertension (Table). After adjustment for demographics, the hazard ratio for incident hypertension was 1.37 (95% CI: 1.17, 1.60) for the highest decile of FGF23 compared to the lowest quintile. After adjustment for behaviors and adiposity the HR was 1.25 (95% CI: 1.07, 1.46). The association was further attenuated in the final model after adjusting for renal function (HR: 1.20, 95% CI: 1.03, 1.41). Complementary log-log models that accounted for interval censoring did not alter results. Conclusions: High levels ( 60.6 pg/mL) of FGF23 are associated with a modestly increased risk of incident hypertension in the general population. Next steps include replication of these findings in other cohorts, and examining the association with a longer follow-up period.


2012 ◽  
Vol 5 (3) ◽  
pp. 240-243 ◽  
Author(s):  
P.-A. Westerberg ◽  
T. Linde ◽  
D. Vanderschueren ◽  
J. Billen ◽  
I. Jans ◽  
...  

2019 ◽  
Vol 20 (3) ◽  
pp. 695 ◽  
Author(s):  
André Mansinho ◽  
Arlindo R. Ferreira ◽  
Sandra Casimiro ◽  
Irina Alho ◽  
Inês Vendrell ◽  
...  

The fibroblast growth factor (FGF) signaling pathway plays a key role in tumorigenesis and is recognized as a potential therapeutic target. In this study, the authors aimed to assess the impact of serum FGF23 levels in the prognosis of patients with cancer and bone metastases from solid tumors. A cohort of 112 patients with cancer and metastatic bone disease were treated with bone-targeted agents (BTA). Serum baseline FGF23 was quantified by ELISA and dichotomized in FGF23high and FGF23low groups. Additionally, the association between FGF23 and overall survival (OS) and time to skeletal-related events (TTSRE) was investigated. Baseline characteristics were balanced between groups, except for the median urinary N-terminal telopeptide (uNTX) level. After a median follow-up of 26.0 months, a median OS of 34.4 and 12.2 months was found in the FGF23low and FGF23high groups, respectively (multivariate HR 0.18, 95% CI 0.07–0.44, p = 0.001; univariate HR 0.27, p = 0.001). Additionally, TTSRE was significantly longer for patients with FGF23low (13.0 vs 2.0 months, p = 0.04). Overall, this study found that patients with FGF23low at baseline had longer OS and TTSRE. Further studies are warranted to define its role as a prognostic biomarker and in the use of drugs targeting the FGF axis.


Circulation ◽  
2020 ◽  
Vol 142 (Suppl_3) ◽  
Author(s):  
Ravi B Patel ◽  
Hongyan Ning ◽  
Ian H de Boer ◽  
Bryan Kestenbaum ◽  
Joao Ac Lima ◽  
...  

Background: While fibroblast growth factor 23 (FGF23) is associated with incident heart failure (HF) and atrial fibrillation (AF), the mechanisms driving these associations are unclear. FGF23 elevation leads to cardiomyocyte calcium handling abnormalities, suggesting that FGF23 may directly reduce myocardial function. Methods: In the Multi-Ethnic Study of Atherosclerosis, a cohort free of cardiovascular disease at recruitment, we evaluated the associations of serum FGF23 (2000-2002) with measures of left ventricular (LV) and left atrial (LA) mechanical function on cardiac magnetic resonance (CMR) at 10-year follow up (2010-2012). Results: Of 2,276 participants with baseline FGF23 and CMR at 10-year follow up, participants with higher FGF23 levels were more likely white race, taking anti-hypertensive medications, and had lower baseline glomerular filtration rate (GFR). After covariate adjustment, baseline FGF23 levels were independently associated with worse LV global circumferential strain, worse LV mid-wall circumferential strain, and lower LA total emptying fraction in later life ( Table ). The association of FGF23 and LV global circumferential strain was consistent across the spectrum of GFR ( Figure ). While higher FGF23 was associated with higher LV mass (β coefficient per SD higher: 1.14, 95% CI: 0.16, 2.12, P= 0.02), it was not associated with the presence of macroscopic myocardial scar (OR per SD higher: 1.12, 95% CI: 0.86-1.45, P= 0.42). Conclusions: Baseline FGF23 is independently associated with lower LV and LA systolic function in later life. These findings provide mechanistic insight driving the associations of FGF23 with development of both HF and AF.


2017 ◽  
Vol 58 (5) ◽  
pp. 981 ◽  
Author(s):  
Ji-Yeon Lee ◽  
Hye-Sun Park ◽  
Seunghee Han ◽  
Jiyu Kelly Lim ◽  
Namki Hong ◽  
...  

PLoS ONE ◽  
2018 ◽  
Vol 13 (4) ◽  
pp. e0196634 ◽  
Author(s):  
Marie Frimodt-Møller ◽  
Bernt Johan von Scholten ◽  
Henrik Reinhard ◽  
Peter Karl Jacobsen ◽  
Tine Willum Hansen ◽  
...  

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