Tumor(s) induced osteomalacia--a curious case of double trouble

J Clin Endocrinol Metab. 2014 Feb;99(2):395-8. doi: 10.1210/jc.2013-3791. Epub 2013 Dec 2.

Abstract

Context: We report a case of tumor-induced osteomalacia with evidence of synchronous multifocal fibroblast growth factor 23 (FGF23) production.

Objective: The aim is to present a case of tumor-induced osteomalacia and to highlight the fact that incomplete removal of multifocal FGF23-producing tumors, which are not entirely picked up by functional imaging, could be the cause of treatment failure.

Setting: The patient was treated in the Department of Endocrinology of a tertiary care center in India.

Patient: We report the case of a 42-year-old male with tumor-induced osteomalacia.

Intervention: We treated the tumor-induced osteomalacia with staged surgery of the two tumors. The 18F-fluorodeoxyglucose (FDG)-avid lesion (considered the sole culprit lesion after functional imaging) was resected first, followed by the non-FDG-avid lesion. The sequential removal of both tumors resulted in complete cure.

Results: The patient had hypophosphatemia and hyperphosphaturia. C-Terminal FGF23 level was elevated. Positron emission tomography-computed tomography showed two lesions-an FDG-avid lesion in the right leg, and a non-avid lesion in the left thigh. After removal of the FDG-avid lesion, the hypophosphatemia persisted, and the FGF23 level showed only modest reduction. The patient had complete clinical and biochemical resolution only after removal of the second non-FDG-avid tumor.

Conclusions: We present the case of a tumor-induced osteomalacia whose biochemical parameters did not improve after removal of the FDG-avid tumor initially. The possibility of multifocal FGF23 production was considered, and the second, non-FDG-avid lesion was resected, which resulted in complete cure. Thorough clinical examination and meticulous follow-up with documentation of the biochemical resolution are necessary for management of all patients with this rare disorder.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Fibroblast Growth Factor-23
  • Humans
  • Hypophosphatemia / blood
  • Hypophosphatemia / diagnosis*
  • Hypophosphatemia / surgery
  • Hypophosphatemia, Familial / blood
  • Hypophosphatemia, Familial / diagnosis*
  • Hypophosphatemia, Familial / surgery
  • India
  • Male
  • Middle Aged
  • Neoplasms, Connective Tissue / blood
  • Neoplasms, Connective Tissue / diagnosis*
  • Neoplasms, Connective Tissue / surgery
  • Osteomalacia
  • Paraneoplastic Syndromes

Supplementary concepts

  • Oncogenic osteomalacia