Tumor-induced hypophosphatemic osteomalacia caused by a mesenchymal tumor of the mandible managed by a segmental mandibulectomy and microvascular reconstruction with a free fibula flap

Head Neck. 2019 Jun;41(6):E93-E98. doi: 10.1002/hed.25657. Epub 2019 Mar 11.

Abstract

Background: Tumor-induced osteomalacia is a rare paraneoplastic syndrome in which patients develop hypophosphatemia and osteomalacia.

Methods and results: Here, we report a unique case of a 42-year-old man who presented to our institution with a 1-year history of pain in his ribs, hips, lower back, and feet. Radiologic examination revealed a decrease in bone density and multiple insufficiency fractures. Laboratory evaluation revealed hypophosphatemia, low serum 1,25 dihydroxy vitamin D3 , and elevated fibroblast growth factor 23 (FGF23). A positron emission tomography/CT scan showed increased uptake in the right mandibular third molar region. Panoramic radiography and CT scanning showed a lytic expansile bone lesion. A mandibular bone biopsy revealed a mixed connective tissue tumor. A right segmental mandibulectomy was performed, followed by microvascular reconstruction. The resection was confirmed by normalization of serum phosphate and FGF23.

Conclusion: Successful management of this condition was achieved, with complete surgical resection of the tumor and reconstructive surgery.

Keywords: alkaline phosphatase; fibroblast growth factor 23; hypophosphatemia; mesenchymal tumor; tumor-induced osteomalacia.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Fibroblast Growth Factor-23
  • Fibula / transplantation
  • Free Tissue Flaps
  • Humans
  • Hypophosphatemia / etiology*
  • Hypophosphatemia / surgery
  • Male
  • Mandibular Neoplasms / complications*
  • Mandibular Neoplasms / surgery*
  • Mandibular Osteotomy*
  • Neoplasms, Connective Tissue / complications
  • Neoplasms, Connective Tissue / etiology*
  • Neoplasms, Connective Tissue / surgery
  • Osteomalacia
  • Paraneoplastic Syndromes

Supplementary concepts

  • Oncogenic osteomalacia