Diagnosis and management of tumor-induced osteomalacia: a single center experience


Hacisahinogullari H., Tekin S., Tanrikulu S., Saribeyliler G., Yalin G., Bilgic B., ...Daha Fazla

ENDOCRINE, cilt.82, sa.2, ss.427-434, 2023 (SCI-Expanded) identifier identifier identifier

  • Yayın Türü: Makale / Tam Makale
  • Cilt numarası: 82 Sayı: 2
  • Basım Tarihi: 2023
  • Doi Numarası: 10.1007/s12020-023-03450-3
  • Dergi Adı: ENDOCRINE
  • Derginin Tarandığı İndeksler: Science Citation Index Expanded (SCI-EXPANDED), Scopus, BIOSIS, CAB Abstracts, Chemical Abstracts Core, EMBASE, MEDLINE, Veterinary Science Database
  • Sayfa Sayıları: ss.427-434
  • Anahtar Kelimeler: Fibroblast growth factor 23, Hypophosphatemia, Phosphaturic mesenchymal tumor, Tumor-induced osteomalacia
  • İstanbul Üniversitesi Adresli: Evet

Özet

PurposeThe aim of this study is to review the clinical and laboratory characteristics, diagnostic and treatment modalities of tumor-induced osteomalacia (TIO) cases managed in a single center.Material methodsDemographic and clinical features, biochemical findings, diagnostic procedures, treatment modalities, and outcomes of nine patients who had the diagnosis of TIO were reviewed retrospectively.ResultsMean age of the study group (F/M: 4/5) was 45.8 & PLUSMN; 10.8 years, and mean time from the onset of symptoms to diagnosis was 4.7 & PLUSMN; 2.8 years. The clinical manifestations were muscle weakness and difficulty in walking (8/9), hip pain (3/9), multiple fractures (2/9), stress fracture (2/9). Mean plasma phosphorus concentration was 1.28 & PLUSMN; 0.4 mg/dl at presentation. We performed radionuclide imaging modalities (18F-FDG PET/CT, Ga68-DOTATATE PET/CT, octreotide scintigraphy) in seven of nine patients, and tumor was detected in all. Lower extremity (n = 6; %67), head region (n = 2; %22) and thorax (n = 1; %11) were the tumor locations of our cases. Eight patients underwent surgery and remission was achieved postoperatively in all of the operated patients and plasma phosphorus level normalized in 4 & PLUSMN; 2 days. Pathological examination revealed mesenchymal tumors with different subtypes. Recurrence occurred in three patients at 13 & PLUSMN; 10.5 months after the first surgery. Two patients were reoperated and radiotherapy was also performed in one of them.ConclusionHypophosphatemia necessitates careful evaluation for the etiology. TIO is one of the important causes of adult-onset hypophosphatemic osteomalacia. Diagnosis of TIO is essential because the laboratory and clinical findings resolve after appropriate treatment.