Tumor-induced osteomalacia: A systematic literature review
Noelia Álvarez-Rivas,
Gloria Lugo-Rodríguez,
Jose Ramón Maneiro,
Carlota Iñiguez-Ubiaga,
Rafael Benito Melero-Gonzalez,
Tania Iglesias-Cabo,
Loreto Carmona,
Carlos García-Porrúa,
Francisco Javier de Toro-Santos
Affiliations
Noelia Álvarez-Rivas
Rheumatology Department, Hospital Público da Mariña, Servizo Galego de Saúde (SERGAS), Burela, Spain; Corresponding author at: Hospital Público da Mariña, Rúa Rafael Vior, s/n, Burela, Lugo. Spain.
Gloria Lugo-Rodríguez
Endocrinology Department, Complexo Hospitalario Universitario de A Coruña (CHUAC), Servizo Galego de Saúde (SERGAS), A Coruña, Spain
Jose Ramón Maneiro
Rheumatology Department, Complexo Hospitalario Universitario de Santiago (CHUS), Servizo Galego de Saúde (SERGAS), Santiago de Compostela, Spain
Carlota Iñiguez-Ubiaga
Rheumatology Department, Hospital Universitario del Bierzo, Servicio de Salud de Castilla y León (SACYL), Ponferrada, Spain
Rafael Benito Melero-Gonzalez
Rheumatology Department, Complexo Hospitalario Universitario de Ourense (CHUO), Servizo Galego de Saúde (SERGAS), Orense, Spain
Tania Iglesias-Cabo
Statistical Consulting Unit, Scientific and Technical Services, University of Oviedo, Oviedo, Spain
Loreto Carmona
Instituto de Salud Musculoesquelética, Madrid, Spain
Carlos García-Porrúa
Rheumatology Department, Complexo Hospitalario Universitario de Lugo (CHULA), Servizo Galego de Saúde (SERGAS), Lugo, Spain
Francisco Javier de Toro-Santos
Instituto de Investigación Biomédica de A Coruña (INIBIC), Universidade de A Coruña, Complexo Hospitalario Universitario de A Coruña (CHUAC), Servizo Galego de Saúde (SERGAS), A Coruña, Spain; Rheumatology Department, Complexo Hospitalario Universitario de A Coruña (CHUAC), Servizo Galego de Saúde (SERGAS), A Coruña, Spain
Introduction: Tumor-induced osteomalacia (TIO), is a rare acquired paraneoplastic syndrome characterized by defective bone mineralization, caused by the overproduction of fibroblast growth factor 23 (FGF23) by a tumor. Material and methods: We conducted a systematic review to identify all case reports of TIO, focusing on those associated with mesenchymal tumors. We followed the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) consensus, and we included patients with a diagnosis of TIO and histological confirmation of phosphaturic mesenchymal tumors or resolution of the condition after treatment of the tumor. Bibliographical searches were carried out until December 2023 in the Cochrane Library, Medline and Embase, as well as congress abstracts online. Results: We identified 769 articles with 1979 cases reported. Most patients were adults, with a higher incidence on men. Disease duration before diagnosis is a mean of 4.8 years. Most tumors were histologically classified as PMT. Lower limbs were the predominant location. Hypophosphatemia was present in 99.8 % of patients. The FGF23 was elevated at diagnosis in 95.5 %. Resection of the tumor was the treatment of choice in most of patients. After resection, there was a clinical improvement in 97.6 % of cases, and serum phosphorus and FGF23 levels returned to normal ranges in 91.5 % and 81.4 % of the patients, respectively. Conclusion: TIO is usually misdiagnosed with rheumatological or musculoskeletal disorders. The diagnosis should be suspected in patients with hypophosphatemic osteomalacia, and the measurement of serum FGF23 can be useful for diagnosis and management.
