J Clin Res Pediatr Endocrinol. 2023; 15(4): 421-425 | DOI: 10.4274/jcrpe.galenos.2022.2021-8-14

Tumor-induced Osteomalacia in a Boy with Maxillary Ossifying Fibroma

Ha Nguyen Thi¹, Cuong Pham Manh², TuanLinh To³, Le Thi Lan Anh¹, Nam Nguyen Thanh⁴, Soamarat Vilaiyuk^5
1Hanoi Medical University, Bach Mai Hospital, Clinic of Pediatrics, Hanoi, Vietnam
²Bach Mai Hospital, Diagnostic Imaging Central, Hanoi, Vietnam
³Viet Duc Hospital, Maxillofacial, Department of Plastic and Aesthetic Surgery, Hanoi, Vietnam
⁴Bach Mai Hospital, Clinic of Pediatrics, Hanoi, Vietnam
⁵Mahidol University Faculty of Medicine; Ramathibodi Hospital, Department of Pediatrics, Division of Rheumatology, Bangkok, Thailand

Tumor-induced osteomalacia (TIO) is a rare, paraneoplastic disorder of hypophosphatemia associated with elevated tumor-produced fibroblast growth factor 23 (FGF23). Maxillofacial tumors are rarely involved in TIO, especially maxillary TIO in children. We present a 14-year-old boy with osteomalacia and high serum levels of FGF23, a hormone associated with decreased phosphate resorption, due to a maxillary tumor. The patient was treated with oral phosphorus and calcitriol, and surgical removal of the tumor was performed. After 21 months follow-up, he was pain free and had returned to full activity. We review the reported pediatric cases of TIO in the maxillofacial and oral region and discuss the management of these patients considering the published evidence.

Keywords: Tumor-induced osteomalacia, fibroblast growth factor-23, maxilla, children

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