J Clin Res Pediatr Endocrinol. Ahead of Print: JCRPE-78941 | DOI: 10.4274/jcrpe.galenos.2022.2021-8-14

Tumor - Induced Osteomalacia Associated with a Maxillary Tumor in Children: A Case Report and Review of the Literature

Ha Nguyen Thi¹, Cuong Pham Manh², Linh To Tuan³, Lan Anh Le Thi¹, Nam Nguyen Thanh⁴, Soamarat Vilaiyuk^5
1Pediatric Department, Bach Mai Hospital. Ha Noi Medical University, Viet Nam
²Diagnostic Imaging Central, Bach Mai Hospital, Viet Nam
³Maxillofacial, Plastic and Aesthetic Surgery Department, Viet Duc Hospital, Viet Nam
⁴Pediatric Department, Bach Mai Hospital, Viet Nam
⁵Rheumatology Division, Pediatric Department. Faculty of Medicine Ramathibodi Hospital. Mahidol University, Thailand

Tumor-induced osteomalacia (TIO) is a rare paraneoplastic disorder of hypophosphatemia associated with a tumor-producing fibroblast growth factor 23 (FGF23). The maxillofacial tumor is rarely involved in TIO, especially maxillary TIO in children. We presented 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 received surgical removal of tumor. After 21 months follow-up, he was pain free and had returned to full activity. We reviewed 5 reports of pediatric patients with TIO in oral and maxillofacial region. Tumors localized in mandible (2/5), maxilla (2/5), both mandible and maxilla (1/5). Mean time from onset of signs to diagnosis was 21,9 months.

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

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