Sinonasal Glomangiopericytoma Causing Oncogenic Osteomalacia

Abstract

A 60-year-old woman suffered from recurrent femur neck fracture. Laboratory data showed serum hypophosphatemia, elevated alkaline phosphatase, normal serum calcium levels, and normal parathyroid hormone levels. Radiological examinations revealed a tumor in the right maxillary alveolar bone. The nasal cavity mass was removed, and the histological features were those of glomangiopericytoma. After removal of the tumor, some of the laboratory data normalized. Based on the clinical features, histopathological diagnosis and postoperative course of events, a diagnosis of glomangiopericytoma causing oncogenic osteomalacia was confirmed. We report a case of oncogenic osteomalacia caused by sinonasal glomangiopericytoma.

Characteristics and prognosis of glomangiopericytomas: A systematic review

“…After full‐text assessment, 139 articles were also excluded: articles containing no histologic findings (n = 20), unclear histology (n = 10), duplicate cases (n = 3), cases not arising in the sinonasal tract (n = 21), other sinonasal tumors, such as solitary fibrous tumor (n = 39), glomus tumor (n = 27), myopericytoma (n = 3), fibroma (n = 1), papilloma (n = 1), and lymphoma (n = 1), and reviews without information about individual cases (n = 13). Finally, 80 articles containing glomangiopericytoma of the sinonasal tract (337 cases) were identified as eligible for this study . Most articles (75/80; 94%) were published in English, except for 5 articles written in Korean …”

“…Sometimes check pain, facial bulging, or proptosis was present. Interestingly, bone fracture or whole body bone pain associated with osteomalacia was seen in 7 patients with glomangiopericytoma …”

“…We observed somewhat interesting clinical features of glomangiopericytomas. Osteomalacia was sometimes observed in patients with glomangiopericytoma . Oncogenic osteomalacia has been related with benign or malignant tumors of the bone and soft tissue, including osteosarcoma, enchondroma, nonossifying fibroma, and hemangioma .…”

“…Production of a humoral factor by the tumor has been considered the most probable pathogenesis of oncogenic osteomalacia. Tumor‐inducing humoral factor inhibits 25‐hydroxyvitamin D3‐hydroxylase activity, leading to a decrease in 1,25‐dihydroxyvitamin D3 synthesis . Although it was associated with a small group of patients with glomangiopericytomas, osteomalacia might be an unusual clinical presentation in patients with glomangiopericytoma.…”

“…Osteomalacia was sometimes observed in patients with glomangiopericytoma. 13,15,23,24,42,51,56 Oncogenic osteomalacia has been related with benign or malignant tumors of the bone and soft tissue, including osteosarcoma, enchondroma, nonossifying fibroma, and hemangioma. 24 Production of a humoral factor by the tumor has been considered the most probable pathogenesis of oncogenic osteomalacia.…”