Tumor induced osteomalacia in head and neck region: single center experience and systematic review

Abstract

Tumor-induced osteomalacia in the head and neck region remains a challenging diagnosis to manage. Literature pertaining to management and outcome details remains sparse. We describe two cohorts: cohort 1 included seven patients from a single center in Western India with tumors located in paranasal sinuses (n = 3), intracranial (n = 2) and maxilla (n = 2). The unique features from our series is the management of persistent disease with radiation therapy (n = 2) and peptide receptor radionuclide therapy (PRRT) (n = 1). Cohort two has 163 patients identified from 109 publications for systematic review. Paranasal sinuses, mandible, intracranial disease, maxilla and oral cavity, in descending order, are reportedly common tumor sites. Within this cohort, mean age was 46 ± 14 years at presentation with 44.1% having local symptoms. Duration of symptoms varied from 1 to 240 months. Pre-surgery mean serum phosphorus was 1.4 ± 0.4 mg/dL and median FGF-23 levels were 3.6 (IQR:1.8–6.8) times of normal upper limit of normal. Majority (97.5%) were managed primarily with surgical excision; however, primary radiotherapy (n = 2) and surgery combined with radiotherapy (n = 2) were also reported. Twenty patients had persistent disease while nine patients had recurrence, more commonly noted with intracranial and oral cavity tumors. Surgery was the most common second mode of treatment employed succeeded by radiotherapy. Four patients had metastatic disease. The most common histopathological diagnosis reported is PMT mixed connective tissue, while the newer terminology ‘PMT mixed epithelial and connective tissue type’ has been described in 15 patients.

Hypophosphatemic Osteomalacia in a Young Adult

“…These tumors are usually slow growing, benign and small. They are commonly found in the craniofacial region or in the extremities [ 4 ]. Other histological types include fibromas, chondrosarcomas, neuroblastomas, and prostate carcinomas.…”

Tumor-induced osteomalacia caused by head and neck tumors: report of an unusual case and literature review

“…A retrospective study found that about 50% of patients showed signs of tenderness and/or swelling at the proposed site of the tumor or swelling. Therefore, careful physical examination can help early detection of tumors in TIO patients (4). Tumors in jaw can lead to pathological loosening of teeth, fall off, sometimes slightly bulging jaw.…”

“…After tumor resection, the blood phosphorus level quickly returned to normal and the symptoms were relieved within few months. To date, less than 200 cases of osteomalacia caused by maxillofacial tumors have been reported in literature (4). There are two main reasons for the low number of reported cases: first, the prevalence of TIO is low and less cases occur in the maxillofacial region; second, clinicians are poorly aware of the relationship between osteomalacia and mesenchymal tumors.…”

Successful Treatment of Tumor-Induced Osteomalacia by Multidisciplinary Therapy with Radiation to Intracranial Fibromyxoid Tumor

“…However, some patients may develop tumor recurrence and benefit from a second excision [2]. Radiotherapy has been rarely used in case of inoperable tumors or incomplete resections [3][4][5][6][7][8][9]. We report the case of a patient who had a multiple recurrent ossifying fibromyxoid tumor in the left frontal sinus causing TIO.…”