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Ameloblastic Fibroodontoma of Mandible Causing Tumor Induced Osteomalacia: A Case Report with Review of 88 Phosphaturic Oral Neoplasms

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Abstract

Tumor induced osteomalacia (TIO) is a rare paraneoplastic syndrome associated with tumors secreting fibroblast growth factor 23, which induces osteomalacia. Microscopically, these tumors most commonly show benign phosphaturic mesenchymal tumors. We report the first case of phosphaturic ameloblastic fibro-odontoma (AFO) manifesting as osteomalacia. Our index patient was a 33-year-old male who was diagnosed with TIO and AFO in the mandible was identified as the cause. Our case is unique as AFO is considered as a hamartoma. To the best of our knowledge, there is no hamartoma reported till date causing phosphaturic osteomalacia. As AFO demonstrates mixed epithelial and mesenchymal origin, we propose a new histopathological subtype of TIO—“phosphaturic tumor of mixed epithelial and mesenchymal origin”. A review of literature focused on TIO caused by oral lesions revealed 88 oral neoplasms which matched our search criteria. Due to the rarity and unpredictable behavior of TIOs, a high index of suspicion, a broad diagnostic approach, detailed history and multidisciplinary investigations are crucial for establishing the definitive diagnosis and proper treatment recommendations.

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Authors and Affiliations

  1. Department of Oral Pathology and Microbiology, Centre for Dental education and Research, All India Institute of Medical Sciences, Delhi, India

    Deepika Mishra & Harpreet Kaur

  2. Department of Radiodiagnosis, All India Institute of Medical Sciences, Delhi, India

    Ashu Seith Bhalla & Smita Manchanda

  3. Department of Oral and Maxillofacial Surgery, Centre for Dental Education and Research, All India Institute of Medical Sciences, Delhi, India

    Jithin Sasikumar, Bhaskar Agarwal & Ajoy Roychoudhury

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  1. Deepika Mishra
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Mishra, D., Kaur, H., Bhalla, A.S. et al. Ameloblastic Fibroodontoma of Mandible Causing Tumor Induced Osteomalacia: A Case Report with Review of 88 Phosphaturic Oral Neoplasms. Head and Neck Pathol 15, 975–988 (2021). https://doi.org/10.1007/s12105-020-01267-4

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