Ossifying epulis in pseudohypo-parathyroidism: a case-based therapeutic approach

Abstract

AIM: The term Pseudohypoparathiroidism indicates a group of rare conditions characterised by end-organ resistance to the action of parathyroid hormone (PTH). Ossifying epulis (OE) is a exophytic gingival lesion characterised by spontaneous bone formation beneath the mucosa, which may affect children and adults: the exophytic, calcified outgrowths can occur in any bone and generally have favorable prognosis. Drug therapy may normalise calcium serum levels, but not completely avoid the occurrence of peripheral ossifying epulis. CASE REPORT: We report a representative case of a peripheral ossifying epulis in the mouth of a patient following a drug treatment protocol for her pseudohypoparathyroidism and to optimise serum markers. An 11-year-old girl was referred to our department, showing a bulky neoformation on the gingival margin of 0.6 mm diameter with sharp margins. The mass was completely excised. Histological analysis revealed distinctive features of a chronic and acute inflammatory microenvironment with plasma cells (positivity for CD38, MUM1, Lambda and Kappa chains) and bone tissue fragments with remodeling aspects referable to flogistic osteolysis. The biopsy result leads to hypothese a change in the patient's drug therapy. Multidisciplinary screening and individualised pharmacological treatment are strongly recommended in the clinical practice in order to improve the therapeutic results.