A UNIQUE CASE OF REGIONAL ODONTODYSPLASIA

Abstract

<h3>BACKGROUND</h3> Regional odontodysplasia is a rare condition involving defective enamel and dentin in a group of adjacent teeth. It may also include calcifications within the dental pulp and/or dental follicle.^1,2 First described by Hitchin in 1934,³ regional odontodysplasia affects dental mesodermal and ectodermal tissues along particular segments of the maxilla and/or mandible in the primary and/or permanent dentitions. There is a sex predilection, affecting women more than men.⁴ The current case is of a 6-year-old girl who presented with asymmetry of the face at a children's hospital in the eastern United States. <h3>CASE REPORT</h3> A 6-year-old female patient presented with a history of extractions of abscessed teeth #H and #I. Tooth #I was extracted because of abscess and facial cellulitis. The patient was admitted for intravenous antibiotics. Medical history was noncontributory. Dental history was positive for a drooping upper left lip and expansion of the upper left alveolar ridge. Panoramic and multiple periapical radiographs were acquired. Radiographically, the permanent maxillary left first and second molars and primary second molar had defective enamel and dentin along with taurodontism. The pulp chamber and canal were elongated. These features had the appearance of "ghost teeth." The bone around the maxillary left posterior aspect was also affected. <h3>DISCUSSION/CONCLUSIONS</h3> The presented tooth specimen exhibited irregular, abnormally distributed dentinal tubules. Globular calcified masses within the pulp were seen. Based on clinical, histopathologic, and radiographic findings, the case was diagnosed as regional odontodysplasia. Treatment included comprehensive dental care including endodontic, prosthodontic, orthodontic, and surgical intervention. Regional odontodysplasia is a rare developmental anomaly. It presents with a slight female predilection in a bimodal age distribution, most commonly affecting a single quadrant, with the maxilla more commonly involved. Clinical manifestations may include hyperplastic gingiva, failure of tooth eruption, periapical inflammation, bony changes, alterations of the dentition, and cutaneous nevi in the affected area.⁵