Cervicofacial infection in a 3-year-old child with regional odontodysplasia: A case report and literature review

Abstract

Regional odontodysplasia (RO) also known as ghost teeth is a rare developmental anomaly affecting the mesodermal and ectodermal components of teeth with characteristic clinical and radiographic findings. The enamel and dentin are hypomineralized and hypocalcified with short roots and open apices. The affected teeth have an abnormal morphology, meaning they are fragile and thin and liable to fracture and decay. Radiographically, there is a marked reduction in radiodensity with little distinction between the enamel and dentin, hence the term “ghost teeth.” RO generally affects one particular segment in one or both arches of the maxilla or mandible and can affect both the primary and permanent dentition. We report a unique case of a 3-year-old female who presented to Alder Hey Children's Hospital acutely unwell with a left-sided cervicofacial swelling from RO affecting the entire portion of the left maxilla.

Clinical, Radiographic and Histologic Evaluation of Regional Odontodysplasia: an Unusual Case Report with 6-Year Follow-Up

“…In this case, multidisciplinary treatment was required (paediatric dentist, maxillofacial surgeon, orthodontist) to achieve correct oral health, preserving the masticatory function, the control of bone development and general dentistry, the monitoring of tooth development with RO for maintenance or surgical treatment if necessary as well as subsequent prosthetic rehabilitation. From 2017 to November 2019, 7 more articles reporting 7 clinical cases were published and provided similar data to those collected by Alotaibi O, et al [9,13,17,19,[24][25][26][27][28].…”

“…Other factors causing OR have been mentioned, such as neural crest cell defects, viral infections, local trauma, Rh incompatibility, hyperpyrexia, ionizing radiation, teratogenic pharmacological agents and nutritional and/or metabolic alterations. OR has been associated with the presence of nevus, hypophosphatasia, hypocalcaemia, ectodermal dysplasia, and hydrocephalus; however, most cases of OR are not associated with medical alterations [9,[14][15][16][17].…”

“…They are also susceptible to fracture. Other common clinical signs after the eruption of RO teeth are gingival enlargement, periapical infections, and abscess formation [17].…”

Regional Odontodysplasia Affecting the Maxilla

“…Regional odontodysplasia is a rare nonhereditary anomaly that affects the development of enamel and dentin of the deciduous and/or permanent dentition [1][2][3]. The first description of RO was probably made by Hitchin in 1934 [4], however, some authors credit to McCall and Wald (1947) [5] the first report of this condition.…”

Odontodisplasia Regional y Agenesia de Premolares. Reporte de un Caso

“…La odontodisplasia regional (OR) es una rara condición y que afecta tanto la dentición temporal como a la permanente. Presenta un desarrollo anormal, no hereditario de los dientes que afecta tanto el tejido mesodérmico como ectodérmico (Bowden et al, 2018).…”