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Otodental syndrome. A case report

L Van Doorne1, G Wackens, M De Maeseneer

  • 1Department of Oral and Maxillofacial Surgery, Free University of Brussels, Belgium.

International Journal of Oral and Maxillofacial Surgery
|May 30, 1998
PubMed
Summary

Otodental syndrome, a rare genetic disorder, presents unique dental and hearing abnormalities. This case highlights previously undocumented features, expanding our understanding of this condition.

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Area of Science:

  • Genetics
  • Ophthalmology
  • Dentistry

Background:

  • Otodental syndrome is a rare genetic disorder characterized by hearing loss and dental anomalies.
  • It follows an autosomal dominant inheritance pattern.

Observation:

  • A 12-year-old boy with otodental syndrome exhibited characteristic dental abnormalities, including bulbous canines, globe-shaped posterior teeth, and agenesis of maxillary premolars.
  • High-frequency sensorineural hearing loss was also noted.

Findings:

  • This patient presented with previously unreported findings: generalized macrodontia, delayed mandibular premolar mineralization, and supplementary permanent maxillary canines.
  • Genetic studies confirmed an autosomal dominant trait.

Implications:

  • These novel findings expand the known clinical spectrum of otodental syndrome.
  • Further research into the genetic basis and phenotypic variability of otodental syndrome is warranted.

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