Hyper IgE syndrome is a rare immunodeficiency that is characterized by high levels of IgE in the serum, recurrent skin infections, frequent respiratory tract infections, and common young age mortality. This multisystem disorder also includes unusual facial, dental and skeletal features. The oral manifestations of patients with hyper IgE syndrome include chronic oral candidiasis and eruption disturbances of permanent teeth due to delayed exfoliation of primary teeth. The possible association between hyper IgE and some of the clinical signs of the syndrome is discussed. Objectives: To demonstrate the oral manifestations of this rare syndrome. Methods: A case of a 10-year-old girl diagnosed with hyper IgE syndrome is presented. Results: The patient showed marked oral candidiasis that included chronic angular chelitis and median rhomboid glossitis, as well as partial retention of the primary dentition. Conclusion: It is extremely important for dental surgeons in general and pediatric dentists in particular, to recognize this syndrome and its oral manifestations to avoid life-treating implications associated with dental and mucosal infections and to ensure accurate eruption of permanent teeth.

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