Objectives:Incontinentia pigmenti is a rare X- linked dominant disease which affects the ectodermal tissues that usually lethal in males. The condition is chacterized by four cutaneous stages which frequently associated with neurological, dental, cardiac and ocular defects. In general positive family history is present. Methods: 16 year old female case was treated in pedodontics department clinics.All decayed teeth filled with amalgam and dental prosthesis prepared. Results:The case was presented with the dental , clinical, radiographical and dermatological findings. Hypodontia and delayed eruption of the primary and permanent dentition were present in the case. Crown morphology at the permanent dentition was showing the microdontic appearence. The patient had also cutaneous manifestations, mental retardation, neurological and ocular defects. A positive family history was not present in this case.Conclusions: In this disease, poor oral health conditions can lead to risks for patient . It is therefore of great importance that these patients are referred to a paediatric dentist for oral examination and treatment.

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