Mowat-Wilson syndrome (MWS) is a genetic disease which simultaneously exhibits various clinical symptoms including characteristic facial features such as external ear malformation, widely spaced eyes, thick and dense eyebrows, pointed chin, elongated face, and prominent nasal bridge, mild to moderate intellectual disabilities, microcephaly, Hirschsprung disease, tapered fingers, hypoplastic corpus callosum, hypospadias, and epilepsy. Regarding characteristic intraoral features, there have been only a few reports on delayed eruption of teeth, high arched palate, cleft uvula, submucous cleft palate, and malalignment, but there is no report describing the morphological characteristics of teeth.
In this study, we performed dental treatment of two patients with MWS and found shared morphological characteristics of teeth. The patients were a 17-year-old male and a 23-year-old male. Neither patient could cooperate in dental treatment because of intellectual disabilities and both were difficult to treat by conventional methods. We measured the size of teeth on CT images and dental radiograms obtained during oral examination and dental treatment under general anesthesia or using extracted wisdom teeth. Even taking into account the error of measurement, the molars of both patients showed greater overall lengths than the Japanese average and, in particular, mild radiculomegaly. In addition, histopathological examination of extracted wisdom teeth revealed that the cause of radiculomegaly was cement hyperplasia. In the present study, radiculomegaly was observed in both patients, which may be an intraoral finding characteristic of MWS.
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