Article

OBJECTIVE: To describe a patient with facial pain, ipsilateral facial dystonia, and phantom supernumerary teeth, beginning after resection of hypertrophic gums. She had familial sensorineural deafness. BACKGROUND: Atypical cranial dystonia subsequent to dental procedures is associated with facial pain, dysesthesias, or phantom phenomena, but not with phantom supernumerary teeth. Patients with migraine are susceptible to experience cephalic phantom phenomena after dental procedures. Wynne syndrome is a hereditary autosomal dominant disorder characterized by congenital sensorineural deafness and supernumerary teeth. METHODS: Clinical examination, computerized tomography of paranasal sinuses and facial bones, magnetic resonance imaging of the brain, cranial electrophysiological testing, and electroencephalogram. RESULTS: A 52-year-old woman with history of migraine without aura for 40 years exhibited focal right facial involuntary tonic contracture accompanying chronic severe pain over the same area after gum resection. She reported a daily sensation of having two extra upper canine teeth pressing on her tongue, simulating vampire's ("Dracula's") teeth. She had high-frequency bilateral sensorineural deafness. Her computerized tomography studies, brain magnetic resonance imaging, and cranial electrophysiological testing were normal. CONCLUSION: This patient with chronic migraine had atypical cranial dystonia beginning after a dental procedure. Her dystonia was complicated by the unusual phenomenon of phantom supernumerary teeth. This condition may be misdiagnosed as atypical or psychogenic facial pain when facial dystonia is localized or subtle and is, therefore, confused with an idiosyncratic gesture or habitual spasm. Her signs and symptoms are reminiscent of Wynne syndrome.

Langue : ANGLAIS

Tiré à part : OUI

Identifiant basis : 2001219330