Miyaoka T, Miura S, Seno H, Inagaki T, Horiguchi J
Department of Psychiatry, Shimane Medical University, Izumo, Japan.
Eur J Neurol. 2003 Nov;10(6):727-9. doi: 10.1046/j.1468-1331.2003.00668.x.
Jaw-opening dystonia (oromandibular dystonia with jaw-opening; Brueghel's syndrome) is a rare condition, and only a limited number of cases have been reported in the literature. However, many patients may remain undiscovered or misdiagnosed, like a patient described previously. A case (40-year-old man) of jaw-opening dystonia (oromandibular dystonia with jaw-opening; Brueghel's syndrome) is reported. In this case, brain anomalies, cavum septi pellucidi and Verga's ventricle, were observed on magnetic resonance imaging of the brain. This case and a review of the literature indicate the presence of organic factors in the etiology of Brueghel's syndrome. The etiological relationship of brain anomalies in Bruegel's syndrome is discussed.