Lower Cranial Dystonia with Inflated Cheeks: A Case of Dystonic Respiratory Failure

We herein report a 76-year-old woman who developed lower cranial dystonia with a peculiar appearance of cheek inflation. The patient showed strong contraction of the orbicularis oris muscles. Consequently, her cheeks were passively inflated by expiration without exit. When the dystonic attack persisted, she developed cyanosis but recovered immediately after passive mouth opening. An autopsy revealed progressive supranuclear palsy. We tentatively named this characteristic dystonia “lower cranial dystonia with inflated cheeks" because of its peculiar appearance of inflated cheeks. This dystonia can cause respiratory failure. Therefore, neurologists should recognize such dystonia as a movement disorder emergency.