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[Idiopathic rabbit syndrome: a case report].
1Department of Neurology, Juntendo University Urayasu Hospital, Chiba, Japan.
No to Shinkei = Brain and Nerve
|December 20, 1999
Summary
This study details a unique case of idiopathic oromandibular tremor, mimicking rabbit syndrome. Tiapride effectively treated the tremor, suggesting dopamine pathway involvement in this rare neurological condition.
Area of Science:
- Neurology
- Movement Disorders
Background:
- Idiopathic oromandibular tremor is a rare condition affecting jaw and perioral muscles.
- Rabbit syndrome is a specific type of tremor characterized by rapid, rhythmic jaw movements.
Observation:
- A 36-year-old woman presented with isolated oromandibular tremor without other neurological deficits.
- Electromyography (EMG) showed synchronized discharges in jaw muscles at 6 Hz.
- No abnormalities were found in MRI or EEG.
Findings:
- The patient's tremor responded well to tiapride, a dopamine receptor antagonist.
- Other medications including diazepam, trihexyphenydil, levodopa, and a beta-blocker were ineffective.
- The tremor presentation was clinically indistinguishable from neuroleptic-induced rabbit syndrome.
Implications:
- The efficacy of tiapride suggests a role for dopaminergic dysfunction in the basal ganglia.
- This case expands the understanding of idiopathic oromandibular tremor and its potential pathophysiology.
- Further research into basal ganglia circuits may elucidate mechanisms of oromandibular tremors.