Nishiyama K, Masuda N, Kurisaki H
Department of Neurology, Tokyo National Hospital.
Rinsho Shinkeigaku. 1993 Jun;33(6):663-5.
A 61-year-old woman developed cerebellar ataxia as an initial symptom when she was 52 years old. Her neurological symptom was gradually followed by autonomic nervous system disturbance, pyramidal sign, and rigidity. She was diagnosed as suffering from multiple system atrophy (MSA), when she was 53 years old. Magnetic resonance imaging revealed atrophy of the cerebellum and brainstem. She was accompanied by rabbit syndrome (RS), when she was 61 years old. She had not been given any neuroleptics, which might produce RS, before she developed RS. Her regular involuntary movement was localized in lips, and its frequency was about 3 Hz. While sleeping, she did not have the involuntary movement. We had a chance to conduct pharmacological examination on RS. The administration of atropine or trihexyphenidyl did not change her symptom of RS, and the intravenous injection of levodopa deteriorated the movement. Haloperidol, sulpiride, or chlorpromazine was significantly effective on her involuntary movement of RS. These results indicated that our patient had the supersensitivity in her dopamine receptor. Such supersensitivity might result from the denervation in MSA, because she had not been administered any neuroleptics. RS is generally considered to be a kind of extrapyramidal sign, and we have not been aware of any report about RS, which levodopa deteriorated and neuroleptics improved. The mechanism of her symptom is discussed.
一名61岁女性在52岁时出现小脑共济失调作为首发症状。其神经症状逐渐伴有自主神经系统紊乱、锥体束征和强直。她在53岁时被诊断为患有多系统萎缩(MSA)。磁共振成像显示小脑和脑干萎缩。她在61岁时出现了兔综合征(RS)。在出现RS之前,她未曾服用过任何可能导致RS的抗精神病药物。她的规律性不自主运动局限于唇部,频率约为3Hz。睡眠时,她没有这种不自主运动。我们有机会对RS进行药理学检查。给予阿托品或苯海索并未改变她的RS症状,静脉注射左旋多巴使运动恶化。氟哌啶醇、舒必利或氯丙嗪对她的RS不自主运动有显著疗效。这些结果表明我们的患者多巴胺受体存在超敏反应。这种超敏反应可能是由于MSA中的去神经支配所致,因为她未曾服用过任何抗精神病药物。RS通常被认为是一种锥体外系体征,我们尚未知晓有任何关于RS的报道,即左旋多巴使其恶化而抗精神病药物使其改善。对她症状的机制进行了讨论。