用极低剂量左旋多巴治疗早发抗精神病药物诱发的迟发性运动障碍(表现为垂头综合征),成功治愈一例自闭症患者

Keiho Owada , Shuhei Ide , Akiko Shibata , Masakazu Mimaki
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引用次数: 0

摘要

背景非典型抗精神病药物通常用于控制与自闭症谱系障碍(ASD)相关的攻击性和情绪不稳定性,但也可能导致严重的副作用,如迟发性肌张力障碍(TD),这是一种通常不可逆的迟发性运动障碍亚型。垂头综合征(DHS)是锥体外系障碍的一种罕见表现,也可能是颈部肌张力障碍的一种罕见形式。病例介绍一名患有严重自闭症和智力障碍的 15 岁男子接受了利培酮(1 毫克/天)治疗,以控制冲动和攻击行为。治疗 2 个月后,患者开始出现 DHS 症状、活动减少和缺乏面部表情。根据观察到的肌张力障碍症状波动,由于停用利培酮 2 个月后症状仍然存在,患者开始接受超低剂量左旋多巴治疗(VLDT;0.5-1 毫克/千克/天)。值得注意的是,患者的面部表情、活动水平和颈部控制能力在两天内均有所改善。讨论和结论:本病例是 DHS 的一种非典型表现,即使短期使用低剂量非典型抗精神病药物,也会出现早发性 TD,这表明多巴胺能系统易受影响。因此,临床医生在使用非典型抗精神病药物治疗 ASD 时应谨慎,并警惕 TD 的发展。此外,肌张力障碍症状的波动性和对VLDT的快速反应表明,该病与多巴反应性肌张力障碍有相似之处,这为类似临床情况下的难治性疾病提供了潜在的治疗途径。
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A case of autism successfully treated with very-low-dose levodopa for early-onset antipsychotic-induced tardive movement disorder manifested by dropped head syndrome

Background

Atypical antipsychotics, often used to manage aggressiveness and mood instability associated with autism spectrum disorder (ASD), can cause severe side effects, such as tardive dystonia (TD), an often irreversible tardive movement disorder subtype. Dropped head syndrome (DHS), a rare manifestation of extrapyramidal disorders, may also represent a rare form of neck TD.

Case Presentation

A 15-year-old man with severe autism and intellectual disability was prescribed risperidone (1 mg/day) to manage impulsive and aggressive behaviors. After 2 months of treatment, the patient started showing DHS symptoms, diminished activity, and lack of facial expressions. Based on the observed fluctuation of dystonic symptoms, very-low-dose levodopa therapy (VLDT; 0.5–1 mg/kg/day) was started due to the persistence of symptoms 2 months after discontinuing risperidone. Remarkably, the patient showed improved facial expression, activity levels, and neck control within 2 days. After 7 months of treatment, the patient maintained complete improvement in overall symptoms.

Discussion and Conclusion

This case presented an atypical manifestation of DHS, with early-onset TD even after short-term exposure to low-dose atypical antipsychotics, indicating susceptibility of the dopaminergic system. Thus, clinicians should be cautious in the administration of atypical antipsychotics for ASD and vigilant in monitoring TD development. Furthermore, the fluctuation in dystonic symptoms and rapid response to VLDT indicate similarities to dopa-responsive dystonia, highlighting a potential therapeutic avenue for refractory conditions in similar clinical scenarios.

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