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病例报告:“接受电休克治疗的紧张症患者中的C型尼曼-匹克病”

Case Report: "Niemann-Pick Disease Type C in a Catatonic Patient Treated With Electroconvulsive Therapy".

作者信息

van Verseveld M, Koens L H, de Koning Tom J, Derikx R L E, van Waarde J A

机构信息

Department of Psychiatry, Rijnstate Hospital, Arnhem, Netherlands.

Department of Neurology, University Medical Center Groningen, University of Groningen, Groningen, Netherlands.

出版信息

Front Psychiatry. 2021 Oct 22;12:745734. doi: 10.3389/fpsyt.2021.745734. eCollection 2021.

Abstract

We describe a case of an adolescent male with Niemann-Pick Type C (NP-C), a neurodegenerative lysosomal lipid storage disorder, who presented with recurrent catatonia which required repeated treatment with electroconvulsive therapy (ECT). During the ECT-course, seizure threshold increased substantially, leading to questions about the influence of NP-C on neuronal excitability. In this exemplary ECT-patient, NP-C was diagnosed not until after the first ECT-course when initial psychopharmacology for catatonia had failed and antipsychotics and benzodiazepines showed significant . Clinicians should be aware of NP-C in patients referred for ECT, especially in the case of treatment resistance, neurological symptoms and intolerance of psychopharmacological drugs. As was shown in our NP-C patient, ECT can be repeatedly effective for catatonic features. In the literature, effectiveness of ECT in patients with NP-C has sparsely been reported. This case demonstrates that detection of NP-C is beneficial for patients because more optimal treatment with ECT can be provided earlier without further exposure to side-effects.

摘要

我们描述了一例患有尼曼-匹克C型(NP-C)病的青少年男性病例,这是一种神经退行性溶酶体脂质贮积症,该患者反复出现紧张症,需要多次接受电休克治疗(ECT)。在ECT治疗过程中,癫痫阈值大幅升高,这引发了关于NP-C对神经元兴奋性影响的疑问。在这个典型的ECT患者中,直到第一个ECT疗程结束后才诊断出NP-C,当时针对紧张症的初始心理药理学治疗失败,抗精神病药物和苯二氮䓬类药物显示出显著效果。临床医生在为ECT转诊的患者中应意识到NP-C,特别是在治疗抵抗、神经系统症状和对心理药物不耐受的情况下。正如我们的NP-C患者所示,ECT对紧张症特征可反复有效。在文献中,ECT对NP-C患者的有效性鲜有报道。该病例表明,检测出NP-C对患者有益,因为可以更早地提供更优化的ECT治疗,而无需进一步遭受副作用。

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Systematic review of psychiatric signs in Niemann-Pick disease type C.尼曼-匹克病 C 型的精神症状系统评价。
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J Inherit Metab Dis. 2015 Jan;38(1):187-99. doi: 10.1007/s10545-014-9794-4. Epub 2014 Nov 26.
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The neuropsychiatry of inborn errors of metabolism.先天性代谢错误的神经精神病学。
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