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唐氏综合征退行性疾病:病例系列研究——临床特征与治疗方法

Down syndrome regression disorder, a case series: Clinical characterization and therapeutic approaches.

作者信息

Bonne Sidney, Iftimovici Anton, Mircher Clotilde, Conte Martine, Louveau Cécile, Legrand Adrien, Danset-Alexandre Charlotte, Cannarsa Costanza, Debril Alexis, Consoli Angèle, Krebs Marie-Odile, Ellul Pierre, Chaumette Boris

机构信息

Centre de Référence pour les Maladies Rares à Expression Psychiatrique, GHU Paris Psychiatrie et Neurosciences, Paris, France.

Université Paris Cité, Institute of Psychiatry and Neuroscience of Paris (IPNP), INSERM U1266, Paris, France.

出版信息

Front Neurosci. 2023 Feb 23;17:1126973. doi: 10.3389/fnins.2023.1126973. eCollection 2023.

Abstract

Down syndrome (DS) is one of the most frequent genetic disorders and represents the first cause of intellectual disability of genetic origin. While the majority of patients with DS follow a harmonious evolution, an unusual neurodevelopmental regression may occur, distinct from that described in the context of autism spectrum disorders, called down syndrome regression disorder (DSRD). Based on four patients, two males and two females, with age range between 20 and 24, treated at the Reference Center for Rare Psychiatric Disorders of the GHU Paris Psychiatry and Neurosciences [Pôle hospitalo-universitaire d'Évaluation Prévention et Innovation Thérapeutique (PEPIT)], we describe this syndrome, discuss its etiologies and propose therapeutic strategies. DSRD often occurs in late adolescence. There is a sudden onset of language disorders, loss of autonomy and daily living skills, as well as behavioral symptoms such as depression, psychosis, or catatonia. These symptoms are non-specific and lead to an overlap with other diagnostic categories, thus complicating diagnosis. The etiologies of the syndrome are not clearly identified but certain predispositions of patients with trisomy 21 have suggested an underlying immune-mediated mechanism. Symptomatic therapeutic approaches (serotonergic antidepressants, atypical antipsychotics, benzodiazepines) were not effective, and generally associated with poor tolerance. Etiological treatments, including anti-inflammatory drugs and corticosteroids, led to partial or good recovery in the four cases. Early recognition of regressive symptoms and rapid implementation of adapted treatments are required to improve the quality of life of patients and their families.

摘要

唐氏综合征(DS)是最常见的遗传性疾病之一,也是遗传性智力残疾的首要原因。虽然大多数唐氏综合征患者病情发展较为平稳,但可能会出现一种不寻常的神经发育倒退,这与自闭症谱系障碍中所描述的情况不同,称为唐氏综合征倒退障碍(DSRD)。基于在巴黎精神病学与神经科学大学医院集团罕见精神疾病参考中心[预防与治疗创新评估大学医院中心(PEPIT)]接受治疗的4例患者(2例男性和2例女性,年龄在20至24岁之间),我们描述了这种综合征,讨论了其病因并提出了治疗策略。DSRD通常发生在青春期后期。会突然出现语言障碍、自主能力和日常生活技能丧失,以及抑郁、精神病或紧张症等行为症状。这些症状不具有特异性,导致与其他诊断类别重叠,从而使诊断变得复杂。该综合征的病因尚未明确,但21三体综合征患者的某些易感性提示了一种潜在的免疫介导机制。对症治疗方法(血清素能抗抑郁药、非典型抗精神病药、苯二氮䓬类药物)无效,且通常耐受性较差。病因治疗,包括抗炎药和皮质类固醇,在这4例患者中导致了部分或良好的恢复。需要早期识别倒退症状并迅速实施适当的治疗,以提高患者及其家庭的生活质量。

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