Mary Santos Silva, Ricardo Nakamura, Marcia Rosana Arjona, Thue Peres Colferai Del Monaco, Mauricio Luiz Malito, Taís Oliveira Sampaio, Samantha Lopes de Oliveira, Juliana Silva de Almeida Magalhães, Marcela Camara Machado-Costa, Helga Cristina Almeida Silva
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引用次数: 0
摘要
STAC3基因先天性肌病和恶性高热症(MH)是神经病学和麻醉学之间的一个重要交叉,及时认识其临床特征,以及神经科和麻醉科医师之间的合作,对于早期诊断和预防不良危重事件至关重要。该基因与先天性肌病有关,首次报道为美洲原住民肌病(NAM),这是一种罕见的疾病,以畸形、挛缩、肌肉不适和脊柱侧凸为特征。作为一种罕见的药物遗传高代谢疾病,MH是由卤化药物和/或琥珀胆碱引发的,与RYR1、CACNA1S或STAC3基因的变异有关。我们的目的是分析巴西与MH相关的STAC3基因肌病病例系列的特征,并回顾以前的报道。我们报告了三例MH危机,两名男孩和一名女孩,年龄在2至15岁。所有患者都接受了卤化药物治疗,其中一人接受了琥珀胆碱治疗。2例患者进行了2 - 4次全身麻醉。这组STAC3基因突变患者的MH危机表现出不同的临床特征(表达性)和发生率(外显率)。提示有STAC3肌病的神经肌肉患者应增加对MH风险的诊断怀疑。相反,仔细评估神经肌肉患者的麻醉前因有助于限制候选基因。此外,巴西神经科医生可以向巴西神经系统疾病登记处(Registro Brasileiro de doenas Neurológicas, REDONE,葡萄牙语)通报麻醉期间有不良事件先例的神经系统患者。
STAC3 gene congenital myopathy and malignant hyperthermia: a crossroads between neurology and anesthesia.
STAC3 gene congenital myopathy and malignant hyperthermia (MH) represent an important crossroads between neurology and anesthesia, where the prompt recognition of the clinical characteristics, and the collaboration between neurologists and anesthesiologists, are essential to early diagnosis and prevention of adverse critical events. This gene is associated with a congenital myopathy first reported as Native American myopathy (NAM), a rare condition characterized by dysmorphisms, contractures, muscular complaints, and scoliosis. As a rare pharmacogenetic hypermetabolic disease, MH is triggered by halogenated agents and/or succinylcholine, linked to variants in the RYR1,CACNA1S, or STAC3 genes. Our objective was to analyze the characteristics of a Brazilian case series of STAC3 gene myopathy associated with MH and to review previous reports. We report three MH crises, in two boys and one girl, 2 to 15 years old. All of them received halogenated agents and one additionally received succinylcholine. Two patients presented two to four previous uneventful general anesthesia. The MH crises in this series of patients with STAC3 gene mutations demonstrated variable clinical characteristics (expressivity) and occurrence (penetrance). Neuromuscular patients with findings suggestive of STAC3 myopathy should increase diagnostic suspicion regarding the risk of MH. Conversely, the careful evaluation of the anesthetic antecedents of neuromuscular patients can help to restrict the candidate genes. Additionally, Brazilian neurologists can notify neurological patients with antecedents of adverse events during anesthesia to the Brazilian Registry of Neurological Diseases (Registro Brasileiro de Doenças Neurológicas, REDONE, in Portuguese).
期刊介绍:
Arquivos de Neuro-Psiquiatria is the official journal of the Brazilian Academy of Neurology. The mission of the journal is to provide neurologists, specialists and researchers in Neurology and related fields with open access to original articles (clinical and translational research), editorials, reviews, historical papers, neuroimages and letters about published manuscripts. It also publishes the consensus and guidelines on Neurology, as well as educational and scientific material from the different scientific departments of the Brazilian Academy of Neurology.
The ultimate goals of the journal are to contribute to advance knowledge in the areas of Neurology and Neuroscience, and to provide valuable material for training and continuing education for neurologists and other health professionals working in the area. These goals might contribute to improving care for patients with neurological diseases. We aim to be the best Neuroscience journal in Latin America within the peer review system.