Malignant Hyperthermia Investigation Unit, Department of Anesthesiology and Pain Management, University Health Network, Toronto, ON, Canada.
Malignant Hyperthermia Unit, Department of Anesthesiology, Pain and Intensive Care, Federal University of São Paulo, São Paulo, Brazil.
Br J Anaesth. 2024 Oct;133(4):759-767. doi: 10.1016/j.bja.2024.05.046. Epub 2024 Aug 5.
Malignant hyperthermia susceptibility (MHS) designates individuals at risk of developing a hypermetabolic reaction triggered by halogenated anaesthetics or the depolarising neuromuscular blocking agent suxamethonium. Over the past few decades, beyond the operating theatre, myopathic manifestations impacting daily life are increasingly recognised as a prevalent phenomenon in MHS patients. At the request of the European Malignant Hyperthermia Group, we reviewed the literature and gathered the opinion of experts to define MHS-related myopathy as a distinct phenotype expressed across the adult lifespan of MHS patients unrelated to anaesthetic exposure; this serves to raise awareness about non-anaesthetic manifestations, potential therapies, and management of MHS-related myopathy. We focused on the clinical presentation, biochemical and histopathological findings, and the impact on patient well-being. The spectrum of symptoms of MHS-related myopathy encompasses muscle cramps, stiffness, myalgias, rhabdomyolysis, and weakness, with a wide age range of onset mainly during adulthood. Histopathological analysis can reveal nonspecific abnormalities suggestive of RYR1 involvement, while metabolic profiling reflects altered energy metabolism in MHS muscle. Myopathic manifestations can significantly impact patient quality of life and lead to functional limitations and socio-economic burden. While currently available therapies can provide symptomatic relief, there is a need for further research into targeted treatments addressing the underlying pathophysiology. Counselling early after establishing the MHS diagnosis, followed by multidisciplinary management involving various medical specialties, is crucial to optimise patient care.
恶性高热易感性(MHS)指的是个体在接触卤代麻醉剂或去极化神经肌肉阻滞剂琥珀酰胆碱后,易发生代谢亢进反应的风险。在过去的几十年中,除了手术室之外,MHS 患者日常生活中影响生活的肌病表现也越来越被认为是一种普遍现象。应欧洲恶性高热组的要求,我们回顾了文献并汇集了专家的意见,将与 MHS 相关的肌病定义为一种与麻醉暴露无关的、在 MHS 患者整个成年期表现出的独特表型;这有助于提高对非麻醉表现、潜在治疗方法和 MHS 相关肌病管理的认识。我们重点关注临床表现、生化和组织病理学发现以及对患者健康状况的影响。MHS 相关肌病的症状谱包括肌肉痉挛、僵硬、肌痛、横纹肌溶解和无力,发病年龄范围广泛,主要在成年期。组织病理学分析可以显示提示 RYR1 参与的非特异性异常,而代谢谱反映了 MHS 肌肉中能量代谢的改变。肌病表现可显著影响患者的生活质量,并导致功能受限和社会经济负担。虽然目前可用的治疗方法可以提供症状缓解,但需要进一步研究针对潜在病理生理的靶向治疗。在确立 MHS 诊断后尽早进行咨询,然后进行涉及多个医学专业的多学科管理,对于优化患者护理至关重要。