Dalle Simon, Dalle Sebastiaan
Anaesthesia Department, Antwerp University Hospital, Wilrijkstraat 10, 2650 Edegem, Belgium.
RMovAnt Research Group, Department of Rehabilitation Sciences & Physiotherapy, University of Antwerp, Universiteitsplein 1, 2610 Antwerp, Belgium; Exercise Physiology Research Group, Department of Movement Sciences, University of Leuven, Tervuursevest 101, 3001 Leuven, Belgium.
Prog Lipid Res. 2025 Jul;99:101342. doi: 10.1016/j.plipres.2025.101342. Epub 2025 Jul 16.
Malignant hyperthermia (MH) is a life-threatening pharmacogenetic disorder triggered by volatile anaesthetics and depolarizing muscle relaxants. MH is characterized by excessive calcium release from the sarcoplasmic reticulum, often due to ryanodine receptor 1 (RYR1) mutations, leading to hypermetabolism, muscle rigidity and hyperthermia. While the RYR1 antagonist dantrolene remains the primary pharmacological treatment, its side effects necessitate exploration of alternative treatment options. Emerging evidence implicates the endocannabinoid system in muscle calcium homeostasis, suggesting its potential role in MH management. The endocannabinoid system comprises endogenous ligands (e.g. anandamide), cannabinoid receptors (e.g. cannabinoid receptor 1, CB1), and can modulate the calcium dynamics. CB1 activation inhibits PKA-mediated phosphorylation of RYR1 and L-type calcium channels, reducing myoplasmic calcium and muscle contractility, a mechanism that could counteract MH pathophysiology. In addition, antagonism or desensitization of the calcium channel transient receptor potential vanilloid 1 lowers calcium release from the sarcoplasmic reticulum. Preclinical studies demonstrate that CB1 agonism lowers body temperature and attenuates cardiovascular stress, aligning with MH therapeutic goals. This review synthesizes molecular insights linking endocannabinoid signaling to MH, highlighting its unexplored potential as an adjunctive therapy. Future research should validate these mechanisms in MH-specific models, including RYR1-mutant human myotubes, to translate ECS modulation into clinical practice.
恶性高热(MH)是一种由挥发性麻醉剂和去极化肌松药引发的危及生命的药物遗传学疾病。MH的特征是肌浆网过度释放钙,这通常是由于兰尼碱受体1(RYR1)突变所致,进而导致代谢亢进、肌肉强直和体温过高。虽然RYR1拮抗剂丹曲林仍然是主要的药物治疗方法,但其副作用使得有必要探索其他治疗选择。新出现的证据表明内源性大麻素系统参与肌肉钙稳态调节,提示其在MH治疗中可能发挥作用。内源性大麻素系统由内源性配体(如花生四烯乙醇胺)、大麻素受体(如大麻素受体1,CB1)组成,并可调节钙动力学。CB1激活可抑制PKA介导的RYR1和L型钙通道磷酸化,减少肌浆钙和肌肉收缩力,这一机制可能抵消MH的病理生理过程。此外,钙通道瞬时受体电位香草酸亚型1的拮抗或脱敏作用可降低肌浆网的钙释放。临床前研究表明,CB1激动可降低体温并减轻心血管应激,这与MH的治疗目标一致。本综述综合了将内源性大麻素信号与MH联系起来的分子见解,强调了其作为辅助治疗尚未被探索的潜力。未来的研究应在包括RYR1突变人肌管在内的MH特异性模型中验证这些机制,以便将内源性大麻素系统调节转化为临床实践。