Huckell V F, Staniloff H M, Britt B A, Waxman M B, Morch J E
Circulation. 1978 Nov;58(5):916-25. doi: 10.1161/01.cir.58.5.916.
Malignant hyperthermia is a disease resulting from defective cellular membranes, usually presenting as drug-induced pyrexic crises. We describe four patients with life threatening ventricular arrhythmias or chest pain in the absence of pyrexic crises. Three presented with life threatening arrhythmias and the fourth with severe atypical chest pain. Two patients had a family history of multiple sudden deaths. Resting CKs were elevated in three patients while CK-MB was elevated in one. Resting ECGs were abnormal in three. Three patients had recurrent ventricular tachycardia, two had recurrent ventricular fibrillation and multiple cardiac arrests. Cardiac catheterization showed abnormal left ventricular wall motion in two and minimal mitral valve prolapse in one while all had normal coronary arteries. Thallium-201 myocardial imaging demonstrated large perfusion defects in the patient with electrocardiographic Q waves and normal coronary arteries. Myocardial involvement has been demonstrated by clinical, electrocardiographic, hemodynamic, angiographic and myocardial imaging abnormalities. Malignant arrhythmias occurred in these patients in the absence of pyrexic crises or drug admininstration. Abnormal calcium release in the myocardium, as documented in skeletal muscle membranes, may be a unifying concept for the various manifestations described.
恶性高热是一种由细胞膜缺陷引起的疾病,通常表现为药物诱导的发热危象。我们描述了4例在无发热危象情况下出现危及生命的室性心律失常或胸痛的患者。其中3例表现为危及生命的心律失常,第4例表现为严重的非典型胸痛。2例患者有多次猝死的家族史。3例患者静息肌酸激酶(CK)升高,1例肌酸激酶同工酶(CK-MB)升高。3例患者静息心电图异常。3例患者出现反复室性心动过速,2例出现反复心室颤动和多次心脏骤停。心脏导管检查显示2例左心室壁运动异常,1例有轻度二尖瓣脱垂,而所有患者冠状动脉均正常。铊-201心肌显像显示,心电图有Q波且冠状动脉正常的患者存在大面积灌注缺损。临床、心电图、血流动力学、血管造影和心肌显像异常均证实了心肌受累。这些患者在无发热危象或未用药的情况下发生了恶性心律失常。如骨骼肌膜所记录的,心肌中异常的钙释放可能是所描述的各种表现的一个统一概念。