3rd Department of Internal Medicine, Semmelweis University, Budapest, Hungary.
Immunopharmacol Immunotoxicol. 2013 Apr;35(2):304-6. doi: 10.3109/08923973.2013.766801. Epub 2013 Feb 15.
The authors report a case of rare chloroquine cardiotoxicity mimicking connective tissue disease heart involvement in a 56-year-old woman with mixed connective tissue disease (MCTD) manifested suddenly as third degree A-V block with QT(c) interval prolongation and short torsade de pointes runs ultimately degenerating into ventricular fibrillation. Immunological tests suggested an MCTD flare, implying that cardiac arrest had resulted from myocardial involvement by MCTD. However, QT(c) prolongation is not a characteristic of cardiomyopathy caused by connective tissue disease, unless anti-Ro/SSA positivity is present, but that was not the case. Therefore, looking for another cause of QT(c) prolongation the possibility of chloroquine cardiotoxicity emerged, which the patient had been receiving for almost two years in supramaximal doses. Biopsy of the deltoid muscle was performed, because in chloroquine toxicity, specific lesions are present both in the skeletal muscle and in the myocardium, and electron microscopy revealed the accumulation of cytoplasmic curvilinear bodies, which are specific to antimalarial-induced myocyte damage and are absent in all other muscle diseases, except neuronal ceroid lipofuscinosis. Thus, the diagnosis of chloroquine cardiotoxicity was established. It might be advisable to supplement the periodic ophthalmological examination, which is currently the only recommendation for patients on long-term chloroquine therapy, with ECG screening.
作者报告了一例罕见的氯喹心脏毒性,表现为 56 岁混合性结缔组织病(MCTD)女性患者出现类似结缔组织病心脏受累的三度房室传导阻滞,伴有 QT(c)间期延长和短扭转性室性心动过速,最终发展为心室颤动。免疫检测提示 MCTD 发作,提示心肌受累导致心脏骤停。然而,QT(c)延长并非结缔组织病引起的心肌病的特征,除非存在抗 Ro/SSA 阳性,但事实并非如此。因此,寻找其他导致 QT(c)延长的原因时,出现了氯喹心脏毒性的可能性,该患者在超最大剂量下已接受了近两年的治疗。对三角肌进行了活检,因为在氯喹毒性中,骨骼肌和心肌都存在特定的病变,电镜显示细胞质卷曲体的积累,这是抗疟原虫引起的心肌损伤的特异性表现,而在其他肌肉疾病中均不存在,除神经元蜡样脂褐质沉积症外。因此,确立了氯喹心脏毒性的诊断。对于长期接受氯喹治疗的患者,除了目前唯一推荐的定期眼科检查外,还可能需要进行心电图筛查。