Kohli Utkarsh, Aziz Zaid, Beaser Andrew D, Nayak Hemal M
Division of Pediatric Cardiology, Department of Pediatrics, Pritzker School of Medicine of the University of Chicago, Chicago, Illinois.
Center for Arrhythmia Care, Heart & Vascular Center, Pritzker School of Medicine of the University of Chicago, Chicago, Illinois.
Pacing Clin Electrophysiol. 2019 Aug;42(8):1146-1154. doi: 10.1111/pace.13668. Epub 2019 Apr 17.
We report a 17-year-old boy with a large RYR2 exon 3 deletion who has a severe catecholaminergic polymorphic ventricular tachycardia (CPVT) phenotype characterized by refractoriness to both nadolol and flecainide which has previously not been reported in this subgroup of CPVT patients. Treatment options in a patient like ours are therefore limited and sympathectomy and implantable cardioverter-defibrillator implantation should be considered early in the treatment course as was done in this patient. In contrast to other CPVT patients who do not usually have structural cardiac abnormalities, these patients are at a high risk of developing left ventricular noncompaction or dilated cardiomyopathy and therefore might benefit from cardiac imaging at regular intervals.
我们报告了一名17岁男孩,其RYR2基因第3外显子存在大片段缺失,患有严重的儿茶酚胺能多形性室性心动过速(CPVT)表型,其特征为对纳多洛尔和氟卡尼均耐药,此前在该亚组CPVT患者中尚未有此报道。因此,像我们这位患者这样的治疗选择有限,交感神经切除术和植入式心脏复律除颤器植入术应在治疗过程早期就予以考虑,本患者就是如此。与其他通常无心脏结构异常的CPVT患者不同,这些患者发生左心室心肌致密化不全或扩张型心肌病的风险很高,因此可能受益于定期的心脏成像检查。