Department of Biomedical Sciences, University of Padova, Viale G. Colombo 3, 35121, Padova, Italy.
Leviev Heart Center, Sheba Medical Center, Tel Hashomer and Sackler School of Medicine, Tel Aviv University, Tel Aviv, Israel.
J Muscle Res Cell Motil. 2020 Sep;41(2-3):251-258. doi: 10.1007/s10974-020-09587-2. Epub 2020 Sep 9.
Homozygous calsequestrin 2 (CASQ2) point mutations leads to catecholaminergic polymorphic ventricular tachycardia: a common pathogenetic feature appears to be the drastic reduction of mutant CASQ2 in spite of normal transcription. Comparative biochemical analysis of R33Q and D307H knock in mutant mice identifies different pathogenetic mechanisms for CASQ2 degradation and different molecular adaptive mechanisms. In particular, each CASQ2 point mutation evokes specific adaptive cellular and molecular processes in each of the four adaptive pathways investigated. Thus, similar clinical phenotypes and identical cellular mechanism for cardiac arrhythmia might imply different molecular adaptive mechanisms.
纯合性肌浆网钙结合蛋白 2(CASQ2)点突变导致儿茶酚胺多形性室性心动过速:尽管转录正常,但似乎普遍存在突变型 CASQ2 急剧减少的共同发病机制。对 R33Q 和 D307H 敲入突变小鼠的比较生化分析确定了 CASQ2 降解的不同发病机制和不同的分子适应机制。特别是,每个 CASQ2 点突变在研究的四种适应途径中的每一种中引发特定的适应性细胞和分子过程。因此,相似的临床表型和相同的心律失常细胞机制可能意味着不同的分子适应机制。