Department of Neurology, Heimer Institute for Muscle Research, BG-University Hospital Bergmannsheil, Ruhr-University Bochum, 44789 Bochum, Germany.
Department of Human Genetics, Ruhr-University Bochum, 44801 Bochum, Germany.
Stem Cell Res. 2024 Sep;79:103459. doi: 10.1016/j.scr.2024.103459. Epub 2024 Jun 10.
Here we present the generation of HIMRi006-A and HIMRi007-A Pompe disease (PD) patient derived human induced pluripotent stem cell (hiPSC) lines. HIMRi006-A represents an infantile onset disease (IOPD) phenotype caused by a homozygous c.307 T > G mutation in the GAA gene. HIMRi007-A is characterized by heterozygous mutations c.-32-13 T > G/c.1716C > G and is associated with an adult onset of disease symptoms (LOPD). Both lines are generated via lentiviral expression of OCT4, SOX2, KLF4, and c-MYC. The lines display a typical embryonic stem cell morphology, express pluripotency markers, retain a normal karyotype (46, XX/XY) and have the differentiation capacity in all three germ layers. Altogether, both lines provide a resource tool to the community for future in depth molecular studies of PD pathomechanism.
在这里,我们展示了两种亨廷顿舞蹈病(HD)患者来源的人诱导多能干细胞(hiPSC)系的生成,即 HIMRi006-A 和 HIMRi007-A。HIMRi006-A 代表一种由 GAA 基因中的纯合 c.307T>G 突变引起的婴儿发病型(IOPD)表型。HIMRi007-A 的特征是杂合突变 c.-32-13T>G/c.1716C>G,与疾病症状的成人发病(LOPD)相关。这两种系都是通过慢病毒表达 OCT4、SOX2、KLF4 和 c-MYC 生成的。这些系表现出典型的胚胎干细胞形态,表达多能性标志物,保持正常的核型(46, XX/XY),并具有三个胚层的分化能力。总之,这两种系为未来深入研究 PD 发病机制提供了一种社区资源工具。