Literature DB >> 34088011

Generation and characterization of three human induced pluripotent stem cell lines (iPSC) from two family members with dilated cardiomyopathy and left ventricular noncompaction (DCM-LVNC) and one healthy heterozygote sibling.

Hadas Ben-Zvi1, Nataly Korover1, Tatiana Rabinski2, Rivka Ofir2, Smadar Cohen3.   

Abstract

Autophagy serves as a master regulator of cellular homeostasis. Hence, expectedly autophagic dysfunction has been documented in many diseases such as cancer, neurodegeneration and cardiovascular disorders. A novel homozygous mutation in PLEKHM2 gene (mPLEKHM2) resulted in dilated cardiomyopathy with left ventricular noncompaction (DCM-LVNC), probably as result of impaired autophagy due to disruption of lysosomal movement assisted by PLEKHM2. Here we report a generation of three iPSC lines, four clones originated from two patients with homozygous mPLEKHM2 and two from a heterozygote sibling. All generated lines highly expressed pluripotency markers, spontaneously differentiated into three germ layers, retained the mutation after reprogramming and displayed normal karyotypes.
Copyright © 2021. Published by Elsevier B.V.

Entities:  

Year:  2021        PMID: 34088011     DOI: 10.1016/j.scr.2021.102382

Source DB:  PubMed          Journal:  Stem Cell Res        ISSN: 1873-5061            Impact factor:   2.020


  2 in total

1.  PLEKHM2 Loss-of-Function Is Associated With Dilated Cardiomyopathy.

Authors:  Jessica Atkins; Cortney Gensemer; Kimberly Foil; Jordan Morningstar; Hannia Ramos; Adrian B Van Bakel; Russell A Norris; Daniel P Judge
Journal:  Circ Genom Precis Med       Date:  2022-07-06

Review 2.  Translational potential of hiPSCs in predictive modeling of heart development and disease.

Authors:  Corrin Mansfield; Ming-Tao Zhao; Madhumita Basu
Journal:  Birth Defects Res       Date:  2022-03-09       Impact factor: 2.661

  2 in total

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