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Establishment and characterization of ZJUCHi003: an induced pluripotent stem cell line from a patient with Temple-Baraitser/Zimmermann-Laband syndrome carrying KCNH1 c.1070G > A (p.R357Q) variant.

Die ChenJimei SuXueying HuangHongyu ChenTiejia JiangChunchun ZhiZuolin ZhouBing ZhangLan YuXiao-Ling Jiang
Published in: Human cell (2024)
Pathogenic variants of the KCNH1 gene can cause dominant-inherited Temple-Baraitser/Zimmermann-Laband syndrome with severe mental retardation, seizure, gingival hyperplasia and nail hypoplasia. This study established an induced pluripotent stem cell (iPSC) line using urinary cells from a girl with KCNH1 recurrent/hotspot pathogenic variant c.1070G > A (p.R357Q). The cell identity, pluripotency, karyotypic integrity, absence of reprogramming virus and mycoplasma contamination, and differential potential to three germ layers of the iPSC line, named as ZJUCHi003, were characterized and confirmed. Furthermore, ZJUCHi003-derived neurons manifested slower action potential repolarization process and wider action potential half-width than the normal neurons. This cell line will be useful for investigating the pathogenic mechanisms of KCNH1 variants-associated symptoms, as well as for evaluating novel therapeutic approaches.
Keyphrases
  • stem cells
  • copy number
  • case report
  • human health
  • diabetic rats
  • spinal cord
  • risk assessment
  • drug induced
  • single cell
  • cell therapy
  • mental health
  • early onset
  • oxidative stress
  • physical activity
  • endothelial cells