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 JiangPublished 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
- high glucose
- human health
- spinal cord
- diabetic rats
- risk assessment
- drug induced
- cell therapy
- single cell
- genome wide
- drinking water
- gene expression
- endothelial cells
- bone marrow
- oxidative stress
- spinal cord injury
- early onset
- dna methylation
- health risk
- depressive symptoms
- heavy metals
- genome wide identification