Genome editing of a low-penetrance albinism-associated variant in TYR in patient-derived pluripotent stem cells
TYR encodes tyrosinase, the enzyme catalysing the initial steps of melanin biosynthesis in melanocytes and retinal pigment epithelia (RPE). TYR c.1205G>A (p.Arg402Gln) is a common genetic variant associated with several pigmentation traits. Notably, when this variant is encountered in specific haplotypic backgrounds in the homozygous state, it predisposes to albinism. We generated an induced pluripotent stem cell (iPSC) line from an affected individual carrying such a homozygous genotype (UMANi255-A), and then used CRISPR-Cas9 to correct the TYR c.1205G>A variant (UMANi255-A-1). The resulting iPSC lines demonstrate capacity for multi-lineage differentiation, providing a useful in vitro model for studying pigmentation biology.
Authors
- Antony Adamson (ORCID: https://orcid.org/0000-0002-5408-0013)
- Steven Paul Woods (ORCID: https://orcid.org/0000-0001-6873-5722)
- Polly Downton (ORCID: https://orcid.org/0000-0002-1617-6153)
- Nicola Bates (ORCID: https://orcid.org/0000-0001-5875-548X)
- Panagiotis I. Sergouniotis
Institutions
- Manchester Academic Health Science Centre (GB)
- University of Manchester (GB)
- Manchester University NHS Foundation Trust (GB)
Publication Details
- Journal
- Stem Cell Research
- Published
- 2025-10-22
- DOI
- https://doi.org/10.1016/j.scr.2025.103855
- Citations
- 1
- Primary Topic
- CRISPR and Genetic Engineering
- Type
- article
- Field-Weighted Citation Impact
- 0.81