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.

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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
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article

Genome editing of a low-penetrance albinism-associated variant in TYR in patient-derived pluripotent stem cells

Antony Adamson, Steven Paul Woods, Polly Downton, Nicola Bates et al.
1 citations
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CRISPR and Genetic Engineering
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article

Genome editing of a low-penetrance albinism-associated variant in TYR in patient-derived pluripotent stem cells

Antony Adamson, Steven Paul Woods, Polly Downton, Nicola Bates, Panagiotis I. Sergouniotis
article en
1 citations

Abstract

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.

Stem Cell ResearchVol. 89
Manchester Academic Health Science Centre (GB), University of Manchester (GB), Manchester University NHS Foundation Trust (GB)
Openalex Percentile: Top 22%
CRISPR and Genetic Engineering
0.81
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