Litcius/Paper detail

Substantial somatic genomic variation and selection for BCOR mutations in human induced pluripotent stem cells

Foad J. Rouhani, Xueqing Zou, Petr Danecek, Cherif Badja, Tauanne Dias Amarante, Gene Ching Chiek Koh, Qianxin Wu, Yasin Memari, Richard Durbin, Iñigo Martincorena, Andrew Bassett, Daniel J. Gaffney, Serena Nik‐Zainal

2022Nature Genetics103 citationsDOIOpen Access PDF

Abstract

We explored human induced pluripotent stem cells (hiPSCs) derived from different tissues to gain insights into genomic integrity at single-nucleotide resolution. We used genome sequencing data from two large hiPSC repositories involving 696 hiPSCs and daughter subclones. We find ultraviolet light (UV)-related damage in ~72% of skin fibroblast-derived hiPSCs (F-hiPSCs), occasionally resulting in substantial mutagenesis (up to 15 mutations per megabase). We demonstrate remarkable genomic heterogeneity between independent F-hiPSC clones derived during the same round of reprogramming due to oligoclonal fibroblast populations. In contrast, blood-derived hiPSCs (B-hiPSCs) had fewer mutations and no UV damage but a high prevalence of acquired BCOR mutations (26.9% of lines). We reveal strong selection pressure for BCOR mutations in F-hiPSCs and B-hiPSCs and provide evidence that they arise in vitro. Directed differentiation of hiPSCs and RNA sequencing showed that BCOR mutations have functional consequences. Our work strongly suggests that detailed nucleotide-resolution characterization is essential before using hiPSCs.

Topics & Concepts

BiologyReprogrammingGeneticsInduced pluripotent stem cellMutagenesisMutationGenomePhenotypeGeneEmbryonic stem cellCRISPR and Genetic EngineeringPluripotent Stem Cells ResearchRenal and related cancers