Two iPSC lines with a heterozygous frameshift mutation in the floating-harbour syndrome locus of the SRCAP gene
We present two CRISPR/Cas9-modified human iPSC lines with a heterozygous frameshift mutation (NM_006662.3:c.7300_7301insA) in the FLHS-locus of the SRCAP gene, which is associated with Floating-Harbor syndrome, a congenital neurodevelopmental disorder with symptoms including short stature and intellectual disability. The iPSCs express the pluripotency markers OCT4, SOX2, NANOG and TRA 1-60. They show differentiation into cells from all 3 germ layers, no chromosomal abnormalities and no off-target mutations in the tested regions. The mutation leads to a stop codon previously found in patients. Thus, either cell line can serve as disease-specific model for studying SRCAP in the context of FLHS.
Authors
- Lisa Hagenau (ORCID: https://orcid.org/0000-0003-2563-2728)
- Lars R. Jensen (ORCID: https://orcid.org/0000-0002-9608-8224)
- Scott V. Edwards (ORCID: https://orcid.org/0000-0003-2535-6217)
- Md Faruk Hossain (ORCID: https://orcid.org/0000-0003-3214-3068)
- Ana Tzvetkova (ORCID: https://orcid.org/0000-0003-1576-6511)
- Andreas W. Kuß (ORCID: https://orcid.org/0000-0002-9401-4627)
- Jennifer M. Rhode
- B. Nowack
Institutions
- Universitätsmedizin Greifswald (DE)
- Universität Greifswald (DE)
Publication Details
- Journal
- Stem Cell Research
- Published
- 2025-05-08
- DOI
- https://doi.org/10.1016/j.scr.2025.103730
- Primary Topic
- Congenital heart defects research
- Type
- article
- Field-Weighted Citation Impact
- 0.00
Funders
- European Regional Development Fund