Developing iPSC models from phenylketonuria patients with varying PAH gene mutations
Phenylketonuria (PKU), a common autosomal recessive metabolic disorder, arises from diverse pathogenic variants in the phenylalanine hydroxylase (PAH) gene, causing phenylalanine accumulation and neurological impairment. The vast spectrum of over 2,200 PAH variants and frequent compound heterozygosity complicate genotype-phenotype prediction, highlighting the need for deeper mechanistic insight. We generated human induced pluripotent stem cells (iPSCs) from peripheral blood mononuclear cells of four PKU patients with distinct PAH genotypes using non-integrating Sendai viruses. The iPSC lines were validated for pluripotency, vector clearance, and genomic integrity. These patient-specific iPSCs provide a valuable platform for elucidating PKU pathophysiology and advancing personalized therapeutic development.
Authors
- Andrew B.J. Prowse (ORCID: https://orcid.org/0000-0003-4113-1720)
- Desi Veleva (ORCID: https://orcid.org/0000-0002-3989-5492)
- Md. Shahidur Rahman Chowdhury (ORCID: https://orcid.org/0000-0003-0210-6581)
- Michael Nafisinia
- Merve Ay
Institutions
- The University of Sydney (AU)
- The University of Queensland (AU)
- Westmead Institute for Medical Research (AU)
Publication Details
- Journal
- Stem Cell Research
- Published
- 2025-12-07
- DOI
- https://doi.org/10.1016/j.scr.2025.103885
- Primary Topic
- Metabolism and Genetic Disorders
- Type
- article
- Field-Weighted Citation Impact
- 0.00