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.

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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
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article

Developing iPSC models from phenylketonuria patients with varying PAH gene mutations

Andrew B.J. Prowse, Desi Veleva, Md. Shahidur Rahman Chowdhury, Michael Nafisinia et al.
Stem Cell Research
Metabolism and Genetic Disorders
article

Developing iPSC models from phenylketonuria patients with varying PAH gene mutations

Andrew B.J. Prowse, Desi Veleva, Md. Shahidur Rahman Chowdhury, Michael Nafisinia, Merve Ay
article en

Abstract

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.

Stem Cell ResearchVol. 90
The University of Sydney (AU), The University of Queensland (AU), Westmead Institute for Medical Research (AU)
Openalex Percentile: Top 59%
Metabolism and Genetic Disorders
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Developing iPSC models from phenylketonuria patients with varying PAH gene mutations — Andrew B.J. Prowse, Desi Veleva, et al. · Stem Cell Research (2025) | TGRS Research Map | TGRS