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March 5, 2026Stem Cell Research0 citationsOpen Access

Generation of a pluripotent embryonic stem cell TAFAZZIN hESC model (WAe009-A-3H) of Barth syndrome

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YLYau Chung LowMurdoch Children's Research InstituteCMCameron L. McKnightMurdoch Children's Research InstituteDEDavid A. ElliottMurdoch Children's Research Institute

Key Points

  • The goal is to create a human embryonic stem cell model of Barth syndrome to study its mechanisms and explore treatment options.
  • Generated a human embryonic stem cell model using CRISPR/Cas9 gene editing.
  • Developed a compound heterozygous variant in TAFAZZIN resulting in exon 3 skipping.
  • Assessed pluripotency through morphology, marker expression, and trilineage potential.
  • The generated TAFAZZIN Δ3 C15 cell line shows typical pluripotent stem cell characteristics.
  • The cell line maintains a normal karyotype, ensuring genetic stability.
  • Potential applications include studying mitochondrial cardiomyopathy and screening preclinical treatments.

Abstract

Barth syndrome is among the most common mitochondrial diseases presenting with cardiomyopathy. We have generated a human embryonic stem cell (hESC) model of Barth syndrome ( TAFAZZIN Δ3 C15) in a female background (H9 hESC) using CRISPR/Cas9 gene editing, with compound heterozygous variants in TAFAZZIN that result in exon 3 skipping in all stable transcripts. This cell line displayed characteristics consistent with pluripotent stem cells, including typical colony morphology, expression of pluripotency markers, trilineage potential, and a normal karyotype. This TAFAZZIN Δ3 C15 line could be used for investigation of disease mechanisms in mitochondrial cardiomyopathy and preclinical therapeutic screening.

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Cite This Study

Low et al. (2026) studied this question.

synapsesocial.com/papers/69a91e57d6127c7a504c233ahttps://doi.org/10.1016/j.scr.2026.103948
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