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March 6, 2026Stem Cell Research0 citationsOpen Access

Generation and characterization of induced pluripotent stem cell (iPSC) lines from patients affected with Tay-Sachs and Sandhoff disease

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VJVukasin M. JovanovicCCCatherine Z. ChenCTCamilo Toro

Key Points

  • This research aims to generate and characterize iPSC lines from patients with Tay-Sachs and Sandhoff diseases to explore disease mechanisms and potential therapies.
  • Generation of iPSC lines from four patients (three with Tay-Sachs, one with Sandhoff disease).
  • Characterization of mutations in the HEXA and HEXB genes.
  • Evaluation of iPSC lines for their potential application in disease modeling.
  • Four distinct iPSC lines were successfully created and characterized.
  • Tay-Sachs patients exhibited homozygous or complex heterozygous mutations in the HEXA gene.
  • A Sandhoff patient was found to carry a heterozygous mutation in the HEXB gene.

Abstract

Tay-Sachs and Sandhoff diseases, are sphingolipidoses caused by rare genetic mutations in the HEXA and HEXB genes, that encode the alpha and beta subunits of lysosomal hexosaminidase, respectively. Here, we report the generation and characterization of three Tay-Sachs and one Sandhoff iPSC lines derived from patients with late-onset disease carrying mutations at the HEXA or HEXB gene. The Tay-Sachs patients carried either homozygous or complex heterozygous mutations in the HEXA gene. The Sandhoff patient carried a heterozygous mutation in the HEXB gene. These four iPSC lines will serve as a valuable resource for the development of in vitro lysosomal storage disease models and therapeutic drug development.

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Cite This Study

Jovanovic et al. (2026) studied this question.

synapsesocial.com/papers/69aa7008531e4c4a9ff59649https://doi.org/10.1016/j.scr.2026.103949
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