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Peer-reviewed veterinary case report

CRISPR-Cas9-Generated TXNDC15 c.560delA Homozygous Mouse Model Exhibits Meckel-Gruber Syndrome Phenotype.

Journal:
Genesis (New York, N.Y. : 2000)
Year:
2026
Authors:
Liu, Yang et al.
Affiliation:
Shenzhen Maternity and Child Healthcare Hospital · China

Abstract

To determine whether TXNDC15 variation causes Meckel-Gruber syndrome (MKS), we assessed the pathogenicity of the frameshift variant c.560delA. A CRISPR-Cas9 generated mouse model carrying the equivalent Txndc15 c.512delA mutation was analyzed at embryonic day 15.5. Homozygous Txndc15embryos displayed the complete MKS phenotype-fetal lethality, exencephaly, omphalocele, post-axial polydactyly, and polycystic kidneys-together with markedly reduced TXNDC15 protein in brain, liver, and kidney. These findings confirm TXNDC15 as a bona fide MKS disease gene.

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Original publication: https://pubmed.ncbi.nlm.nih.gov/41518077/