Transposon insertion causes ctnnb2 transcript instability that results in the maternal effect zebrafish ichabod (ich) mutation DOI Creative Commons
Zsombor Varga, Ferenc Kagan,

S. Maegawa

и другие.

bioRxiv (Cold Spring Harbor Laboratory), Год журнала: 2025, Номер unknown

Опубликована: Март 4, 2025

Abstract The maternal-effect mutation ichabod ( ich ) results in ventralized zebrafish embryos due to impaired induction of the dorsal canonical Wnt-signaling pathway. While previous studies linked phenotype reduced ctnnb2 transcript levels, causative remained unidentified. Using long-read sequencing, we discovered that stems from insertion a non-autonomous CMC-Enhancer/Suppressor-mutator (CMC-EnSpm) transposon 3’UTR gene. Through reporter assays, demonstrate while wild type mRNAs exhibit remarkably high stability throughout early stages development, dramatically reduces stability. Genome-wide mapping CMC-EnSpm transposons across multiple strains also indicated ongoing transposition activity genome. Our findings not only resolve molecular basis but highlight continuing mutagenic potential endogenous and reveal unexpected aspects maternal regulation during development.

Язык: Английский

Transposon insertion causes ctnnb2 transcript instability that results in the maternal effect zebrafish ichabod (ich) mutation DOI Creative Commons
Zsombor Varga, Ferenc Kagan,

S. Maegawa

и другие.

bioRxiv (Cold Spring Harbor Laboratory), Год журнала: 2025, Номер unknown

Опубликована: Март 4, 2025

Abstract The maternal-effect mutation ichabod ( ich ) results in ventralized zebrafish embryos due to impaired induction of the dorsal canonical Wnt-signaling pathway. While previous studies linked phenotype reduced ctnnb2 transcript levels, causative remained unidentified. Using long-read sequencing, we discovered that stems from insertion a non-autonomous CMC-Enhancer/Suppressor-mutator (CMC-EnSpm) transposon 3’UTR gene. Through reporter assays, demonstrate while wild type mRNAs exhibit remarkably high stability throughout early stages development, dramatically reduces stability. Genome-wide mapping CMC-EnSpm transposons across multiple strains also indicated ongoing transposition activity genome. Our findings not only resolve molecular basis but highlight continuing mutagenic potential endogenous and reveal unexpected aspects maternal regulation during development.

Язык: Английский

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