PUBLICATION

The XRN1-regulated RNA helicase activity of YTHDC2 ensures mouse fertility independently of m6A recognition

Authors
Li, L., Krasnykov, K., Homolka, D., Gos, P., Mendel, M., Fish, R.J., Pandey, R.R., Pillai, R.S.
ID
ZDB-PUB-220320-9
Date
2022
Source
Molecular Cell   82(9): 1678-1690.e12 (Journal)
Registered Authors
Fish, Richard
Keywords
DExH helicase, MEIOC, RBM46, RNA helicase, XRN1, YTH, YTHDC2, m(6)A, oogenesis, spermatogenesis
MeSH Terms
  • Animals
  • Fertility/genetics
  • Mammals/metabolism
  • Meiosis
  • Mice
  • RNA/genetics
  • RNA Helicases*/genetics
  • RNA Helicases*/metabolism
  • Zebrafish*/genetics
PubMed
35305312 Full text @ Mol. Cell
Abstract
The functional consequence of N6-methyladenosine (m6A) RNA modification is mediated by "reader" proteins of the YTH family. YTH domain-containing 2 (YTHDC2) is essential for mammalian fertility, but its molecular function is poorly understood. Here, we identify U-rich motifs as binding sites of YTHDC2 on 3' UTRs of mouse testicular RNA targets. Although its YTH domain is an m6A-binder in vitro, the YTH point mutant mice are fertile. Significantly, the loss of its 3'→5' RNA helicase activity causes mouse infertility, with the catalytic-dead mutation being dominant negative. Biochemical studies reveal that the weak helicase activity of YTHDC2 is enhanced by its interaction with the 5'→3' exoribonuclease XRN1. Single-cell transcriptomics indicate that Ythdc2 mutant mitotic germ cells transition into meiosis but accumulate a transcriptome with mixed mitotic/meiotic identity that fail to progress further into meiosis. Finally, our demonstration that ythdc2 mutant zebrafish are infertile highlights its conserved role in animal germ cell development.
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Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping