ArticleDevelopment (Cambridge, England)2021
Trp53 ablation fails to prevent microcephaly in mouse pallium with impaired minor intron splicing.
Article in Development (Cambridge, England), 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 10 citations in OpenAlex.
- WATER reveals heterochrony of molecular programs underlies developmental failure caused by minor spliceosome inhibition.bioRxiv : the preprint server for biology · 2026Article
- Minor spliceosome inhibition via ablation of its U11 snRNA gene results in multiple defects in murine spermatogenesis.Nucleic acids research · 2026Article
- Identity rather than 3D position informs splicing of rare introns in the human genome.iScience · 2026Article
- Minor intron splicing is critical for survival of lethal prostate cancer.Molecular cell · 2023Article
- P53 independent pathogenic mechanisms contribute to BubR1 microcephaly.Frontiers in cell and developmental biology · 2023Article
- The emerging significance of splicing in vertebrate development.Development (Cambridge, England) · 2022Review
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
Abstract
Minor spliceosome inhibition due to mutations in RNU4ATAC are linked to primary microcephaly. Ablation of Rnu11, which encodes a minor spliceosome snRNA, inhibits the minor spliceosome in the developing mouse pallium, causing microcephaly. There, cell cycle defects and p53-mediated apoptosis in response to DNA damage resulted in loss of radial glial cells (RGCs), underpinning microcephaly. Here, we ablated Trp53 to block cell death in Rnu11 cKO mice. We report that Trp53 ablation failed to prevent microcephaly in these double knockout (dKO) mice. We show that the transcriptome of the dKO pallium was more similar to the control compared with the Rnu11 cKO. We find aberrant minor intron splicing in minor intron-containing genes involved in cell cycle regulation, resulting in more severely impaired mitotic progression and cell cycle lengthening of RGCs in the dKO that was detected earlier than in the Rnu11 cKO. Furthermore, we discover a potential role of p53 in causing DNA damage in the developing pallium, as detection of γH2aX+ was delayed in the dKO. Thus, we postulate that microcephaly in minor spliceosome-related diseases is primarily caused by cell cycle defects.
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