ArticlePLoS genetics2023
MIWI N-terminal RG motif promotes efficient pachytene piRNA production and spermatogenesis independent of LINE1 transposon silencing.
Article in PLoS genetics, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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Who cites it
15 citing papers in PubMed, 14 citations in OpenAlex.
- Article
- The pachytene-specific lncRNANon-coding RNA research · 2026Article
- Review
- Positive Selection Targeted Primate Genes that Encode Transposable Element Repressors.Genome biology and evolution · 2026Article
- Spermatogenesis Beyond DNA: Integrated RNA Control of the Epitranscriptome and Three-Dimensional Genome Architecture.Current issues in molecular biology · 2026Review
- Spatiotemporal control of PIWI compartmentalization by mitochondrial scaffolds defines pachytene piRNA pathway organization.bioRxiv : the preprint server for biology · 2025Article
- PIWI proteins tether the piRNA biogenesis machinery to mitochondria during mammalian spermatogenesis.The EMBO journal · 2025Article
- Direct identification ofbioRxiv : the preprint server for biology · 2025Article
- Tdrd15 is dispensable for male fertility and spermatogenesis in the golden hamster.European journal of medical research · 2025Article
- CARF regulates the alternative splicing and piwi/piRNA complexes during mouse spermatogenesis through PABPC1.Acta biochimica et biophysica Sinica · 2024Article
- PNLDC1 catalysis and postnatal germline function are required for piRNA trimming, LINE1 silencing, and spermatogenesis in mice.PLoS genetics · 2024Article
- Inherited defects of piRNA biogenesis cause transposon de-repression, impaired spermatogenesis, and human male infertility.Nature communications · 2024Article
- MIWI N-terminal arginines orchestrate generation of functional pachytene piRNAs and spermiogenesis.Nucleic acids research · 2024Article
- MIWI arginines orchestrate generation of functional pachytene piRNAs and spermiogenesis.bioRxiv : the preprint server for biology · 2024Article
- PNLDC1 catalysis and postnatal germline function are required for piRNA trimming, LINE1 silencing, and spermatogenesis in mice.bioRxiv : the preprint server for biology · 2023Article
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Authors and funding
10 authors at 3 institutions in 2 countries.
Funding
Abstract
PIWI proteins and their associated piRNAs act to silence transposons and promote gametogenesis. Murine PIWI proteins MIWI, MILI, and MIWI2 have multiple arginine and glycine (RG)-rich motifs at their N-terminal domains. Despite being known as docking sites for the TDRD family proteins, the in vivo regulatory roles for these RG motifs in directing PIWI in piRNA biogenesis and spermatogenesis remain elusive. To investigate the functional significance of RG motifs in mammalian PIWI proteins in vivo, we genetically engineered an arginine to lysine (RK) point mutation of a conserved N-terminal RG motif in MIWI in mice. We show that this tiny MIWI RG motif is indispensable for piRNA biogenesis and male fertility. The RK mutation in the RG motif disrupts MIWI-TDRKH interaction and impairs enrichment of MIWI to the intermitochondrial cement (IMC) for efficient piRNA production. Despite significant overall piRNA level reduction, piRNA trimming and maturation are not affected by the RK mutation. Consequently, MiwiRK mutant mice show chromatoid body malformation, spermatogenic arrest, and male sterility. Surprisingly, LINE1 transposons are effectively silenced in MiwiRK mutant mice, indicating a LINE1-independent cause of germ cell arrest distinctive from Miwi knockout mice. These findings reveal a crucial function of the RG motif in directing PIWI proteins to engage in efficient piRNA production critical for germ cell progression and highlight the functional importance of the PIWI N-terminal motifs in regulating male fertility.
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