ArticlePLoS biology2019
Targeted genetic screening in mice through haploid embryonic stem cells identifies critical genes in bone development.
Article in PLoS biology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers.
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Who cites it
21 citing papers in PubMed, 30 citations in OpenAlex.
- TRIM37 interacts with TRIM28 to maintain primordial germ cell identity during migration.Cell research · 2026Article
- hnRNPK condensates facilitate enhancer-promoter looping and RNA polymerase II recruitment.Nature genetics · 2026Article
- Metabolic Imbalance Triggers Adaptive Remodeling to Accelerate Diploidization in Murine Haploid Embryonic Stem Cells.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Effect of Small Molecules on Blastocyst Development and Outgrowth Establishment of Bovine Haploid Parthenogenetic Embryos.Animals : an open access journal from MDPI · 2026Article
- Identification and functional validation of a novel FBN1 variant in a Marfan syndrome family using a zebrafish model.BMC genomics · 2025Article
- GRAMMAR-Lambda Delivers Efficient Understanding of the Genetic Basis for Head Size in Catfish.Biology · 2025Article
- In vitro gametogenesis: Towards competent oocytes: Limitations and future improvements for generating oocytes from pluripotent stem cells in culture.BioEssays : news and reviews in molecular, cellular and developmental biology · 2025Review
- Glial maturation factor-β deficiency prevents oestrogen deficiency-induced bone loss by remodelling the actin network to suppress adipogenesis of bone marrow mesenchymal stem cells.Cell death & disease · 2024Article
- A CRISPR/RfxCas13d-mediated strategy for efficient RNA knockdown in mouse embryonic development.Science China. Life sciences · 2024Article
- Development and application of haploid embryonic stem cells.Stem cell research & therapy · 2024Review
- The role of Clec11a in bone construction and remodeling.Frontiers in endocrinology · 2024Review
- Strategies of Macrophages to Maintain Bone Homeostasis and Promote Bone Repair: A Narrative Review.Journal of functional biomaterials · 2022Review
- IRX5 promotes adipogenesis of hMSCs by repressing glycolysis.Cell death discovery · 2022Article
- The RNA-binding protein Musashi2 governs osteoblast-adipocyte lineage commitment by suppressing PPARγ signaling.Bone research · 2022Article
- The Relationship Between Bone and Reproductive Hormones Beyond Estrogens and Androgens.Endocrine reviews · 2021Review
- Osteocyte transcriptome mapping identifies a molecular landscape controlling skeletal homeostasis and susceptibility to skeletal disease.Nature communications · 2021Article
- Applications ofStem cells international · 2021Review
- Review
- Dysregulated Gene Expression of Imprinted and X-Linked Genes: A Link to Poor Development of Bovine Haploid Androgenetic Embryos.Frontiers in cell and developmental biology · 2021Article
- Progress of orthopaedic research in China over the last decade.Journal of orthopaedic translation · 2020Article
Corrections and comments
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Authors and funding
11 authors at 4 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Mutagenic screening is powerful for identifying key genes involved in developmental processes. However, such screens are successful only in lower organisms. Here, we develop a targeted genetic screening approach in mice through combining androgenetic haploid embryonic stem cells (AG-haESCs) and clustered regularly interspaced palindromic repeats/CRISPR-associated protein 9 (CRISPR-Cas9) technology. We produced a mutant semi-cloned (SC) mice pool by oocyte injection of AG-haESCs carrying constitutively expressed Cas9 and an single guide RNA (sgRNA) library targeting 72 preselected genes in one step and screened for bone-development-related genes through skeletal analysis at birth. This yielded 4 genes: Zic1 and Clec11a, which are required for bone development, and Rln1 and Irx5, which had not been previously considered. Whereas Rln1-/- mice exhibited small skeletal size only at birth, Irx5-/- mice showed skeletal abnormalities both in postnatal and adult phases due to decreased bone mass and increased bone marrow adipogenesis. Mechanistically, iroquois homeobox 5 (IRX5) promotes osteoblastogenesis and inhibits adipogenesis by suppressing peroxisome proliferator activated receptor γ (PPARγ) activation. Thus, AG-haESC-mediated functional mutagenic screening opens new avenues for genetic interrogation of developmental processes in mice.
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