ArticleBone research2014
High-throughput screening of mouse gene knockouts identifies established and novel skeletal phenotypes.
Article in Bone research, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 66 papers.
What it found
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Who cites it
66 citing papers in PubMed.
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- Klotho Protein: A Multifaceted Guardian of Healthy Aging and Its Therapeutic Potential.International journal of nanomedicine · 2025Review
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- Skeletal Phenotyping of Period-1-Deficient Melatonin-Proficient Mice.Journal of pineal research · 2024Article
- Integrative Analysis Reveals the Diverse Effects of 3D Stiffness upon Stem Cell Fate.International journal of molecular sciences · 2023Article
- Leucine Repeat Rich Kinase 1 Controls Osteoclast Activity by Managing Lysosomal Trafficking and Secretion.Biology · 2023Article
- Skeletal phenotypes in secreted frizzled-related protein 4 gene knockout mice mimic skeletal architectural abnormalities in subjects with Pyle's disease from SFRP4 mutations.Bone research · 2023Article
- The International Mouse Phenotyping Consortium: comprehensive knockout phenotyping underpinning the study of human disease.Nucleic acids research · 2023Article
- The role of Meteorin-like in skeletal development and bone fracture healing.Journal of orthopaedic research : official publication of the Orthopaedic Research Society · 2022Article
- Review
- Sfrp4 and the Biology of Cortical Bone.Current osteoporosis reports · 2022Review
- Review
- Estrogen receptor alpha and NFATc1 bind to a bone mineral density-associated SNP to repress WNT5B in osteoblasts.American journal of human genetics · 2022Article
- Mice LackingDiabetes, metabolic syndrome and obesity : targets and therapy · 2022Article
- Deletion of Gremlin-2 alters estrous cyclicity and disrupts female fertility in mice†.Biology of reproduction · 2021Article
- Proteomic Comparison of Bone Marrow Derived Osteoblasts and Mesenchymal Stem Cells.International journal of molecular sciences · 2021Article
- Deciphering the LRRK code: LRRK1 and LRRK2 phosphorylate distinct Rab proteins and are regulated by diverse mechanisms.The Biochemical journal · 2021Article
6 more citing papers are in PubMed but not listed here.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
9 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Screening gene function in vivo is a powerful approach to discover novel drug targets. We present high-throughput screening (HTS) data for 3 762 distinct global gene knockout (KO) mouse lines with viable adult homozygous mice generated using either gene-trap or homologous recombination technologies. Bone mass was determined from DEXA scans of male and female mice at 14 weeks of age and by microCT analyses of bones from male mice at 16 weeks of age. Wild-type (WT) cagemates/littermates were examined for each gene KO. Lethality was observed in an additional 850 KO lines. Since primary HTS are susceptible to false positive findings, additional cohorts of mice from KO lines with intriguing HTS bone data were examined. Aging, ovariectomy, histomorphometry and bone strength studies were performed and possible non-skeletal phenotypes were explored. Together, these screens identified multiple genes affecting bone mass: 23 previously reported genes (Calcr, Cebpb, Crtap, Dcstamp, Dkk1, Duoxa2, Enpp1, Fgf23, Kiss1/Kiss1r, Kl (Klotho), Lrp5, Mstn, Neo1, Npr2, Ostm1, Postn, Sfrp4, Slc30a5, Slc39a13, Sost, Sumf1, Src, Wnt10b), five novel genes extensively characterized (Cldn18, Fam20c, Lrrk1, Sgpl1, Wnt16), five novel genes with preliminary characterization (Agpat2, Rassf5, Slc10a7, Slc26a7, Slc30a10) and three novel undisclosed genes coding for potential osteoporosis drug targets.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.