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MCPH3 is caused by biallelic variants in the cyclin\u2010dependent kinase 5 regulatory subunit\u2010associated protein 2 gene <jats:italic>CDK5RAP2<\/jats:italic>. In the corresponding <jats:italic>Cdk5rap2<\/jats:italic> mutant or Hertwig's anemia mouse model, congenital microcephaly as well as defects in the hematopoietic system, germ cells and eyes have been reported. The reduction in brain volume, particularly affecting gray matter, has been attributed mainly to disturbances in the proliferation and survival of early neuronal progenitors. In addition, defects in dendritic development and synaptogenesis exist that affect the excitation\u2010inhibition balance. Here, we studied proteomic changes in cerebral cortices of <jats:italic>Cdk5rap2<\/jats:italic> mutant mice.<\/jats:p><\/jats:sec><jats:sec><jats:title>Material and methods<\/jats:title><jats:p>: We used large\u2010gel two\u2010dimensional gel (2\u2010DE) electrophoresis to separate cortical proteins. 2\u2010DE gels were visualized by a trained observer on a light box. Spot changes were considered with respect to presence\/absence, quantitative variation and altered mobility.<\/jats:p><\/jats:sec><jats:sec><jats:title>Result<\/jats:title><jats:p>: We identified a reduction in more than 30 proteins that play a role in processes such as cell cytoskeleton dynamics, cell cycle progression, ciliary functions and apoptosis. These proteome changes in the MCPH3 model can be associated with various functional and morphological alterations of the developing brain.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>: Our results shed light on potential protein candidates for the disease\u2010associated phenotype reported in MCPH3.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ahg.12489","type":"journal-article","created":{"date-parts":[[2022,11,30]],"date-time":"2022-11-30T08:02:50Z","timestamp":1669795370000},"page":"50-62","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Proteome changes in autosomal recessive primary microcephaly"],"prefix":"10.1111","volume":"87","author":[{"given":"Sami","family":"Zaqout","sequence":"first","affiliation":[{"name":"Department of Basic Medical Sciences, College of Medicine, QU Health Qatar University  Doha 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