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We systematically prioritized likely pathogenic CNVs (LP\u2010CNVs) in <jats:italic>PRKN<\/jats:italic> and examined their association with SCZ and ASD.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>In total, 3014 SCZ cases (96.9%), 1205 ASD cases (97.5%), and 2671 controls (98.5%) passed quality control. We found that monoallelic carriers of LP\u2010CNVs in <jats:italic>PRKN<\/jats:italic> were common (70\/6890, 1.02%) and were not at higher risk of SCZ (<jats:italic>p<\/jats:italic>\u2009=\u20090.29) or ASD (<jats:italic>p<\/jats:italic>\u2009=\u20090.72). We observed that the distribution pattern of LP\u2010CNVs in the Japanese population was consistent with those in other populations. We also identified a patient diagnosed with SCZ and early\u2010onset Parkinson's disease carrying biallelic pathogenic CNVs in <jats:italic>PRKN<\/jats:italic>. The absence of Parkinson's symptoms in 10 other monoallelic carriers of the same pathogenic CNV further reflects the lack of effect of monoallelic pathogenic variants in <jats:italic>PRKN<\/jats:italic> in the absence of a second hit.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The present findings suggest that monoallelic CNVs in <jats:italic>PRKN<\/jats:italic> do not confer a significant risk for SCZ or ASD. However, further studies to investigate the association between biallelic CNVs in <jats:italic>PRKN<\/jats:italic> and SCZ and ASD are warranted.<\/jats:p><\/jats:sec>","DOI":"10.1002\/npr2.12370","type":"journal-article","created":{"date-parts":[[2023,11,2]],"date-time":"2023-11-02T05:52:00Z","timestamp":1698904320000},"page":"42-50","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Association between copy number variations in parkin (<i>PRKN<\/i>) and schizophrenia and autism spectrum disorder: A case\u2013control study"],"prefix":"10.1002","volume":"44","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8982-4318","authenticated-orcid":false,"given":"Tzuyao","family":"Lo","sequence":"first","affiliation":[{"name":"Department of Psychiatry Nagoya University Graduate School of Medicine  Nagoya 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