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Structural copy number variants (CNVs) have been implicated in LSL without extra\u2010cardiac features; however, non\u2010penetrance and variable expressivity have created uncertainty over the use of CNV analyses in such patients. High\u2010density SNP microarray genotyping data were used to infer large, likely\u2010pathogenic, autosomal CNVs in a cohort of 1,139 probands with LSL and their families. CNVs were molecularly confirmed and the medical records of individual carriers reviewed. The gene content of novel CNVs was then compared with public CNV data from CHD patients. Large CNVs (&gt;1\u2009MB) were observed in 33 probands (\u223c3%). Six of these were\n                      <jats:italic>de novo<\/jats:italic>\n                      and 14 were not observed in the only available parent sample. Associated cardiac phenotypes spanned a broad spectrum without clear predilection. Candidate CNVs were largely non\u2010recurrent, associated with heterozygous loss of copy number, and overlapped known CHD genomic regions. Novel CNV regions were enriched for cardiac development genes, including seven that have not been previously associated with human CHD. CNV analysis can be a clinically useful and molecularly informative tool in LSLs without obvious extra\u2010cardiac defects, and may identify a clinically relevant genomic disorder in a small but important proportion of these individuals.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ajmg.a.38309","type":"journal-article","created":{"date-parts":[[2017,6,27]],"date-time":"2017-06-27T08:14:41Z","timestamp":1498551281000},"page":"2176-2188","source":"Crossref","is-referenced-by-count":17,"title":["Assessment of large copy number variants in patients with apparently isolated congenital left\u2010sided cardiac lesions reveals clinically relevant genomic events"],"prefix":"10.1002","volume":"173","author":[{"given":"Neil A.","family":"Hanchard","sequence":"first","affiliation":[{"name":"Department of Human and Molecular Genetics Baylor College of Medicine Houston Texas"},{"name":"USDA\/ARS\/Children's Nutrition Research Center Baylor College of Medicine 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