Genetic compensation in a human genomic disorder.

Carelle-Calmels, Nadège; Saugier-Veber, Pascale; Girard-Lemaire, Françoise; Rudolf, Gabrielle; Doray, Bérénice; Guérin, Eric; Kuhn, Pierre; Arrivé, Mathilde et al. · N Engl J Med · 2009

case_report · Level V

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Abstract

Cytogenetic studies of the parents of a girl with the DiGeorge (or velocardiofacial) syndrome, who carried a deletion at 22q11.2, revealed an unexpected rearrangement of both 22q11.2 regions in the unaffected father. He carried a 22q11.2 deletion on one copy of chromosome 22 and a reciprocal 22q11.2 duplication on the other copy of chromosome 22. Genetic compensation, which is consistent with the normal phenotype of the father, was shown through quantitative-expression analyses of genes located within the genetic region associated with the DiGeorge syndrome. This finding has implications for genetic counseling and represents a case of genetic compensation in a human genomic disorder.

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