Literature DB >> 21358631

Mutations in origin recognition complex gene ORC4 cause Meier-Gorlin syndrome.

Duane L Guernsey1, Makoto Matsuoka, Haiyan Jiang, Susan Evans, Christine Macgillivray, Mathew Nightingale, Scott Perry, Meghan Ferguson, Marissa LeBlanc, Jean Paquette, Lysanne Patry, Andrea L Rideout, Aidan Thomas, Andrew Orr, Chris R McMaster, Jacques L Michaud, Cheri Deal, Sylvie Langlois, Duane W Superneau, Sandhya Parkash, Mark Ludman, David L Skidmore, Mark E Samuels.   

Abstract

Meier-Gorlin syndrome is a rare autosomal recessive genetic condition whose primary clinical hallmarks include small stature, small external ears and small or absent patellae. Using marker-assisted mapping in multiple families from a founder population and traditional coding exon sequencing of positional candidate genes, we identified three different mutations in the gene encoding ORC4, a component of the eukaryotic origin recognition complex, in five individuals with Meier-Gorlin syndrome. In two such individuals that were negative for mutations in ORC4, we found potential mutations in ORC1 and CDT1, two other genes involved in origin recognition. ORC4 is well conserved in eukaryotes, and the yeast equivalent of the human ORC4 missense mutation was shown to be pathogenic in functional assays of cell growth. This is the first report, to our knowledge, of a germline mutation in any gene of the origin recognition complex in a vertebrate organism.

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Year:  2011        PMID: 21358631     DOI: 10.1038/ng.777

Source DB:  PubMed          Journal:  Nat Genet        ISSN: 1061-4036            Impact factor:   38.330


  33 in total

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Authors:  J P Fryns
Journal:  Clin Dysmorphol       Date:  1998-07       Impact factor: 0.816

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Journal:  Genes Dev       Date:  2000-07-15       Impact factor: 11.361

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Review 9.  The origin recognition complex protein family.

Authors:  Bernard P Duncker; Igor N Chesnokov; Brendan J McConkey
Journal:  Genome Biol       Date:  2009-03-17       Impact factor: 13.583

10.  QuantiSNP: an Objective Bayes Hidden-Markov Model to detect and accurately map copy number variation using SNP genotyping data.

Authors:  Stefano Colella; Christopher Yau; Jennifer M Taylor; Ghazala Mirza; Helen Butler; Penny Clouston; Anne S Bassett; Anneke Seller; Christopher C Holmes; Jiannis Ragoussis
Journal:  Nucleic Acids Res       Date:  2007-03-06       Impact factor: 16.971

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  85 in total

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Journal:  Cold Spring Harb Perspect Biol       Date:  2014-01-01       Impact factor: 10.005

4.  MCM5: a new actor in the link between DNA replication and Meier-Gorlin syndrome.

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Review 8.  Small organelle, big responsibility: the role of centrosomes in development and disease.

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Review 9.  Genetic evaluation of short stature.

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Review 10.  Complex Phenotypes: Mechanisms Underlying Variation in Human Stature.

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