Literature DB >> 9437321

Autosomal dominant iris hypoplasia is caused by a mutation in the Rieger syndrome (RIEG/PITX2) gene.

W L Alward1, E V Semina, J W Kalenak, E Héon, B P Sheth, E M Stone, J C Murray.   

Abstract

PURPOSE: To determine whether autosomal dominant iris hypoplasia is caused by mutations in the newly described gene for Rieger syndrome (RIEG/PITX2).
METHOD: Mutation screening and sequence analysis was performed in a single family.
RESULTS: A novel mutation in the RIEG/PITX2 gene was found in all affected but no unaffected individuals. This mutation would be expected to result in an arginine to tryptophan amino acid change in the homeodomain of solurshin, the RIEG/ITX2 gene product.
CONCLUSION: Autosomal dominant iris hypoplasia is caused by a defect in the same gene that is defective in many cases of Rieger syndrome.

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Year:  1998        PMID: 9437321     DOI: 10.1016/s0002-9394(99)80242-6

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  27 in total

1.  Analyses of the effects that disease-causing missense mutations have on the structure and function of the winged-helix protein FOXC1.

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Journal:  Am J Hum Genet       Date:  2001-03       Impact factor: 11.025

Review 2.  Glaucoma genetics.

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Authors:  M Hermina Strungaru; Tim Footz; Yi Liu; Fred B Berry; Pascal Belleau; Elena V Semina; Vincent Raymond; Michael A Walter
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5.  Potential novel mechanism for Axenfeld-Rieger syndrome: deletion of a distant region containing regulatory elements of PITX2.

Authors:  Bethany A Volkmann; Natalya S Zinkevich; Aki Mustonen; Kala F Schilter; Dmitry V Bosenko; Linda M Reis; Ulrich Broeckel; Brian A Link; Elena V Semina
Journal:  Invest Ophthalmol Vis Sci       Date:  2011-03-18       Impact factor: 4.799

Review 6.  Genetics of atrial fibrillation: implications for future research directions and personalized medicine.

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7.  Structural and biophysical insights into the ligand-free Pitx2 homeodomain and a ring dermoid of the cornea inducing homeodomain mutant.

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Journal:  Biochemistry       Date:  2012-01-06       Impact factor: 3.162

8.  Identification of dominant FOXE3 and PAX6 mutations in patients with congenital cataract and aniridia.

Authors:  Dominique Brémond-Gignac; Pierre Bitoun; Linda M Reis; Henri Copin; Jeffrey C Murray; Elena V Semina
Journal:  Mol Vis       Date:  2010-08-22       Impact factor: 2.367

9.  The canonical Wnt signaling antagonist DKK2 is an essential effector of PITX2 function during normal eye development.

Authors:  Philip J Gage; Min Qian; Dianqing Wu; Kevin I Rosenberg
Journal:  Dev Biol       Date:  2008-03-04       Impact factor: 3.582

10.  Human PRKC apoptosis WT1 regulator is a novel PITX2-interacting protein that regulates PITX2 transcriptional activity in ocular cells.

Authors:  Moulinath Acharya; David J Lingenfelter; Lijia Huang; Philip J Gage; Michael A Walter
Journal:  J Biol Chem       Date:  2009-10-02       Impact factor: 5.157

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