Literature DB >> 8588587

Autosomal dominant optic nerve colobomas, vesicoureteral reflux, and renal anomalies.

L A Schimmenti1, M E Pierpont, B L Carpenter, C E Kashtan, M R Johnson, W B Dobyns.   

Abstract

We describe a father and 3 sons with optic nerve colobomas, vesicoureteral reflux, and renal anomalies. The youngest son had congenital renal failure and ultimately underwent renal transplantation. The father and one son had high frequency hearing loss. There were no other affected relatives. We conclude that the association of optic nerve colobomas, renal anomalies, and vesicoureteral reflux comprises a unique autosomal dominant syndrome. Molecular investigations have determined this disorder to be associated with a single nucleotide deletion in the PAX2 gene.

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Year:  1995        PMID: 8588587     DOI: 10.1002/ajmg.1320590217

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  17 in total

1.  Localisation of a 10q breakpoint within the PAX2 gene in a patient with a de novo t(10;13) translocation and optic nerve coloboma-renal disease.

Authors:  K Narahara; E Baker; S Ito; Y Yokoyama; S Yu; D Hewitt; G R Sutherland; M R Eccles; R I Richards
Journal:  J Med Genet       Date:  1997-03       Impact factor: 6.318

2.  The orphan nuclear receptor Tlx regulates Pax2 and is essential for vision.

Authors:  R T Yu; M Y Chiang; T Tanabe; M Kobayashi; K Yasuda; R M Evans; K Umesono
Journal:  Proc Natl Acad Sci U S A       Date:  2000-03-14       Impact factor: 11.205

3.  Clinical utility gene card for: renal coloboma (Papillorenal) syndrome.

Authors:  Matthew Bower; Michael Eccles; Laurence Heidet; Lisa A Schimmenti
Journal:  Eur J Hum Genet       Date:  2011-02-16       Impact factor: 4.246

4.  Further delineation of renal-coloboma syndrome in patients with extreme variability of phenotype and identical PAX2 mutations.

Authors:  L A Schimmenti; H E Cunliffe; L A McNoe; T A Ward; M C French; H H Shim; Y H Zhang; W Proesmans; A Leys; K A Byerly; S R Braddock; M Masuno; K Imaizumi; K Devriendt; M R Eccles
Journal:  Am J Hum Genet       Date:  1997-04       Impact factor: 11.025

Review 5.  Renal coloboma syndrome.

Authors:  Lisa A Schimmenti
Journal:  Eur J Hum Genet       Date:  2011-06-08       Impact factor: 4.246

Review 6.  PAX2 in human kidney malformations and disease.

Authors:  Lyndsay A Harshman; Patrick D Brophy
Journal:  Pediatr Nephrol       Date:  2011-12-03       Impact factor: 3.714

7.  Two novel EGFP insertion alleles reveal unique aspects of Pax2 function in embryonic and adult kidneys.

Authors:  Abdul Soofi; Inna Levitan; Gregory R Dressler
Journal:  Dev Biol       Date:  2012-03-03       Impact factor: 3.582

8.  Papillorenal syndrome-causing missense mutations in PAX2/Pax2 result in hypomorphic alleles in mouse and human.

Authors:  Ramakrishna P Alur; Camasamudram Vijayasarathy; Jacob D Brown; Mohit Mehtani; Ighovie F Onojafe; Yuri V Sergeev; Elangovan Boobalan; Marypat Jones; Ke Tang; Haiquan Liu; Chun-Hong Xia; Xiaohua Gong; Brian P Brooks
Journal:  PLoS Genet       Date:  2010-03-05       Impact factor: 5.917

9.  The prevalence of PAX2 mutations in patients with isolated colobomas or colobomas associated with urogenital anomalies.

Authors:  H E Cunliffe; L A McNoe; T A Ward; K Devriendt; H G Brunner; M R Eccles
Journal:  J Med Genet       Date:  1998-10       Impact factor: 6.318

10.  Groucho suppresses Pax2 transactivation by inhibition of JNK-mediated phosphorylation.

Authors:  Yi Cai; Patrick D Brophy; Inna Levitan; Stefano Stifani; Gregory R Dressler
Journal:  EMBO J       Date:  2003-10-15       Impact factor: 11.598

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