Literature DB >> 17896311

Novel mutations in the KCNV2 gene in patients with cone dystrophy and a supernormal rod electroretinogram.

Sureka Thiagalingam1, Terri L McGee, Richard G Weleber, Michael A Sandberg, Karmen M Trzupek, Eliot L Berson, Thaddeus P Dryja.   

Abstract

PURPOSE: To identify mutations in KCNV2 in patients with a form of cone dystrophy characterized by a supernormal rod electroretinogram (ERG).
METHODS: The 2 exons and flanking intron DNA of KCNV2 from 8 unrelated patients were PCR amplified and sequenced.
RESULTS: We found 1 frameshift, 2 nonsense, 1 non-stop, and 6 missense mutations. Every patient had one or two mutations identified. Of the missense mutations, 4 affected residues were in the amino terminal region of the protein, and two in the pore region.
CONCLUSIONS: KCNV2 mutations account for most if not all cases of cone dystrophy with a supernormal rod ERG.

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Year:  2007        PMID: 17896311     DOI: 10.1080/13816810701503681

Source DB:  PubMed          Journal:  Ophthalmic Genet        ISSN: 1381-6810            Impact factor:   1.803


  18 in total

Review 1.  Electrical resonance with voltage-gated ion channels: perspectives from biophysical mechanisms and neural electrophysiology.

Authors:  Lin Ge; Xiao-dong Liu
Journal:  Acta Pharmacol Sin       Date:  2016-01       Impact factor: 6.150

2.  Two-color pupillometry in KCNV2 retinopathy.

Authors:  Frederick T Collison; Jason C Park; Gerald A Fishman; Edwin M Stone; J Jason McAnany
Journal:  Doc Ophthalmol       Date:  2019-03-29       Impact factor: 2.379

3.  Phenotypic characteristics including in vivo cone photoreceptor mosaic in KCNV2-related "cone dystrophy with supernormal rod electroretinogram".

Authors:  Ajoy Vincent; Tom Wright; Yaiza Garcia-Sanchez; Marsha Kisilak; Melanie Campbell; Carol Westall; Elise Héon
Journal:  Invest Ophthalmol Vis Sci       Date:  2013-01-30       Impact factor: 4.799

4.  Voltage-gated potassium channel KCNV2 (Kv8.2) contributes to epilepsy susceptibility.

Authors:  Benjamin S Jorge; Courtney M Campbell; Alison R Miller; Elizabeth D Rutter; Christina A Gurnett; Carlos G Vanoye; Alfred L George; Jennifer A Kearney
Journal:  Proc Natl Acad Sci U S A       Date:  2011-03-14       Impact factor: 11.205

5.  Establishing baseline rod electroretinogram values in achromatopsia and cone dystrophy.

Authors:  Isaac Wang; Naheed W Khan; Kari Branham; B Wissinger; Susanne Kohl; J R Heckenlively
Journal:  Doc Ophthalmol       Date:  2012-08-19       Impact factor: 2.379

6.  Homozygosity mapping reveals PDE6C mutations in patients with early-onset cone photoreceptor disorders.

Authors:  Alberta A H J Thiadens; Anneke I den Hollander; Susanne Roosing; Sander B Nabuurs; Renate C Zekveld-Vroon; Rob W J Collin; Elfride De Baere; Robert K Koenekoop; Mary J van Schooneveld; Tim M Strom; Janneke J C van Lith-Verhoeven; Andrew J Lotery; Norka van Moll-Ramirez; Bart P Leroy; L Ingeborgh van den Born; Carel B Hoyng; Frans P M Cremers; Caroline C W Klaver
Journal:  Am J Hum Genet       Date:  2009-07-16       Impact factor: 11.025

7.  Functional analysis of missense mutations in Kv8.2 causing cone dystrophy with supernormal rod electroretinogram.

Authors:  Katie E Smith; Susan E Wilkie; Joseph T Tebbs-Warner; Bradley J Jarvis; Linn Gallasch; Martin Stocker; David M Hunt
Journal:  J Biol Chem       Date:  2012-10-31       Impact factor: 5.157

8.  KCNV2-Associated Retinopathy: Genetics, Electrophysiology, and Clinical Course-KCNV2 Study Group Report 1.

Authors:  Michalis Georgiou; Anthony G Robson; Kaoru Fujinami; Shaun M Leo; Ajoy Vincent; Fadi Nasser; Thales Antônio Cabral De Guimarães; Samer Khateb; Nikolas Pontikos; Yu Fujinami-Yokokawa; Xiao Liu; Kazushige Tsunoda; Takaaki Hayashi; Mauricio E Vargas; Alberta A H J Thiadens; Emanuel R de Carvalho; Xuan-Thanh-An Nguyen; Gavin Arno; Omar A Mahroo; Maria Inmaculada Martin-Merida; Belen Jimenez-Rolando; Gema Gordo; Ester Carreño; Ayuso Carmen; Dror Sharon; Susanne Kohl; Rachel M Huckfeldt; Bernd Wissinger; Camiel J F Boon; Eyal Banin; Mark E Pennesi; Arif O Khan; Andrew R Webster; Eberhart Zrenner; Elise Héon; Michel Michaelides
Journal:  Am J Ophthalmol       Date:  2020-12-11       Impact factor: 5.258

Review 9.  The role of voltage-gated ion channels in visual function and disease in mammalian photoreceptors.

Authors:  Rabab Rashwan; David M Hunt; Livia S Carvalho
Journal:  Pflugers Arch       Date:  2021-07-13       Impact factor: 3.657

10.  Molecular, Cellular and Functional Changes in the Retinas of Young Adult Mice Lacking the Voltage-Gated K+ Channel Subunits Kv8.2 and K2.1.

Authors:  Xiaotian Jiang; Rabab Rashwan; Valentina Voigt; Jeanne Nerbonne; David M Hunt; Livia S Carvalho
Journal:  Int J Mol Sci       Date:  2021-05-05       Impact factor: 5.923

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