Literature DB >> 20006823

Leber congenital amaurosis: clinical correlations with genotypes, gene therapy trials update, and future directions.

Daniel C Chung1, Elias I Traboulsi.   

Abstract

Leber congenital amaurosis comprises a group of early onset childhood retinal dystrophies, characterized by vision loss, nystagmus, and severe retinal dysfunction. To date, 15 causative genes have been identified that account for the heterogeneous presentation and clinical course. Knowledge of key aspects of phenotype and clinical course can contribute to the determination of a precise genetic etiology using genetic testing. Gene-based therapies are emerging, and knowledge of a patient's genotype is essential. A review of clinical presentation and disease course, their correlation to specific genotypes, and underlying physiological mechanisms, coupled with the latest results of human gene therapy trials, will assist the clinician in patient diagnosis and counseling.

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Year:  2009        PMID: 20006823     DOI: 10.1016/j.jaapos.2009.10.004

Source DB:  PubMed          Journal:  J AAPOS        ISSN: 1091-8531            Impact factor:   1.220


  41 in total

1.  Chemical chaperone TUDCA preserves cone photoreceptors in a mouse model of Leber congenital amaurosis.

Authors:  Tao Zhang; Wolfgang Baehr; Yingbin Fu
Journal:  Invest Ophthalmol Vis Sci       Date:  2012-06-05       Impact factor: 4.799

Review 2.  Gene therapy and genome surgery in the retina.

Authors:  James E DiCarlo; Vinit B Mahajan; Stephen H Tsang
Journal:  J Clin Invest       Date:  2018-06-01       Impact factor: 14.808

3.  Neuronal deficiency of ARV1 causes an autosomal recessive epileptic encephalopathy.

Authors:  Elizabeth E Palmer; Kelsey E Jarrett; Rani K Sachdev; Fatema Al Zahrani; Mais Omar Hashem; Niema Ibrahim; Hugo Sampaio; Tejaswi Kandula; Rebecca Macintosh; Rajat Gupta; Donna M Conlon; Jeffrey T Billheimer; Daniel J Rader; Kouichi Funato; Christopher J Walkey; Chang Seok Lee; Christine Loo; Susan Brammah; George Elakis; Ying Zhu; Michael Buckley; Edwin P Kirk; Ann Bye; Fowzan S Alkuraya; Tony Roscioli; William R Lagor
Journal:  Hum Mol Genet       Date:  2016-06-06       Impact factor: 6.150

Review 4.  Paradigm Shifts in Ophthalmic Diagnostics.

Authors:  J Sebag; Alfredo A Sadun; Eric A Pierce
Journal:  Trans Am Ophthalmol Soc       Date:  2016-08

Review 5.  Photoreceptor Cilia and Retinal Ciliopathies.

Authors:  Kinga M Bujakowska; Qin Liu; Eric A Pierce
Journal:  Cold Spring Harb Perspect Biol       Date:  2017-10-03       Impact factor: 10.005

Review 6.  The Status of RPE65 Gene Therapy Trials: Safety and Efficacy.

Authors:  Eric A Pierce; Jean Bennett
Journal:  Cold Spring Harb Perspect Med       Date:  2015-01-29       Impact factor: 6.915

Review 7.  The chick eye in vision research: An excellent model for the study of ocular disease.

Authors:  C Ellis Wisely; Javed A Sayed; Heather Tamez; Chris Zelinka; Mohamed H Abdel-Rahman; Andy J Fischer; Colleen M Cebulla
Journal:  Prog Retin Eye Res       Date:  2017-06-28       Impact factor: 21.198

8.  Current concepts in the treatment of retinitis pigmentosa.

Authors:  Maria A Musarella; Ian M Macdonald
Journal:  J Ophthalmol       Date:  2010-10-11       Impact factor: 1.909

9.  Functional and behavioral restoration of vision by gene therapy in the guanylate cyclase-1 (GC1) knockout mouse.

Authors:  Shannon E Boye; Sanford L Boye; Jijing Pang; Renee Ryals; Drew Everhart; Yumiko Umino; Andy W Neeley; Joseph Besharse; Robert Barlow; William W Hauswirth
Journal:  PLoS One       Date:  2010-06-25       Impact factor: 3.240

10.  Novel mutations in CRB1 and ABCA4 genes cause Leber congenital amaurosis and Stargardt disease in a Swedish family.

Authors:  Frida Jonsson; Marie S Burstedt; Ola Sandgren; Anna Norberg; Irina Golovleva
Journal:  Eur J Hum Genet       Date:  2013-02-27       Impact factor: 4.246

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