نتایج جستجو برای: lebers congenital amaurosis

تعداد نتایج: 120868  

Journal: :The British journal of ophthalmology 1987
C I Phillips A M Levy M Newton N L Stokoe

Of 99 children in the Royal Blind School, Edinburgh (which serves Scotland and part of N E England), 15 had optic atrophy (hydrocephalus 4, intracranial haemorrhage 2, prematurity 2, fetal distress 2, birth asphyxia 2, cerebral atrophy 1, cardiac arrest during hernia operation 1, and leukaemia 1). Fourteen had congenital cataract, 12 congenital retinal aplasia (Leber's congenital amaurosis) and...

Arezou Karamzade, Elaheh Alavinejad, Hassan Khojasteh Jafari, Mohammad Keramatipour, Mohammad Saberi, Mona Entezam, Yeganeh Eshaghkhani, Zahra Golchehre,

Background: Leber congenital amaurosis (LCA) is a rare inherited retinal disease causing severe visual impairment in infancy. It has been reported that 9-15% of LCA cases have mutations in CRB1 gene. The complex of CRB1 protein with other associated proteins affects the determination of cell polarity, orientation, and morphogenesis of photoreceptors. Here, we report three novel pathogenic varia...

Journal: :Archives of ophthalmology 2010
Martin McKibbin Manir Ali Moin D Mohamed Adam P Booth Fiona Bishop Bishwanath Pal Kelly Springell Yasmin Raashid Hussain Jafri Chris F Inglehearn

OBJECTIVES To report the genetic basis of Leber congenital amaurosis (LCA) in northern Pakistan and to describe the phenotype. METHODS DNA from 14 families was analyzed using single-nucleotide polymorphism and microsatellite genotyping and direct sequencing to determine the genes and mutations involved. The history and examination findings from 64 affected individuals were analyzed to show ge...

2011
Adeel Ahmad Shakeela Daud Naseebullah Kakar Gudrun Nürnberg Peter Nürnberg Masroor Ellahi Babar Michaela Thoenes Christian Kubisch Jamil Ahmad Hanno Jörn Bolz

PURPOSE To determine the cause of Leber congenital amaurosis (LCA) and developmental cataracts in a consanguineous Pakistani family. METHODS The diagnosis was established in all affected individuals of a Pakistani LCA family by medical history, funduscopy, and standard ERG. We performed genome-wide linkage analysis for mapping the disease locus in this family. RESULTS Congenitally severely ...

2011
Donna S. Mackay Arundhati Dev Borman Phillip Moradi Robert H. Henderson Zheng Li Genevieve A. Wright Naushin Waseem Mamatha Gandra Dorothy A. Thompson Shomi S. Bhattacharya Graham E. Holder Andrew R. Webster Anthony T. Moore

PURPOSE To identify patients with autosomal recessive retinal dystrophy caused by mutations in the gene, retinal dehydrogenase 12 (RDH12), and to report the associated phenotype. METHODS After giving informed consent, all patients underwent full clinical evaluation. Patients were selected for mutation analysis based upon positive results from the Asper Ophthalmics Leber congenital amaurosis a...

2013
Mirjana A. Janicijevic-Petrovic Tatjana S. Sarenac-Vulovic Katarina M. Janicijevic Dragan I. Vujic Dejan D. Vulovic

CONFLICT OF INTEREST none declared. INTRODUCTION Authors are from reference documentation to archive at Organization of Federation of blind and visually impaired in central Serbia (Kragujevac), by retrograde analysis, of 2007-2012, comprehend two groups by etiology-clinical characteristics of congenital blindness and visually impaired, caused infection or non infection example. AIM to analy...

Journal: :Molecular vision 1999
R Koenekoop A L Pina M Loyer J Davidson J Robitaille I Maumenee J Tombran-Tink

PURPOSE Leber congenital amaurosis (LCA) has been mapped to chromosome 17p13.1. From the candidate genes mapped to this region, thus far, only Retinal Guanylate Cyclase (RetGC), has been found to have pathogenic LCA mutations, in families from North African origin. However, early reports, demonstrated eight LCA families linked to 17p13.1, but only four of them showed mutations in RetGC. Mapped ...

Journal: :Molecular therapy : the journal of the American Society of Gene Therapy 2004
Nadine S Dejneka Enrico M Surace Tomas S Aleman Artur V Cideciyan Arkady Lyubarsky Andrey Savchenko T Michael Redmond Waixing Tang Zhangyong Wei Tonia S Rex Ernest Glover Albert M Maguire Edward N Pugh Samuel G Jacobson Jean Bennett

The congenital retinal blindness known as Leber congenital amaurosis (LCA) can be caused by mutations in the RPE65 gene. RPE65 plays a critical role in the visual cycle that produces the photosensitive pigment rhodopsin. Recent evidence from human studies of LCA indicates that earlier rather than later intervention may be more likely to restore vision. We determined the impact of in utero deliv...

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