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Ophthalmologica ; 225(4): 228-35, 2011.
Artículo en Inglés | MEDLINE | ID: mdl-21412020

RESUMEN

PURPOSE: To describe the genotype and phenotype in a 9-year-old boy with bilateral retinopathy. METHODS: The patient, his healthy (by history) nonconsanguineous parents and his sister were examined by best-corrected visual acuity, matrix frequency doubling technology, monocular static field analysis, fundus autofluorescence imaging, optical coherence tomography, Ganzfeld electroretinography (ERG), pattern ERG, multifocal ERG, electro-oculography and genotyping of the BEST1 gene. RESULTS: The patient presented with an Arden ratio of 1.25, an unremarkable ERG and fluorescent yellow deposits distributed throughout the fundus suggestive of autosomal recessive bestrophinopathy (ARB). Genotyping revealed a homozygous nonsense mutation in BEST1 (p.R200X). The parents and the sister, who were heterozygous mutation carriers, presented with normal ophthalmological function. CONCLUSIONS: ARB is a rare retinal disorder. We contribute a novel patient report indicative of ARB, assessed by clinical examination and confirmed by genotyping of BEST1, to the short list of ARB cases in the literature.


Asunto(s)
ADN/genética , Proteínas del Ojo/genética , Mutación , Enfermedades de la Retina/genética , Niño , Canales de Cloruro/genética , Electrooculografía , Electrorretinografía , Femenino , Predisposición Genética a la Enfermedad , Genotipo , Humanos , Masculino , Linaje , Retina/fisiopatología , Enfermedades de la Retina/diagnóstico , Enfermedades de la Retina/fisiopatología , Tomografía de Coherencia Óptica , Agudeza Visual
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