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Am J Hum Genet ; 75(5): 822-31, 2004 Nov.
Artículo en Inglés | MEDLINE | ID: mdl-15372378

RESUMEN

Motor neuron diseases (MNDs) are a group of neurodegenerative disorders with involvement of upper and/or lower motor neurons, such as amyotrophic lateral sclerosis (ALS), spinal muscular atrophy (SMA), progressive bulbar palsy, and primary lateral sclerosis. Recently, we have mapped a new locus for an atypical form of ALS/MND (atypical amyotrophic lateral sclerosis [ALS8]) at 20q13.3 in a large white Brazilian family. Here, we report the finding of a novel missense mutation in the vesicle-associated membrane protein/synaptobrevin-associated membrane protein B (VAPB) gene in patients from this family. Subsequently, the same mutation was identified in patients from six additional kindreds but with different clinical courses, such as ALS8, late-onset SMA, and typical severe ALS with rapid progression. Although it was not possible to link all these families, haplotype analysis suggests a founder effect. Members of the vesicle-associated proteins are intracellular membrane proteins that can associate with microtubules and that have been shown to have a function in membrane transport. These data suggest that clinically variable MNDs may be caused by a dysfunction in intracellular membrane trafficking.


Asunto(s)
Esclerosis Amiotrófica Lateral/genética , Proteínas de Unión al Calcio/genética , Cromosomas Humanos Par 20/genética , Expresión Génica , Atrofia Muscular Espinal/genética , Adulto , Secuencia de Aminoácidos , Brasil , Células Cultivadas , Mapeo Cromosómico , Cartilla de ADN , Efecto Fundador , Proteínas Fluorescentes Verdes , Humanos , Proteínas de Interacción con los Canales Kv , Persona de Mediana Edad , Modelos Moleculares , Datos de Secuencia Molecular , Mutación Missense/genética , Linaje , Estructura Terciaria de Proteína , Alineación de Secuencia , Análisis de Secuencia de ADN , Proteínas de Transporte Vesicular
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