Brown-Vialetto-Van Laere syndrome, a ponto-bulbar palsy with deafness, is caused by mutations in c20orf54.

نویسندگان

  • Peter Green
  • Matthew Wiseman
  • Yanick J Crow
  • Henry Houlden
  • Shelley Riphagen
  • Jean-Pierre Lin
  • F Lucy Raymond
  • Anne-Marie Childs
  • Eamonn Sheridan
  • Sian Edwards
  • Dragana J Josifova
چکیده

Brown-Vialetto-Van Laere syndrome is a rare neurological disorder with a variable age at onset and clinical course. The key features are progressive ponto-bulbar palsy and bilateral sensorineural deafness. A complex neurological phenotype with a mixed picture of upper and lower motor neuron involvement reminiscent of amyotrophic lateral sclerosis evolves with disease progression. We identified a candidate gene, C20orf54, by studying a consanguineous family with multiple affected individuals and subsequently demonstrated that mutations in this gene were the cause of disease in other, unrelated families.

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منابع مشابه

Late-onset and acute presentation of Brown-Vialetto-Van Laere syndrome in a Brazilian family

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Brown-Vialetto-van Laere syndrome; the first Turkish case.

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Pontobulbar palsy and neurosensory deafness (Brown-Vialetto-Van Laere syndrome) with possible autosomal dominant inheritance.

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Exome sequencing reveals riboflavin transporter mutations as a cause of motor neuron disease.

Brown-Vialetto-Van Laere syndrome was first described in 1894 as a rare neurodegenerative disorder characterized by progressive sensorineural deafness in combination with childhood amyotrophic lateral sclerosis. Mutations in the gene, SLC52A3 (formerly C20orf54), one of three known riboflavin transporter genes, have recently been shown to underlie a number of severe cases of Brown-Vialetto-Van ...

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عنوان ژورنال:
  • American journal of human genetics

دوره 86 3  شماره 

صفحات  -

تاریخ انتشار 2010