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Jpn J Clin Oncol ; 44(10): 1004-8, 2014 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-25129392

RESUMO

Juvenile polyposis syndrome is an autosomal dominant inherited disorder characterized by multiple juvenile polyps arising in the gastrointestinal tract and an increased risk of gastrointestinal cancers, specifically colon cancer. BMPR1A and SMAD4 germline mutations have been found in patients with juvenile polyposis syndrome. We identified a BMPR1A mutation, which involves a duplication of coding exon 3 (c.230+452_333+441dup1995), on multiple ligation dependent probe amplification in a patient with juvenile polyposis syndrome. The mutation causes a frameshift, producing a truncated protein (p.D112NfsX2). Therefore, the mutation is believed to be pathogenic. We also identified a duplication breakpoint in which Alu sequences are located. These results suggest that the duplication event resulted from recombination between Alu sequences. To our knowledge, partial duplication in the BMPR1A gene has not been reported previously. This is the first case report to document coding exon 3 duplication in the BMPR1A gene in a patient with juvenile polyposis syndrome.


Assuntos
Receptores de Proteínas Morfogenéticas Ósseas Tipo I/genética , Duplicação Gênica , Mutação em Linhagem Germinativa , Polipose Intestinal/congênito , Síndromes Neoplásicas Hereditárias/genética , Proteína Smad4/genética , Adolescente , Éxons , Mutação da Fase de Leitura , Humanos , Polipose Intestinal/genética , Masculino , Linhagem
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