Your browser doesn't support javascript.
loading
Mostrar: 20 | 50 | 100
Resultados 1 - 1 de 1
Filtrar
Más filtros

Banco de datos
Tipo de estudio
Tipo del documento
Asunto de la revista
País de afiliación
Intervalo de año de publicación
1.
Am J Med Genet A ; 185(7): 2180-2189, 2021 07.
Artículo en Inglés | MEDLINE | ID: mdl-33878224

RESUMEN

A 57-year-old man with a family history of aortic aneurysm was found, during assessment of unexplained fever, to have an infrarenal aortic aneurysm requiring immediate repair. Dilatation of popliteal and iliac arteries was also present. Progressive aortic root dilatation with aortic regurgitation was documented from 70 years leading to valve-sparing aortic root replacement at 77 years, at which time genetic studies identified a likely pathogenic FBN1 missense variant c.6916C > T (p.Arg2306Cys) in exon 56. The proband's lenses were normally positioned and the Marfan syndrome (MFS) systemic score was 0/20. Cascade genetic testing identified 15 other family members with the FBN1 variant, several of whom had unsuspected aortic root dilatation; none had ectopia lentis or MFS systemic score ≥ 7. Segregation analysis resulted in reclassification of the FBN1 variant as pathogenic. The combination of thoracic aortic aneurysm and dissection (TAAD) and a pathogenic FBN1 variant in multiple family members allowed a diagnosis of MFS using the revised Ghent criteria. At 82 years, the proband's presenting abdominal aortic aneurysm was diagnosed retrospectively to have resulted from IgG4-related inflammatory aortopathy.


Asunto(s)
Aneurisma de la Aorta/genética , Fibrilina-1/genética , Predisposición Genética a la Enfermedad , Síndrome de Marfan/genética , Anciano , Anciano de 80 o más Años , Aneurisma de la Aorta/complicaciones , Aneurisma de la Aorta/patología , Exones , Femenino , Pruebas Genéticas , Humanos , Inmunoglobulina G/genética , Masculino , Síndrome de Marfan/complicaciones , Síndrome de Marfan/fisiopatología , Persona de Mediana Edad , Mutación
SELECCIÓN DE REFERENCIAS
DETALLE DE LA BÚSQUEDA