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Symptomatic mosaicism for a novel FBN1 splice site variant in a parent causing inherited neonatal Marfan syndrome.
Postma, Julianne K; Altamirano-Diaz, Luis; Rupar, C Anthony; Siu, Victoria M.
Afiliación
  • Postma JK; Division of Medical Genetics, Department of Pediatrics, Children's Hospital of Eastern Ontario, Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.
  • Altamirano-Diaz L; Division of Pediatric Cardiology, Department of Pediatrics, London Health Sciences Centre, Schulich School of Medicine and Dentistry, Western University, London, Ontario, Canada.
  • Rupar CA; Departments of Pathology and Laboratory Medicine, Pediatrics and Biochemistry, Children's Health Research Institute, London Health Sciences Centre, Western University, London, Ontario, Canada.
  • Siu VM; Division of Medical Genetics, Department of Pediatrics, London Health Sciences Centre, Schulich School of Medicine and Dentistry, Western University, London, Ontario, Canada.
Am J Med Genet A ; 185(8): 2507-2513, 2021 08.
Article en En | MEDLINE | ID: mdl-33988295
Neonatal Marfan syndrome is a severe, early onset presentation of pathogenic variants in FBN1. Because of the significant cardiac involvement and early mortality, nearly all reported cases have been de novo, and the disorder has not been documented to be inherited from a symptomatic parent. Here, we present a female infant with neonatal Marfan syndrome who was born to a father with Marfan syndrome. Prior to the birth of his daughter, the father had been found to have an FBN1 missense variant of uncertain clinical significance. Initial familial variant testing of the infant did not reveal the same missense variant, but Sanger sequencing of FBN1 subsequently identified a pathogenic splice site variant. The father was then found to have 10%-20% mosaicism for the same splice site variant.
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Texto completo: 1 Base de datos: MEDLINE Asunto principal: Sitios de Empalme de ARN / Fibrilina-1 / Síndrome de Marfan / Mosaicismo / Mutación Tipo de estudio: Diagnostic_studies / Prognostic_studies Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2021 Tipo del documento: Article

Texto completo: 1 Base de datos: MEDLINE Asunto principal: Sitios de Empalme de ARN / Fibrilina-1 / Síndrome de Marfan / Mosaicismo / Mutación Tipo de estudio: Diagnostic_studies / Prognostic_studies Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2021 Tipo del documento: Article