Symptomatic mosaicism for a novel FBN1 splice site variant in a parent causing inherited neonatal Marfan syndrome.
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.
Palabras clave
Texto completo:
1
Base de datos:
MEDLINE
Asunto principal:
Sitios de Empalme de ARN
/
Fibrilina-1
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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