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Genetic diagnosis of childhood sensorineural hearing loss.
Reda Del Barrio, Sara; García Fernández, Alfredo; Quesada-Espinosa, Juan Francisco; Sánchez-Calvín, María Teresa; Gómez-Manjón, Irene; Sierra-Tomillo, Olalla; Juárez-Rufián, Alexandra; de Vergas Gutiérrez, Joaquín.
Afiliación
  • Reda Del Barrio S; Servicio de Otorrinolaringología, Hospital Universitario 12 de Octubre, Madrid, Spain. Electronic address: sara.reda@salud.madrid.org.
  • García Fernández A; Servicio de Otorrinolaringología, Hospital Universitario 12 de Octubre, Madrid, Spain.
  • Quesada-Espinosa JF; Servicio de Genética, Hospital Universitario 12 de Octubre, Madrid, Spain.
  • Sánchez-Calvín MT; Servicio de Genética, Hospital Universitario 12 de Octubre, Madrid, Spain.
  • Gómez-Manjón I; Servicio de Genética, Hospital Universitario 12 de Octubre, Madrid, Spain.
  • Sierra-Tomillo O; Servicio de Genética, Hospital Universitario 12 de Octubre, Madrid, Spain.
  • Juárez-Rufián A; Servicio de Genética, Hospital Universitario 12 de Octubre, Madrid, Spain.
  • de Vergas Gutiérrez J; Servicio de Otorrinolaringología, Hospital Universitario 12 de Octubre, Madrid, Spain.
Article en En | MEDLINE | ID: mdl-38224868
ABSTRACT

INTRODUCTION:

Congenital/early-onset sensorineural hearing loss (SNHL) is one of the most common hereditary disorders in our environment. There is increasing awareness of the importance of an etiologic diagnosis, and genetic testing with next-generation sequencing (NGS) has the highest diagnostic yield. Our study shows the genetic results obtained in a cohort of patients with bilateral congenital/early-onset SNHL. MATERIALS AND

METHODS:

We included 105 children with bilateral SNHL that received genetic testing between 2019 and 2022. Genetic tests were performed with whole exome sequencing, analyzing genes related to hearing loss (virtual panel with 244 genes).

RESULTS:

48% (50/105) of patients were genetically diagnosed. We identified pathogenic and likely pathogenic variants in 26 different genes, and the most frequently mutated genes were GJB2, USH2A and STRC. 52% (26/50) of variants identified produced non-syndromic hearing loss, 40% (20/50) produced syndromic hearing loss, and the resting 8% (4/50) could produce both non-syndromic and syndromic hearing loss.

CONCLUSIONS:

Genetic testing plays a vital role in the etiologic diagnosis of bilateral SNHL. Our cohort shows that genetic testing with NGS has a high diagnostic yield and can provide useful information for the clinical workup of patients.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Pruebas Genéticas / Síndromes de Usher Tipo de estudio: Diagnostic_studies Límite: Child / Humans Idioma: En Revista: Acta Otorrinolaringol Esp (Engl Ed) Año: 2024 Tipo del documento: Article Pais de publicación: España

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Pruebas Genéticas / Síndromes de Usher Tipo de estudio: Diagnostic_studies Límite: Child / Humans Idioma: En Revista: Acta Otorrinolaringol Esp (Engl Ed) Año: 2024 Tipo del documento: Article Pais de publicación: España