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A Cockayne-Syndrome-Like Phenotype with a Homozygous Truncating UVSSA Variant: Might This Be a New Cause?
Bahap, Yusuf; Kayhan, Gulsum.
Afiliación
  • Bahap Y; Medical Genetics Department, Gazi University Hospital, Ankara, Turkey.
  • Kayhan G; Medical Genetics Department, Gazi University Hospital, Ankara, Turkey.
Mol Syndromol ; 15(4): 324-327, 2024 Aug.
Article en En | MEDLINE | ID: mdl-39119453
ABSTRACT

Introduction:

UV-sensitive syndrome and Cockayne syndrome (CS) are rare autosomal recessive and transcription-coupled nucleotide excision repair disorders with different clinical manifestations, although some types are allelic. Case Presentation We report on a patient who passed away at 15 years old with a progeroid-like appearance, cachexia, hearing loss, and dental anomalies, which led us to the diagnosis of Cockayne-like progeroid syndromes. Our clinical exome sequencing including all the known genes of progeroid syndromes revealed a homozygous stop-gain variant in the UVSSA gene.

Conclusion:

Although truncating variants in the UVSSA are known to cause UVsS3, their association with CS has not yet been defined. This case might be the first report of a CS-like phenotype caused by a defective UVSSA.
Palabras clave

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Mol Syndromol Año: 2024 Tipo del documento: Article País de afiliación: Turquía Pais de publicación: Suiza

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Mol Syndromol Año: 2024 Tipo del documento: Article País de afiliación: Turquía Pais de publicación: Suiza