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Exome Analysis Reveals Novel Missense and Deletion Variants in the CC2D2A Gene as Causative of Joubert Syndrome.

Rute Luísa Cabrita PintoSilvia ViaggiEdoardo CanaleMarina Martinez PoppleValeria CapraGiuseppina ConteducaBarbara TestaDomenico A CovielloAngela Elvira Covone
Published in: Genes (2023)
The CC2D2A gene is essential for primary cilia formation, and its disruption has been associated with Joubert Syndrome-9 (JBTS9), a ciliopathy with typical neurodevelopmental features. Here, we describe an Italian pediatric patient with typical features of Joubert Syndrome (JBTS): "Molar Tooth Sign", global developmental delay, nystagmus, mild hypotonia, and oculomotor apraxia. Whole exome sequencing and segregation analysis identified in our infant patient a novel heterozygous germline missense variant c.3626C > T; p.(Pro1209Leu) inherited from the father and a novel 7.16 kb deletion inherited from the mother. To the best of our knowledge, this is the first report showing a novel missense and deletion variant involving exon 30 of the CC2D2A gene.
Keyphrases
  • copy number
  • case report
  • genome wide
  • intellectual disability
  • genome wide identification
  • healthcare
  • gene expression
  • genome wide analysis
  • transcription factor