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Rare mosaic variant of GJA1 in a patient with a neurodevelopmental disorder.

Rina ShimomuraTomoe YanagishitaKumiko IshiguroMinobu ShichijiTakatoshi SatoKeiko Shimojima YamamotoMiho NagataYasuki IshiharaYohei MiyashitaKeiko IshigakiSatoru NagataYoshihiro AsanoToshiyuki Yamamoto
Published in: Human genome variation (2024)
GJA1 is the causative gene for oculodentodigital dysplasia (ODDD). A novel de novo GJA1 variant, NM 000165:c263C > T [p.P88L], was identified in a mosaic state in a patient with short stature, seizures, delayed myelination, mild hearing loss, and tooth enamel hypoplasia. Although the patient exhibited severe neurodevelopmental delay, other clinical features of ODDD, including limb anomalies, were mild. This may be due to differences in the mosaic ratios in different organs.
Keyphrases
  • case report
  • hearing loss
  • photodynamic therapy
  • genome wide
  • gene expression
  • dna methylation
  • copy number
  • drug induced