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Congenital hypothyroidism, cardiac defects, and pancreatic agenesis in an infant with GATA6 mutation.

Nikhil RaghuramAshish MarwahaMary-Louise C GreerEstelle GaudaDavid Chitayat
Published in: American journal of medical genetics. Part A (2020)
GATA6 pathogenic variants primarily manifest a phenotype with pancreatic agenesis and cardiac malformations. However, additional congenital malformations affecting the biliary system, congenital diaphragmatic hernia and developmental delay have been reported. We report a newborn, prenatally diagnosed with truncus arteriosus and intrauterine growth restriction, who was postnatally found to have pancreatic agenesis associated with neonatal diabetes and hepatobiliary abnormalities. Whole exome sequencing identified a de novo, heterozygous mutation in the GATA6 gene (c.1366C>T; p.Arg456Cys). Further investigations revealed abnormalities not previously associated with GATA6 mutation, including unilateral thyroid lobe agenesis associated with congenital hypothyroidism, absent gall bladder, possible adrenal insufficiency, thrombocytopenia, and neonatal stroke.
Keyphrases
  • transcription factor
  • copy number
  • type diabetes
  • left ventricular
  • spinal cord injury
  • atrial fibrillation
  • single cell
  • heart failure
  • insulin resistance
  • brain injury
  • skeletal muscle
  • weight loss