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PUS3-related disorder: Report of a novel patient and delineation of the phenotypic spectrum.

Alessandro BorghesiMassimo PlumariElena RossiClaudia ViganòRosa Maria CerboAlessia Claudia CodazziEnza Maria ValenteSimone Gana
Published in: American journal of medical genetics. Part A (2021)
PUS3 encodes the pseudouridylate synthase 3, an enzyme catalyzing the formation of tRNA pseudouridine, which plays a critical role in tRNA structure, function, and stability. Biallelic pathogenic variants of PUS3 have been previously associated with severe intellectual disability, microcephaly, epilepsy, and short stature. We identified a novel homozygous PUS3 frameshift variant in a child with facial dysmorphisms, growth failure, microcephaly, retinal dystrophy, cerebellar hypoplasia, congenital heart defect, and right kidney hypoplasia. This patient further expands the phenotypic spectrum of PUS3-related disorders to include a more severe syndromic presentation.
Keyphrases
  • intellectual disability
  • autism spectrum disorder
  • case report
  • early onset
  • zika virus
  • mental health
  • diabetic retinopathy
  • genome wide
  • growth hormone