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Bi-allelic loss-of-function variants in KIF21A cause severe fetal akinesia with arthrogryposis multiplex.

Ruth J FalbAmelie J MüllerWolfram KleinMona GrimmelUte GrasshoffStephanie SprangerPetra StöbeDarja GauckAlma KuechlerNicola DikowEva M C SchwaiboldChristoph SchmidtLuisa AverdunkRebecca BuchertTilman HeinrichNatalia ProdanJoohyun ParkMartin KehrerMarc SturmOlga KelemenSilke HartmannDenise HornDirk EmmerichNina HirtArmin NeumannGlen KristiansenUlrich GembruchSusanne HaenReiner SiebertSabine HentzeMarkus HoopmannStephan OssowskiStephan WaldmüllerStefanie Beck-WödlDieter GläserIsmail TekesinFelix DistelmaierOlaf RiessKarl-Oliver KaganAndreas DufkeTobias B Haack
Published in: Journal of medical genetics (2021)
Our study underlines the broad locus heterogeneity of FA with well-established and atypical genotype-phenotype associations. We describe KIF21A as a new factor implicated in the pathogenesis of severe neurogenic FA sequence with arthrogryposis of multiple joints, pulmonary hypoplasia and facial dysmorphisms. This hypothesis is further corroborated by a recent report on overlapping phenotypes observed in Kif21a null piglets.
Keyphrases
  • early onset
  • pulmonary hypertension
  • spinal cord injury
  • high throughput
  • single cell
  • copy number
  • drug induced
  • gene expression
  • soft tissue
  • dna methylation
  • genome wide