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A novel truncating variant in the FGD1 gene associated with Aarskog-Scott syndrome in a family previously diagnosed with Tel Hashomer camptodactyly.

Irena KesselAlina GermanAmir Pelegnull nullClaudia Gonzaga-JaureguiTamar PapernaNina EkhilevitchAlina KurolapHagit Baris FeldmanLena Sagi-Dain
Published in: American journal of medical genetics. Part A (2021)
Tel Hashomer camptodactyly syndrome is a long-known entity characterized by camptodactyly with muscular hypoplasia, skeletal dysplasia, and abnormal palmar creases. Currently, the genetic basis for this disorder is unknown, thus there is a possibility that this clinical presentation may be contained within another genetic diagnosis. Here, we present a multiplex family with a previous clinical diagnosis of Tel Hashomer camptodactyly syndrome. Whole exome sequencing and pedigree-based analysis revealed a novel hemizygous truncating variant c.269_270dup (p.Phe91Alafs*34) in the FGD1 gene (NM_004463.3) in all three symptomatic patients, congruous with a diagnosis of Aarskog-Scott syndrome. Our report adds to the limited data on Aarskog-Scott syndrome, and emphasizes the importance of unbiased comprehensive molecular testing toward establishing a diagnosis for genetic syndromes with unknown genetic basis.
Keyphrases
  • genome wide
  • copy number
  • case report
  • newly diagnosed
  • chronic kidney disease
  • dna methylation
  • photodynamic therapy
  • single cell
  • prognostic factors
  • artificial intelligence