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Overlapping phenotype comprising Kenny-Caffey type 2 and Sanjad-Sakati syndromes: The first case report.

Thiago Rodrigues CavoleEduardo PerroneMaria de Fatima de Faria SoaresMagnus Régios Dias da SilvaSergio Setsuo MaedaMarise Lazaretti-CastroAna Beatriz Alvarez Perez
Published in: American journal of medical genetics. Part A (2020)
Kenny-Caffey syndrome (KCS) is a rare hereditary skeletal disorder involving hypoparathyroidism. The autosomal dominant form (KCS2), caused by heterozygous pathogenic variants in the FAM111A gene, is distinguished from the autosomal recessive form (KCS1) and Sanjad-Sakati syndrome (SSS), both caused by pathogenic variants in the tubulin folding cofactor E (TBCE) gene, by the absence of microcephaly and intellectual disability. We present a patient with KCS2 caused by a de novo pathogenic variant c.1706G>A (p.Arg569His) in FAM111A gene, presenting intellectual disability and microcephaly, which are considered to be typical signs of SSS. We suggest that KCS1, KCS2, and SSS may not represent mutually exclusive clinical entities, but possibly an overlapping spectrum.
Keyphrases
  • intellectual disability
  • case report
  • copy number
  • autism spectrum disorder
  • genome wide
  • genome wide identification
  • zika virus
  • dna methylation
  • gene expression
  • single molecule
  • transcription factor