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Novel compound heterozygous ABCA2 variants cause IDPOGSA, a variable phenotypic syndrome with intellectual disability.

Yuta InoueNaomi TsuchidaChong Ae KimBruno de Oliveira StephanMatheus Augusto Araujo CastroRachel Sayuri HonjoDebora Romeo BertolaYuri UchiyamaKohei HamanakaAtsushi FujitaEriko KoshimizuKazuharu MisawaSatoko MiyatakeTakeshi MizuguchiNaomichi Matsumoto
Published in: Journal of human genetics (2024)
The gene for ATP binding cassette subfamily A member 2 (ABCA2) is located at chromosome 9q34.3. Biallelic ABCA2 variants lead to intellectual developmental disorder with poor growth and with or without seizures or ataxia (IDPOGSA). In this study, we identified novel compound heterozygous ABCA2 variants (NM_001606.5:c.[5300-17C>A];[6379C>T]) by whole exome sequencing in a 28-year-old Korean female patient with intellectual disability. These variants included intronic and nonsense variants of paternal and maternal origin, respectively, and are absent from gnomAD. SpliceAI predicted that the intron variant creates a cryptic acceptor site. Reverse transcription-PCR using RNA extracted from a lymphoblastoid cell line of the patient confirmed two aberrant transcripts. Her clinical features are compatible with those of IDPOGSA.
Keyphrases
  • intellectual disability
  • copy number
  • autism spectrum disorder
  • early onset
  • case report
  • genome wide
  • dna methylation
  • photodynamic therapy
  • body mass index
  • gene expression