Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Genetic Services Laboratory, |
RCV001818408 | SCV002069317 | pathogenic | not provided | 2018-12-31 | criteria provided, single submitter | clinical testing | DNA sequence analysis of the KDM6A gene demonstrated a four base pair deletion in exon 17, c.2515_2518del. This pathogenic sequence change results in an amino acid frameshift and creates a premature stop codon 26 amino acids downstream of the mutation, p.Asn839Valfs*27. This pathogenic sequence change is predicted to result in an abnormal transcript, which may be degraded, or may lead to the production of a truncated KDM6A protein with potentially abnormal function. This pathogenic sequence change has previously been described in a family with KDM6A-related Kabuki syndrome (PMID: 24664873). In this family, two brothers with Kabuki syndrome were found to be hemizygous for the c.2515_2518del sequence change; their mother and maternal grandmother were heterozygous for the c.2515_2518del pathogenic sequence change and exhibited mild features of Kabuki syndrome." DNA sequence analysis of the KDM6A gene demonstrated a four base pair deletion in exon 17, c.2515_2518del. This pathogenic sequence change results in an amino acid frameshift and creates a premature stop codon 26 amino acids downstream of the mutation, p.Asn839Valfs*27. This pathogenic sequence change is predicted to result in an abnormal transcript, which may be degraded, or may lead to the production of a truncated KDM6A protein with potentially abnormal function. This pathogenic sequence change has previously been described in a family with KDM6A-related Kabuki syndrome (PMID: 24664873). In this family, two brothers with Kabuki syndrome were found to be hemizygous for the c.2515_2518del sequence change; their mother and maternal grandmother were heterozygous for the c.2515_2518del pathogenic sequence change and exhibited mild features of Kabuki syndrome. |
OMIM | RCV000170468 | SCV000222896 | pathogenic | Kabuki syndrome 2 | 2014-05-01 | no assertion criteria provided | literature only |