TY - JOUR
T1 - Jumonji domain containing 1C (JMJD1C) sequence variants in seven patients with autism spectrum disorder, intellectual disability and seizures
AU - Slavotinek, Anne
AU - van Hagen, Johanna M.
AU - Kalsner, Louisa
AU - Pai, Shashidhar
AU - Davis-Keppen, Laura
AU - Ohden, Lisa
AU - Weber, Yvonne G.
AU - Macke, Erica L.
AU - Klee, Eric W.
AU - Morava, Eva
AU - Gunderson, Lauren
AU - Person, Richard
AU - Liu, Shuxi
AU - Weiss, Marjan
N1 - Publisher Copyright:
© 2020 Elsevier Masson SAS
PY - 2020/4
Y1 - 2020/4
N2 - The Jumonji domain containing 1C (JMJD1C) gene encodes the Jumonji domain-containing protein 1C (JMJD1C) and is a member of the jmJC domain-containing protein family involved in histone demethylation that is expressed in the brain. We report seven, unrelated patients with developmental delays or intellectual disability and heterozygous, de novo sequence variants in JMJD1C. All patients had developmental delays, but there were no consistent additional findings. Two patients were reported to have seizures for which there was no other identified cause. De novo, deleterious sequence variants in JMJD1C have previously been reported in patients with autism spectrum disorder and a phenotype resembling classical Rett syndrome, but only one JMJD1C variant has undergone functional evaluation. In all of the seven patients in this report, there was a plausible, alternative explanation for the neurocognitive phenotype or a modifying factor, such as an additional potentially pathogenic variant, presence of the variant in a population database, heteroplasmy for a mitochondrial variant or mosaicism for the JMJD1C variant. Although the de novo variants in JMJD1C are likely to be relevant to the developmental phenotypes observed in these patients, we conclude that further data supporting the association of JMJD1C variants with intellectual disability is still needed.
AB - The Jumonji domain containing 1C (JMJD1C) gene encodes the Jumonji domain-containing protein 1C (JMJD1C) and is a member of the jmJC domain-containing protein family involved in histone demethylation that is expressed in the brain. We report seven, unrelated patients with developmental delays or intellectual disability and heterozygous, de novo sequence variants in JMJD1C. All patients had developmental delays, but there were no consistent additional findings. Two patients were reported to have seizures for which there was no other identified cause. De novo, deleterious sequence variants in JMJD1C have previously been reported in patients with autism spectrum disorder and a phenotype resembling classical Rett syndrome, but only one JMJD1C variant has undergone functional evaluation. In all of the seven patients in this report, there was a plausible, alternative explanation for the neurocognitive phenotype or a modifying factor, such as an additional potentially pathogenic variant, presence of the variant in a population database, heteroplasmy for a mitochondrial variant or mosaicism for the JMJD1C variant. Although the de novo variants in JMJD1C are likely to be relevant to the developmental phenotypes observed in these patients, we conclude that further data supporting the association of JMJD1C variants with intellectual disability is still needed.
KW - Autism spectrum disorder
KW - Intellectual disability
KW - JMJD1C
KW - Jumonji domain-containing protein 1C
UR - http://www.scopus.com/inward/record.url?scp=85078359873&partnerID=8YFLogxK
U2 - 10.1016/j.ejmg.2020.103850
DO - 10.1016/j.ejmg.2020.103850
M3 - Article
C2 - 31954878
AN - SCOPUS:85078359873
SN - 1769-7212
VL - 63
JO - European Journal of Medical Genetics
JF - European Journal of Medical Genetics
IS - 4
M1 - 103850
ER -