Institute for Genomic Medicine, Columbia University Medical Center, New York, NY, USA.
Department of Genetics, The Institute of Life Sciences, The Hebrew University of Jerusalem, Jerusalem, Israel.
J Hum Genet. 2021 Mar;66(3):339-343. doi: 10.1038/s10038-020-00846-1. Epub 2020 Sep 18.
Childhood-onset schizophrenia (COS) is a rare form of schizophrenia with an onset before 13 years of age. There is rising evidence that genetic factors play a major role in COS etiology, yet, only a few single gene mutations have been discovered. Here we present a diagnostic whole-exome sequencing (WES) in an Israeli Jewish female with COS and additional neuropsychiatric conditions such as obsessive-compulsive disorder (OCD), anxiety, and aggressive behavior. Variant analysis revealed a de novo novel stop gained variant in GRIA2 gene (NM_000826.4: c.1522 G > T (p.Glu508Ter)). GRIA2 encodes for a subunit of the AMPA sensitive glutamate receptor (GluA2) that functions as ligand-gated ion channel in the central nervous system and plays an important role in excitatory synaptic transmission. GluA2 subunit mutations are known to cause variable neurodevelopmental phenotypes including intellectual disability, autism spectrum disorder, epilepsy, and OCD. Our findings support the potential diagnostic role of WES in COS, identify GRIA2 as possible cause to a broad psychiatric phenotype that includes COS as a major manifestation and expand the previously reported GRIA2 loss of function phenotypes.
儿童期发病精神分裂症(COS)是一种罕见的精神分裂症,发病年龄在 13 岁之前。越来越多的证据表明,遗传因素在 COS 的发病机制中起主要作用,但仅发现了少数几个单基因突变。在这里,我们介绍了一位以色列裔犹太女性的 COS 及其他神经精神疾病(如强迫症、焦虑症和攻击性行为)的全外显子组测序(WES)诊断。变异分析显示,GRIA2 基因中存在一个新的无义获得性变异(NM_000826.4:c.1522G>T(p.Glu508Ter))。GRIA2 基因编码 AMPA 敏感型谷氨酸受体(GluA2)的一个亚基,该受体在中枢神经系统中作为配体门控离子通道发挥作用,在兴奋性突触传递中发挥重要作用。已知 GluA2 亚基突变可引起多种神经发育表型,包括智力障碍、自闭症谱系障碍、癫痫和强迫症。我们的研究结果支持 WES 在 COS 中的潜在诊断作用,确定 GRIA2 可能是包括 COS 在内的广泛精神疾病表型的原因,并扩展了先前报道的 GRIA2 功能丧失表型。