Xiong Juan, Peng Jing, Duan Hao-Lin, Chen Chen, Wang Xiao-Le, Chen Shi-Meng, Yin Fei
Department of Pediatrics, Xiangya Hospital, Central South University, Changsha 410008, China.
Zhongguo Dang Dai Er Ke Za Zhi. 2018 Feb;20(2):154-157. doi: 10.7499/j.issn.1008-8830.2018.02.014.
A 4-month-old girl developed convulsion in the neonatal period, which was focal motor seizures in the initial stage and later became spasm and tonic spasm. And the girl also had psychomotor retardation and recurrent pulmonary infection. Electroencephalography showed hypsarrhythmia, normal results were obtained from cranial magnetic resonance imaging, cerebrospinal fluid examination, and urine organic acid analysis, as well as the spectral analyses of blood ammonia, blood lactic acid, blood amino acids, and acylcarnitines. Gene detection revealed a de novo heterozygous mutation, c.607G>A (p.G203R) , in GNAO1. The girl was then diagnosed with GNAO1-associated early infantile epileptic encephalopathy (EIEE type 17). The seizures were well controlled by topiramate and vigabatrin, but there was no improvement in psychomotor development. She also suffered from recurrent pulmonary infection and died at the age of 12 months due to severe pneumonia. For children with unexplained early infantile epileptic encephalopathy, GNAO1 gene mutations should be considered and genetic tests should be performed as early as possible. Recurrent pulmonary infection should also be taken seriously.
一名4个月大的女孩在新生儿期出现惊厥,初期为局灶性运动性发作,后来发展为痉挛和强直性痉挛。该女孩还存在精神运动发育迟缓及反复肺部感染。脑电图显示高度失律,头颅磁共振成像、脑脊液检查、尿有机酸分析以及血氨、血乳酸、血氨基酸和酰基肉碱的谱分析结果均正常。基因检测发现GNAO1基因存在一个新发杂合突变,即c.607G>A(p.G203R)。该女孩随后被诊断为GNAO1相关的早期婴儿型癫痫性脑病(EIEE 17型)。托吡酯和氨己烯酸能很好地控制癫痫发作,但精神运动发育无改善。她还反复发生肺部感染,最终在12个月大时因重症肺炎死亡。对于不明原因的早期婴儿型癫痫性脑病患儿,应考虑GNAO1基因突变并尽早进行基因检测。反复肺部感染也应予以重视。