Lee Inn-Chi, Chen Jia-Yuh, Chen Yung-Jung, Yu Ju-Shan, Su Pen-Hua
Division of Pediatric Neurology, Department of Pediatrics, College of Medicine, Chung-Shan Medical University Hospital, Institute of Medicine of Chung-Shan Medical University, Taichung, Taiwan.
Pediatr Neurol. 2009 May;40(5):387-91. doi: 10.1016/j.pediatrneurol.2008.12.004.
Benign familial neonatal convulsions are a rare, autosomal-dominant form of neonatal epileptic syndrome. It can occur 1 week after birth, and usually involves frequent episodes, but with a benign course. The diagnosis depends on family history and clinical features. The mutant gene locates at 20q13, a voltage-gated potassium-channel gene (KCNQ2). Our patient exhibited an uneventful delivery course and onset of seizures at age 2 days. The general tonic seizures were unique and asymmetric, with frequencies of >20 per day. Results of examinations were within normal limits, including biochemistry and brain magnetic resonance imaging. Abnormalities included a small ventricular septum defect on cardiac sonography unrelated to the seizures, and nonspecific, multiple, high-voltage sharp waves and spike waves occurring infrequently in the central region on electroencephalogram. After phenobarbital and phenytoin use, the seizures persisted. On day 12, another antiepileptic drug, vigabatrin (unavailable in the United States), was used, and seizures decreased. A novel mutation of KCNQ2 was identified from a blood sample. The baby had occasional seizures with drug treatment at age 3 months. Benign familial neonatal convulsion should be considered in a baby with a unique seizure pattern and positive family history. Genetic counseling and diagnosis are mandatory.
良性家族性新生儿惊厥是一种罕见的常染色体显性遗传形式的新生儿癫痫综合征。它可在出生后1周出现,通常发作频繁,但病程良性。诊断取决于家族史和临床特征。突变基因定位于20q13,即一个电压门控钾通道基因(KCNQ2)。我们的患者分娩过程顺利,2日龄时出现惊厥。全身性强直发作独特且不对称,每天发作次数>20次。包括生化检查和脑磁共振成像在内的各项检查结果均在正常范围内。异常情况包括心脏超声检查发现的小室间隔缺损,与惊厥无关,以及脑电图显示在中央区域偶尔出现的非特异性、多个高电压尖波和棘波。使用苯巴比妥和苯妥英后,惊厥仍持续发作。在第12天,使用了另一种抗癫痫药物氨己烯酸(在美国无法获得),惊厥次数减少。从一份血样中鉴定出KCNQ2的一种新突变。该婴儿在3个月大时接受药物治疗后仍偶尔发作惊厥。对于具有独特惊厥模式且家族史阳性的婴儿,应考虑良性家族性新生儿惊厥。必须进行遗传咨询和诊断。