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阿曼的翁韦里希特-伦德伯格进行性肌阵挛癫痫

Unverricht-Lundborg progressive myoclonus epilepsy in Oman.

作者信息

Santoshkumar Balagopal, Turnbull Julie, Minassian Berge A

机构信息

Department of Neurology, Royal Hospital, Muscat, Oman.

出版信息

Pediatr Neurol. 2008 Apr;38(4):252-5. doi: 10.1016/j.pediatrneurol.2007.11.006.

Abstract

We analyzed the clinical, electrophysiologic, and genetic features of Omani Arab patients suspected of manifesting the Unverricht-Lundborg form of progressive myoclonus epilepsy. Ten patients (five boys, five girls; mean age at onset, 10.2 years) were evaluated. Unverricht-Lundborg disease was confirmed in all by detection of dodecamer repeat expansion mutations in the EPM1 gene. There was no correlation between age at onset or severity of disease with sizes of dodecamer repeats. Myoclonic seizures were the presenting symptom in 70% of patients. Myoclonus was severe in adolescence, but remained stable or improved beyond 5-6 years of disease onset. No significant cognitive decline occurred. Nearly 75% of patients exhibited mild to moderate cerebellar dysfunction, which was nonprogressive after adulthood. Slowing of background activity, generalized spike-wave discharges, and photoparoxysmal responses were evident in all patients' electroencephalograms. Spike-wave discharges and photoparoxysmal responses tended to disappear in adulthood. This cluster of progressive myoclonus epilepsy patients manifested typical Unverricht-Lundborg disease. All cases had mutations in EPM1, the known gene for this disorder, and therefore do not contribute to identifying the gene in a second Unverricht-Lundborg disease locus recently mapped in Arab patients from Israel. Although Unverricht-Lundborg disease is very severe in adolescence, its clinical signs stabilize and improve somewhat in adulthood in this so-called "progressive epilepsy."

摘要

我们分析了疑似表现为翁韦里希特-伦德伯格型进行性肌阵挛癫痫的阿曼阿拉伯患者的临床、电生理和遗传特征。对10名患者(5名男孩,5名女孩;平均发病年龄10.2岁)进行了评估。通过检测EPM1基因中的十二聚体重复扩增突变,在所有患者中确诊为翁韦里希特-伦德伯格病。发病年龄或疾病严重程度与十二聚体重复序列的大小之间无相关性。70%的患者以肌阵挛发作作为首发症状。肌阵挛在青春期严重,但在发病5至6年后保持稳定或有所改善。未出现明显的认知功能下降。近75%的患者表现出轻度至中度的小脑功能障碍,成年后无进展。所有患者的脑电图均显示背景活动减慢、广泛性棘波放电和光阵发性反应。棘波放电和光阵发性反应在成年后往往消失。这组进行性肌阵挛癫痫患者表现出典型的翁韦里希特-伦德伯格病。所有病例均在EPM1(该疾病的已知基因)中存在突变,因此对于在最近从以色列阿拉伯患者中定位的第二个翁韦里希特-伦德伯格病基因座中鉴定基因没有帮助。尽管翁韦里希特-伦德伯格病在青春期非常严重,但其临床症状在成年期会在这种所谓的“进行性癫痫”中有所稳定和改善。

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