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非酮性高甘氨酸血症所致早发性肌阵挛性脑病的生酮饮食治疗。

Ketogenic diet in early myoclonic encephalopathy due to non ketotic hyperglycinemia.

机构信息

Neurology Unit, "Bambino Gesù" Children's Hospital, IRCCS, Rome, Italy.

出版信息

Eur J Paediatr Neurol. 2012 Sep;16(5):509-13. doi: 10.1016/j.ejpn.2011.12.015. Epub 2012 Jan 18.

Abstract

Non ketotic hyperglycinemia is a rare inborn error of glycine metabolism due to deficient activity of glycine cleavage system, a multienzymatic complex consisting of four protein subunits: the P-protein, the H-protein, the T-protein and the L-protein. The neonatal form of non ketotic hyperglycinemia presents in the first days of life with encephalopathy, seizures, multifocal myoclonus and characteristic "hiccups". Rapid progression may lead to intractable seizures, coma and respiratory failure requiring mechanical ventilation. Clinical trial with scavenges drugs decreasing glycine levels such as sodium benzoate, and with drugs reducing NMDA receptors excitatory properties, such as ketamine and dextromethorphan, have been tried but the outcome is usually poor; antiepileptic therapy, moreover, is unable to control epileptic seizures. Ketogenic diet has been successfully tried for refractory epilepsy in pediatric patients. We report three cases affected by neonatal non ketotic hyperglycinemia and early myoclonic encephalopathy treated with ketogenic diet. In our patients ketogenic diet, in association with standard pharmacological therapy, determined dramatic reduction of seizures and improved quality of life.

摘要

非酮症高甘氨酸血症是一种罕见的甘氨酸代谢先天性错误,由于甘氨酸裂解系统活性缺乏所致,甘氨酸裂解系统是一种由四个蛋白亚基组成的多酶复合物:P 蛋白、H 蛋白、T 蛋白和 L 蛋白。新生儿型非酮症高甘氨酸血症在生命的头几天出现脑病、癫痫发作、多灶性肌阵挛和特征性“呃逆”。快速进展可能导致难治性癫痫发作、昏迷和需要机械通气的呼吸衰竭。已经尝试了临床试验,使用清除甘氨酸水平的药物,如苯甲酸钠,以及降低 NMDA 受体兴奋性的药物,如氯胺酮和右美沙芬,但结果通常较差;此外,抗癫痫治疗无法控制癫痫发作。生酮饮食已成功用于治疗儿科患者的难治性癫痫。我们报告了三例新生儿非酮症高甘氨酸血症和早发性肌阵挛性脑病患者接受生酮饮食治疗的情况。在我们的患者中,生酮饮食联合标准药物治疗,显著减少了癫痫发作次数,并提高了生活质量。

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