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由于中链酰基辅酶A脱氢酶缺陷导致的二羧酸尿症。儿童低血糖的一个病因。

Dicarboxylic aciduria due to medium chain acyl CoA dehydrogenase defect. A cause of hypoglycemia in childhood.

作者信息

Divry P, David M, Gregersen N, Kølvraa S, Christensen E, Collet J P, Dellamonica C, Cotte J

出版信息

Acta Paediatr Scand. 1983 Nov;72(6):943-9. doi: 10.1111/j.1651-2227.1983.tb09849.x.

Abstract

Dicarboxylic aciduria was found during hypoglycemic episode in a 14 months old girl. Her brother had died at the age of 4 years during febrile illness. A ketogenic diet induced in this patient a severe hypoglycemia. Urinary organic acid profile exhibited abnormal excretion of the C6-C10 dicarboxylic acids (adipic-suberic-sebacic) and related metabolites (5 hydroxyhexanoic, hexanoylglycine, suberyl glycine). This pattern suggested a defect in fatty acids beta oxidation. Plasma carnitine values was within control limits. Similar clinical findings and urinary organic acids excretion have been described in 6 patients since the initial case of Gregersen. Enzymatic studies on cultivated fibroblasts from our patient showed a defect in medium chain CoA dehydrogenase. The treatment of this disease consists of glucose infusion during attacks and prevention of fasting. This rare disease must be considered in a child with non ketotic hypoglycemia or Reye's syndrome.

摘要

在一名14个月大的女孩低血糖发作期间发现了二羧酸尿症。她的哥哥在4岁时因发热性疾病去世。对该患者采用生酮饮食引发了严重低血糖。尿有机酸谱显示C6 - C10二羧酸(己二酸 - 辛二酸 - 癸二酸)及相关代谢产物(5 - 羟基己酸、己酰甘氨酸、辛二酰甘氨酸)排泄异常。这种模式提示脂肪酸β氧化存在缺陷。血浆肉碱值在正常范围内。自格雷格森首例病例以来,已有6例患者出现类似的临床发现和尿有机酸排泄情况。对我们患者培养的成纤维细胞进行的酶学研究显示中链辅酶A脱氢酶存在缺陷。该疾病的治疗包括发作时输注葡萄糖以及预防禁食。对于患有非酮症低血糖或瑞氏综合征的儿童,必须考虑这种罕见疾病。

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