先天性高氨血症:鸟氨酸转氨基甲酰基酶(OTC)缺乏症的有症状的携带者女孩及其无症状的杂合母亲:杂合基因状态检测的特异性酶诊断和动力学调查

Acta paediatrica Hungarica Pub Date : 1991-01-01
A László, T Karsai, A Várkonyi
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引用次数: 0

摘要

在一名患有慢性高氨血症的2岁女孩的肝脏活检标本和其父母的红细胞和白细胞均质液中测定了遗传性高氨血症综合征的特异性酶(氨甲酰磷酸合成酶CPS)、鸟氨酸转氨基甲酰基酶(OTC)、精氨酸-琥珀酸合成酶(ASS)、精氨酸-琥珀酸裂解酶(ASL)和精氨酸酶(ASE)的活性。OTC在患者肝脏匀浆中的活性为62.9%;与对照组相比,父母的白细胞比例分别为78.5%(母亲)和102%(父亲)。我们的病人被证明是OTC缺乏症的症状携带者,她的母亲被证明是一个无症状的携带者。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Congenital hyperammonemia: symptomatic carrier girl patient and her asymptomatic heterozygous mother for ornithine transcarbamylase (OTC) deficiency: specific enzyme diagnostic and kinetic investigations for the detection of heterozygous genostatus.

Activities of the specific enzymes of the inherited hyperammonemic syndromes (carbamoyl-phosphate synthetase CPS), ornithine transcarbamylase (OTC), arginine-succinate-synthetase (ASS), arginine-succinate-lyase (ASL) and arginase (ASE) were measured in a liver biopsy specimen of a 2 years-old girl suffering from chronic hyperammonemia and in the erythrocyte- and leukocyte-homogenisate of her parents. The activity of OTC in liver homogenisate of the patient was 62.9 percent; in the leukocytes of the parents it was 78.5 percent (in mother) and 102 per cent (in the father) as compared to the controls. Our patient proved to be a symptomatic carrier of OTC deficiency and her mother proved to be an asymptomatic carrier.

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