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马尔济斯犬的丙二酸尿症:成纤维细胞中甲基丙二酸浓度正常及丙二酰辅酶A脱羧酶活性正常

Malonic aciduria in Maltese dogs: normal methylmalonic acid concentrations and malonyl-CoA decarboxylase activity in fibroblasts.

作者信息

O'Brien D P, Barshop B A, Faunt K K, Johnson G C, Gibson K M, Shelton G D

机构信息

Department of Veterinary Medicine and Surgery, University of Missouri, Columbia 65211, USA.

出版信息

J Inherit Metab Dis. 1999 Dec;22(8):883-90. doi: 10.1023/a:1005635306257.

Abstract

A family of Maltese dogs with malonic aciduria is reported. The propositus presented at 3 years of age with episodes of seizures and stupor with hypoglycaemia, acidosis, and ketonuria. Urinary organic acid assays showed elevated malonic acid without elevation of methylmalonic acid. Cultured fibroblasts had normal malonyl-CoA decarboxylase activity. Treatment with frequent feedings of a low-fat diet high in medium-chain triglycerides resulted in normalization of clinical signs and a resolution of the malonic aciduria. Two full siblings of the propositus had died at a young age of undiagnosed metabolic and neurological disease. Urine organic acid assays were performed on other family members. A half-sister showed mild malonic aciduria and other organic acid changes similar to the propositus, while the mother and half-brother showed mildly elevated ketone bodies. This family suggests further genetic and clinical heterogeneity in the malonic acidurias.

摘要

报道了一窝患有丙二酸尿症的马尔济斯犬。先证者3岁时出现癫痫发作和昏迷,伴有低血糖、酸中毒和酮尿症。尿有机酸检测显示丙二酸升高,而甲基丙二酸未升高。培养的成纤维细胞具有正常的丙二酰辅酶A脱羧酶活性。频繁喂食富含中链甘油三酯的低脂饮食进行治疗后,临床症状恢复正常,丙二酸尿症得到缓解。先证者的两个同胞全在幼年时死于未确诊的代谢和神经疾病。对其他家庭成员进行了尿有机酸检测。一个同父异母的妹妹表现出轻度丙二酸尿症以及与先证者相似的其他有机酸变化,而母亲和同父异母的弟弟则表现出酮体轻度升高。这个家系提示丙二酸尿症存在进一步的遗传和临床异质性。

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