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肉毒碱酰基转移酶Ⅰ缺乏症伴新突变。

Malonyl coenzyme A decarboxylase deficiency with a novel mutation.

机构信息

Department of Pediatric Metabolism, Ankara Yildirim Beyazit University, Ankara City Hospital, Ankara, Turkey.

Department of Pediatric Metabolism, Ankara City Hospital, Ankara, Turkey.

出版信息

Cardiol Young. 2021 Sep;31(9):1535-1537. doi: 10.1017/S104795112100113X. Epub 2021 Mar 22.

Abstract

Malonyl-CoA, a product of acetyl-CoA carboxylase is a metabolic intermediate in lipogenic tissues that include liver and adipose tissue, where it is involved in the de novo fatty acid synthesis and elongation. Malonyl-CoA decarboxylase (MLYCD, E.C.4.1.1.9), a 55-kDa enzyme catalyses the conversion of malonyl-CoA to acetyl-CoA and carbon dioxide, thus providing a route for disposal of malonyl-CoA from mitochondria and peroxisomes, whereas in the cytosol, the malonyl-CoA pool is regulated by the balance of MLYCD and acetyl-CoA carboxylase activities. So far, 34 cases with different MLYCD gene defects comprising point mutations, stop codons, and frameshift mutations have been reported in the literature. Here, we describe the follow-up of a patient affected by malonic aciduria upon neonatal onset. Molecular analysis showed novel homozygous mutations in the MLYCD gene. Our findings expand the number of reported cases and add a novel variant to the repertoire of MLYCD mutations.

摘要

丙二酰辅酶 A 是乙酰辅酶 A 羧化酶的产物,是包括肝脏和脂肪组织在内的脂肪组织中的代谢中间产物,它参与脂肪酸的从头合成和延伸。丙二酰辅酶 A 脱羧酶(MLYCD,EC4.1.1.9),一种 55kDa 的酶,催化丙二酰辅酶 A 转化为乙酰辅酶 A 和二氧化碳,从而提供了一种从线粒体和过氧化物酶体中清除丙二酰辅酶 A 的途径,而在细胞质中,丙二酰辅酶 A 池的调节取决于 MLYCD 和乙酰辅酶 A 羧化酶的活性平衡。到目前为止,文献中已经报道了 34 例不同的 MLYCD 基因突变病例,包括点突变、无义突变和移码突变。在这里,我们描述了一例新生儿起病的丙二酸尿症患者的随访情况。分子分析显示 MLYCD 基因存在新型纯合突变。我们的发现扩展了报道病例的数量,并为 MLYCD 突变谱增加了一种新的变异体。

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