Suppr超能文献

一例具有轻度临床特征的丙二酰辅酶A脱羧酶缺乏症新病例。

A new case of malonyl-CoA decarboxylase deficiency with mild clinical features.

作者信息

Liu Huan, Tan Dongqiong, Han Lianshu, Ye Jun, Qiu Wenjuan, Gu Xuefan, Zhang Huiwen

机构信息

Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute for Pediatric Research, Shanghai Jiao Tong University School of Medicine, Shanghai, China.

出版信息

Am J Med Genet A. 2016 May;170A(5):1347-51. doi: 10.1002/ajmg.a.37590. Epub 2016 Feb 9.

Abstract

Malonyl-CoA decarboxylase deficiency is an extremely rare autosomal recessive inborn error of fatty acid metabolism. It usually follows a severe disease course and presents poor prognosis without treatment. Here, we report an affected female juvenile with a mild clinical and biochemical phenotype who mainly featured poor schooling without cardiomyopathy and metabolic acidosis. She was suspected of malonyl-CoA decarboxylase deficiency due to a 57-kb deletion in 16q23.3 encompassing the MLCYD gene revealed by chromosome microarray. Malonyl-CoA decarboxylase deficiency was then confirmed by acylcarnitine analysis and organic acid analysis. Real-time PCR analysis of the patient revealed the first three exon deletion of the MLYCD gene, which was maternally inherited. DNA sequencing of the MLYCD gene of the patient identified a novel heterozygous mutation (c.911G>A, p.G304E) in exon 4 that was paternally inherited. The patient urine malonic acid dissolved and had a better school record in 6 month after initiation of fat-limited diet. At 1 year post treatment, the blood malonylcarnitine level decreased remarkably. Our result expands the phenotype of malonyl-CoA decarboxylase deficiency and suggests attentions should be paid to the mild form of disorders, for example, malonyl-CoA decarboxylase deficiency, which usually present a severe disease course.

摘要

丙二酰辅酶A脱羧酶缺乏症是一种极其罕见的常染色体隐性遗传的脂肪酸代谢先天性缺陷病。通常病情严重,若不治疗,预后较差。在此,我们报告一名患病的女性青少年,其临床和生化表型较轻,主要表现为学业不佳,无心肌病和代谢性酸中毒。因染色体微阵列检测发现16q23.3区域存在一个57 kb的缺失,涵盖MLCYD基因,故怀疑她患有丙二酰辅酶A脱羧酶缺乏症。随后通过酰基肉碱分析和有机酸分析确诊为丙二酰辅酶A脱羧酶缺乏症。对该患者进行实时PCR分析发现MLYCD基因的前三个外显子缺失,此为母系遗传。对患者MLYCD基因进行DNA测序,在第4外显子中发现一个新的杂合突变(c.911G>A,p.G304E),此为父系遗传。开始低脂饮食6个月后,患者尿液中的丙二酸溶解,学业成绩有所改善。治疗1年后,血液中丙二酰肉碱水平显著下降。我们的结果扩展了丙二酰辅酶A脱羧酶缺乏症的表型,并提示应关注疾病的轻症形式,例如通常病情严重的丙二酰辅酶A脱羧酶缺乏症。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验