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韩国遗传性尿素循环障碍中精氨琥珀酸裂解酶缺乏症的患病率较低。

Low prevalence of argininosuccinate lyase deficiency among inherited urea cycle disorders in Korea.

机构信息

Department of Pediatrics, Asan Medical Center Children's Hospital, University of Ulsan College of Medicine, Seoul, Korea.

Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.

出版信息

J Hum Genet. 2018 Jul;63(8):911-917. doi: 10.1038/s10038-018-0467-2. Epub 2018 May 17.

Abstract

Argininosuccinic aciduria (ASA), which is considered to be the second most common urea cycle disorder (UCD), is caused by an argininosuccinate lyase deficiency and is biochemically characterized by elevation of argininosuccinic acid and arginine deficiency. In addition to hyperammonemia, other characteristic features of ASA include hepatic fibrosis, hypertension, neurocognitive deficiencies, and trichorrhexis nodosa. Herein, we retrospectively reviewed the clinical findings, biochemical profiles, and genotypic characteristics of five Korean patients with ASA, who showed typical phenotypes and biochemical findings of the disease. Molecular analysis of these patients revealed six novel ASL mutations. Next, we investigated the prevalence of all types of UCDs in Korea. Of note, over a two decade periods, ASA was only detected in 6.3% of patients with a UCD, which made it the fourth most common UCD in Korea. In comparison with Caucasians, in whom ASA is the second most common UCD, ASA is comparatively rare in East Asian populations, including Japanese and Koreans. These findings suggest the possibility of geographic variation in UCDs among ethnic groups.

摘要

精氨琥珀酸尿症(ASA)被认为是第二常见的尿素循环障碍(UCD),由精氨琥珀酸裂解酶缺乏引起,其生化特征为精氨琥珀酸和精氨酸升高。除高氨血症外,ASA 的其他特征性表现包括肝纤维化、高血压、神经认知缺陷和结节性脆发病。在此,我们回顾性分析了 5 例韩国 ASA 患者的临床发现、生化特征和基因型特征,这些患者表现出疾病的典型表型和生化表现。对这些患者的分子分析显示了 6 种新的 ASL 突变。接下来,我们调查了韩国所有类型 UCD 的患病率。值得注意的是,在 20 多年的时间里,ASA 仅在 6.3%的 UCD 患者中被检出,使其成为韩国第四大常见的 UCD。与白种人相比,ASA 是第二常见的 UCD,在包括日本人在内的东亚人群中,ASA 较为罕见。这些发现提示 UCD 在不同种族之间存在地理变异的可能性。

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