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血浆输注联合螯合疗法可缓解单一 Arg778Leu 杂合突变所致暴发性威尔逊病。

Plasma transfusion combined with chelating therapy alleviates fulminant Wilson's disease with a single Arg778Leu heterozygote mutation.

机构信息

Institute of Hepatology, The Third People's Hospital of Changzhou, Changzhou, Jiangsu 213000, China; Department of Liver Diseases, The Third People's Hospital of Changzhou, Changzhou, Jiangsu 213000, China.

Department of Liver Diseases, The Third People's Hospital of Changzhou, Changzhou, Jiangsu 213000, China.

出版信息

Ann Hepatol. 2019 Mar-Apr;18(2):393-396. doi: 10.1016/j.aohep.2018.07.001. Epub 2019 Apr 12.

Abstract

Wilson's disease (WD), resulting from homozygote and compound heterozygote mutations in ATB7B, is an autosomal recessive disease. WD associated acute liver failure (ALF) is fatal, and a revised Wilson's disease prognostic index (RWPI) >11 is a reliable indication of liver transplantation (LT) or artificial liver support (ALS). We described a WD patient who initially presented with ALF and severe hemolytic anemia. A single heterozygote c.2333G>T mutation (p. Arg778Leu, R778L) in ATP7B was screened by whole exome sequencing and validated by Sanger sequencing. Rapid diagnostic criteria (ALP/TBIL <4 and AST/ALT >2.2) are suitable for early diagnosis. Although the RWPI amounted to 15, the patient recovered after intermittent plasma transfusion and subsequent chelating therapy without LT or ALS. In conclusion, WD patients with a single R778L heterozygote mutation can present with ALF as the initial clinical manifestation, and intermittent plasma transfusion combined with chelating therapy may alleviate fulminant WD without LT or ALS.

摘要

威尔逊病(WD)是一种常染色体隐性遗传病,由 ATB7B 基因的纯合子和复合杂合子突变引起。WD 相关的急性肝衰竭(ALF)是致命的,修订后的威尔逊病预后指数(RWPI)>11 是肝移植(LT)或人工肝支持(ALS)的可靠指征。我们描述了一位 WD 患者,最初表现为 ALF 和严重溶血性贫血。通过外显子组测序筛选出 ATP7B 中的单个杂合子 c.2333G>T 突变(p.Arg778Leu,R778L),并通过 Sanger 测序进行验证。快速诊断标准(ALP/TBIL<4 和 AST/ALT>2.2)适用于早期诊断。尽管 RWPI 达到 15,但患者在间歇性血浆输注和随后的螯合治疗后无需 LT 或 ALS 即可恢复。总之,携带单个 R778L 杂合突变的 WD 患者可表现为 ALF 作为首发临床表现,间歇性血浆输注联合螯合治疗可能缓解无 LT 或 ALS 的暴发性 WD。

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