ABCB4基因有两种变异的患者复发性早发性严重产科胆汁淤积。

IF 1.2 Q4 GASTROENTEROLOGY & HEPATOLOGY
Hepatology Forum Pub Date : 2023-09-20 eCollection Date: 2023-01-01 DOI:10.14744/hf.2023.2023.0008
Mohammad Fawad Khattak, Sam Thomson
{"title":"ABCB4基因有两种变异的患者复发性早发性严重产科胆汁淤积。","authors":"Mohammad Fawad Khattak,&nbsp;Sam Thomson","doi":"10.14744/hf.2023.2023.0008","DOIUrl":null,"url":null,"abstract":"<p><p>A 36-year-old patient presented with severe early-onset obstetric cholestasis on a background of having pre-term induction of labor at 33 weeks during her only previous pregnancy. The patient had significantly abnormal liver biochemistry with a bilirubin of 78 µmol/L, ALP of 318 u/L, ALT of 280 µmol/L, and bile acid levels of 420 µmol/L. The patient received ursodeoxycholic acid 750 mg 3 times a day, rifampicin 500 mg twice a day, aspirin 150 mg once a day, and metformin 500 mg 3 times a day. However, despite this, the patient still suffered from intractable pruritus and her bile acid level was still above the 100 µmol/L target that the obstetrics team was aiming for to avoid early delivery at 32 weeks. Due to the nature and severity of her cholestasis, the patient had a number of investigations done postnatally including genetic analysis, which confirmed that the patient was heterozygous for a pathogenic variant of the ATP-binding cassette subfamily B member 4 gene (c.959C>T [p.Ser320Phe]) and also a variant of unknown significance (c.1679C>T [p.Thr560Met]).</p>","PeriodicalId":29722,"journal":{"name":"Hepatology Forum","volume":null,"pages":null},"PeriodicalIF":1.2000,"publicationDate":"2023-09-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/31/68/hf-4-142.PMC10564254.pdf","citationCount":"0","resultStr":"{\"title\":\"Recurrent early-onset severe obstetric cholestasis in a patient with two variants in the ABCB4 gene.\",\"authors\":\"Mohammad Fawad Khattak,&nbsp;Sam Thomson\",\"doi\":\"10.14744/hf.2023.2023.0008\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>A 36-year-old patient presented with severe early-onset obstetric cholestasis on a background of having pre-term induction of labor at 33 weeks during her only previous pregnancy. The patient had significantly abnormal liver biochemistry with a bilirubin of 78 µmol/L, ALP of 318 u/L, ALT of 280 µmol/L, and bile acid levels of 420 µmol/L. The patient received ursodeoxycholic acid 750 mg 3 times a day, rifampicin 500 mg twice a day, aspirin 150 mg once a day, and metformin 500 mg 3 times a day. However, despite this, the patient still suffered from intractable pruritus and her bile acid level was still above the 100 µmol/L target that the obstetrics team was aiming for to avoid early delivery at 32 weeks. Due to the nature and severity of her cholestasis, the patient had a number of investigations done postnatally including genetic analysis, which confirmed that the patient was heterozygous for a pathogenic variant of the ATP-binding cassette subfamily B member 4 gene (c.959C>T [p.Ser320Phe]) and also a variant of unknown significance (c.1679C>T [p.Thr560Met]).</p>\",\"PeriodicalId\":29722,\"journal\":{\"name\":\"Hepatology Forum\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":1.2000,\"publicationDate\":\"2023-09-20\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/31/68/hf-4-142.PMC10564254.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Hepatology Forum\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.14744/hf.2023.2023.0008\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2023/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q4\",\"JCRName\":\"GASTROENTEROLOGY & HEPATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Hepatology Forum","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.14744/hf.2023.2023.0008","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2023/1/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"GASTROENTEROLOGY & HEPATOLOGY","Score":null,"Total":0}
引用次数: 0

摘要

一名36岁的患者在其唯一一次怀孕的33周时进行了早产,她出现了严重的早发性产科胆汁淤积。该患者的肝脏生物化学明显异常,胆红素为78µmol/L,ALP为318 u/L,ALT为280µmol/L,胆汁酸水平为420µmol/L。患者接受熊去氧胆酸750 mg,每日3次,利福平500 mg,每日2次,阿司匹林150 mg,二甲双胍500 mg,每天3次。然而,尽管如此,患者仍患有顽固性瘙痒症,胆汁酸水平仍高于产科团队为避免在32周时提前分娩而设定的100µmol/L目标。由于其胆汁淤积的性质和严重程度,患者在出生后进行了多项研究,包括基因分析,证实患者是ATP结合盒亚家族B成员4基因致病性变体的杂合子(c.959C>T[p.Ser320Phe])和意义未知的变体(c.1679C>T[p.Thr560Met])。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Recurrent early-onset severe obstetric cholestasis in a patient with two variants in the ABCB4 gene.

A 36-year-old patient presented with severe early-onset obstetric cholestasis on a background of having pre-term induction of labor at 33 weeks during her only previous pregnancy. The patient had significantly abnormal liver biochemistry with a bilirubin of 78 µmol/L, ALP of 318 u/L, ALT of 280 µmol/L, and bile acid levels of 420 µmol/L. The patient received ursodeoxycholic acid 750 mg 3 times a day, rifampicin 500 mg twice a day, aspirin 150 mg once a day, and metformin 500 mg 3 times a day. However, despite this, the patient still suffered from intractable pruritus and her bile acid level was still above the 100 µmol/L target that the obstetrics team was aiming for to avoid early delivery at 32 weeks. Due to the nature and severity of her cholestasis, the patient had a number of investigations done postnatally including genetic analysis, which confirmed that the patient was heterozygous for a pathogenic variant of the ATP-binding cassette subfamily B member 4 gene (c.959C>T [p.Ser320Phe]) and also a variant of unknown significance (c.1679C>T [p.Thr560Met]).

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
CiteScore
1.90
自引率
12.50%
发文量
0
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信