IF 1.7 4区 医学 Q3 HEMATOLOGY Acta Haematologica Pub Date : 2023-01-01 DOI:10.1159/000527952
Shengchen Ge, Yuqing Luo, Rujiao Dong, Xiaoli Guo, Mingshan Wang, Yi Chen
{"title":"Two Novel Heterozygous Mutations (p.γPhe230Val and p.AαAsn839Thr) Cause Hereditary Hypodysfibrinogenemia in Two Chinese Independent Families.","authors":"Shengchen Ge,&nbsp;Yuqing Luo,&nbsp;Rujiao Dong,&nbsp;Xiaoli Guo,&nbsp;Mingshan Wang,&nbsp;Yi Chen","doi":"10.1159/000527952","DOIUrl":null,"url":null,"abstract":"<p><p>The objective of this study was to explore the molecular defects in two Chinese families with hypodysfibrinogenemia. The coagulation method and immunoturbidimetric method were used to detect plasma fibrinogen activity and plasma fibrinogen antigen. The fibrinogen genes were amplified by PCR, and suspected mutations were confirmed by reverse sequencing. Bioinformatics and model analysis were used to study the conservatism and harm of the mutations. Study showed that the Fg:C and Fg:Ag of the probands of the two families were reduced, respectively, to 0.80g/L, 0.92g/L and 1.35g/L, 1.42g/L; gene analysis revealed that the proband 1 had a heterozygous missense mutation of c.688T>G (p.γPhe230Val) in exon 7 of the FGG gene; the c.2516A>C (p.AαAsn839Thr) heterozygous missense mutation in exon 6 of the FGA gene was got by the proband 2. These mutations found in this study might be related to the hypodysfibrinogenemia.</p>","PeriodicalId":6981,"journal":{"name":"Acta Haematologica","volume":"146 3","pages":"252-258"},"PeriodicalIF":1.7000,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Acta Haematologica","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1159/000527952","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"HEMATOLOGY","Score":null,"Total":0}
引用次数: 0

摘要

采用凝血法和免疫比浊法检测血浆纤维蛋白原活性和血浆纤维蛋白原抗原。采用PCR扩增纤维蛋白原基因,反向测序证实可疑突变。利用生物信息学和模型分析方法研究突变的保守性和危害。研究表明,两个家族先证者的Fg:C和Fg:Ag分别降至0.80g/L、0.92g/L和1.35g/L、1.42g/L;基因分析显示,先证者1在FGG基因7外显子上发生c.688T>G (p.γPhe230Val)杂合错义突变;先证者2在FGA基因6外显子上发现C . 2516a >C (p.AαAsn839Thr)杂合错义突变。本研究发现的这些突变可能与低纤维蛋白异常血症有关。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
Two Novel Heterozygous Mutations (p.γPhe230Val and p.AαAsn839Thr) Cause Hereditary Hypodysfibrinogenemia in Two Chinese Independent Families.

The objective of this study was to explore the molecular defects in two Chinese families with hypodysfibrinogenemia. The coagulation method and immunoturbidimetric method were used to detect plasma fibrinogen activity and plasma fibrinogen antigen. The fibrinogen genes were amplified by PCR, and suspected mutations were confirmed by reverse sequencing. Bioinformatics and model analysis were used to study the conservatism and harm of the mutations. Study showed that the Fg:C and Fg:Ag of the probands of the two families were reduced, respectively, to 0.80g/L, 0.92g/L and 1.35g/L, 1.42g/L; gene analysis revealed that the proband 1 had a heterozygous missense mutation of c.688T>G (p.γPhe230Val) in exon 7 of the FGG gene; the c.2516A>C (p.AαAsn839Thr) heterozygous missense mutation in exon 6 of the FGA gene was got by the proband 2. These mutations found in this study might be related to the hypodysfibrinogenemia.

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
Acta Haematologica
Acta Haematologica 医学-血液学
CiteScore
4.90
自引率
0.00%
发文量
61
审稿时长
6-12 weeks
期刊介绍: ''Acta Haematologica'' is a well-established and internationally recognized clinically-oriented journal featuring balanced, wide-ranging coverage of current hematology research. A wealth of information on such problems as anemia, leukemia, lymphoma, multiple myeloma, hereditary disorders, blood coagulation, growth factors, hematopoiesis and differentiation is contained in first-rate basic and clinical papers some of which are accompanied by editorial comments by eminent experts. These are supplemented by short state-of-the-art communications, reviews and correspondence as well as occasional special issues devoted to ‘hot topics’ in hematology. These will keep the practicing hematologist well informed of the new developments in the field.
期刊最新文献
Shorter duration of Blinatumomab administration to 14 days has same efficacy and safety profile in treatment of relapsed/refractory B-cell precursor acute lymphoblastic leukemia: A retrospective single-center study. Thiamine-Responsive Megaloblastic Anemia Syndrome Mimicking Myelodysplastic Neoplasm. Efficacy of haploidentical allogeneic hematopoietic cell transplantation following two courses of venetoclax and azacytidine therapy in patients over 55 years old with acute myelogenous leukemia. Real-World Use of Ruxolitinib in Patients with Myelofibrosis and Anemia or Thrombocytopenia at Diagnosis. Sudden Blast Crisis in a chronic myeloid leukemia patient in Treatment Free Remission: A case report and literature review.
×
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
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 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 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1