Literature DB >> 19923982

Inherited dysfibrinogenemia: clinical phenotypes associated with five different fibrinogen structure defects.

Wolfgang Miesbach1, Inge Scharrer, Agnes Henschen, Marguerite Neerman-Arbez, Silvia Spitzer, Dennis Galanakis.   

Abstract

Hereditary dysfibrinogenemia is a rare clotting disorder, which results from mutations in at least one of the three fibrinogen genes. We examined the frequency of hemostatic clinical and laboratory anomalies at presentation of 37 probands from 12 unrelated families with five different defects (Aalpha R16C, gamma A357T, gamma318-319 del, gamma M310T, and Aalpha R16S), among. The median age was 51 years (11-86 years). Among 62% who were women three (13%) had experienced one or more spontaneous abortion. More than half of the probands had experienced one or more undue bleeding episode, easy bruising being by far the most common. In 19% of probands (9/37, all above age of 50 years), had experienced at least one episode of arterial or venous thrombosis. Among these, were two (7%) with deep venous thrombosis, seven with arterial thrombosis, and five (14%) had experienced both. We propose that the higher frequency of prolonged PT than aPTT, in ours and in other reported studies, reflects the polymerization delay, which in aPTT is attenuated owing to contact activation prior to calcium addition.

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Year:  2010        PMID: 19923982     DOI: 10.1097/MBC.0b013e328331e6db

Source DB:  PubMed          Journal:  Blood Coagul Fibrinolysis        ISSN: 0957-5235            Impact factor:   1.276


  8 in total

Review 1.  Thrombosis in Inherited Fibrinogen Disorders.

Authors:  Wolfgang Korte; Man-Chiu Poon; Alfonso Iorio; Michael Makris
Journal:  Transfus Med Hemother       Date:  2017-03-14       Impact factor: 3.747

2.  Fibrinogen Tokushima II: a new case of congenital dysfibrinogenemia with a γ methionine-310 to threonine substitution.

Authors:  Toshio Shigekiyo; Etsuko Sekimoto; Atsuhisa Shirakami; Hiroshi Yamaguchi; Hironobu Shibata; Shuji Ozaki; Masahiko Maegawa; Ken-ichi Aihara
Journal:  Int J Hematol       Date:  2012-07-27       Impact factor: 2.490

Review 3.  New findings on venous thrombogenesis.

Authors:  James R Byrnes; Alisa S Wolberg
Journal:  Hamostaseologie       Date:  2016-11-23       Impact factor: 1.778

4.  Natural history of patients with congenital dysfibrinogenemia.

Authors:  Alessandro Casini; Marc Blondon; Aurélien Lebreton; Jérémie Koegel; Véronique Tintillier; Emmanuel de Maistre; Philippe Gautier; Christine Biron; Marguerite Neerman-Arbez; Philippe de Moerloose
Journal:  Blood       Date:  2014-10-15       Impact factor: 22.113

Review 5.  Abnormal fibrinogen with an Aα 16Arg → Cys substitution is associated with multiple cerebral infarctions.

Authors:  Meiling Luo; Aiqiu Wei; Liqun Xiang; Jie Yan; Lin Liao; Xuelian Deng; Donghong Deng; Peng Cheng; Faquan Lin
Journal:  J Thromb Thrombolysis       Date:  2018-10       Impact factor: 2.300

6.  c.259A>C in the fibrinogen gene of alpha chain (FGA) is a fibrinogen with thrombotic phenotype.

Authors:  Ophira Salomon; Ortal Barel; Eran Eyal; Reut Shnerb Ganor; Yeroham Kleinbaum; Mordechai Shohat
Journal:  Appl Clin Genet       Date:  2019-02-28

7.  A Chinese family with congenital Dysfibrinogenemia carries a heterozygous missense mutation in FGA: Concerning the genetic abnormality and clinical treatment.

Authors:  Jihao Zhou; Peng Zhu; Xinyou Zhang
Journal:  Pak J Med Sci       Date:  2017 Jul-Aug       Impact factor: 1.088

8.  Three cases of congenital dysfibrinogenemia in unrelated Chinese families: heterozygous missense mutation in fibrinogen alpha chain Argl6His.

Authors:  Meiling Luo; Donghong Deng; Liqun Xiang; Peng Cheng; Lin Liao; Xuelian Deng; Jie Yan; Faquan Lin
Journal:  Medicine (Baltimore)       Date:  2016-09       Impact factor: 1.889

  8 in total

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