Literature DB >> 2987704

Detection and sequence of mutations in the factor VIII gene of haemophiliacs.

J Gitschier, W I Wood, E G Tuddenham, M A Shuman, T M Goralka, E Y Chen, R M Lawn.   

Abstract

The most common inherited bleeding disorder in man, haemophilia A, is caused by defect in factor VIII, a component in the blood coagulation pathway. The X-chromosome-linked disease almost certainly stems from a heterogeneous collection of genetic lesions. Because, without proper treatment, haemophilia can be a fatal disease, new mutations are necessary to account for its constant frequency in the population. In addition, haemophilia A displays a wide range of severity, and some 15% of haemophiliacs generate high levels of antibodies against factor VIII ('inhibitor patients'). The present work elucidates the molecular genetic basis of haemophilia in some individuals. Using the recently cloned factor VIII gene as a probe, we have identified two different nonsense point mutations in the factor VIII gene of haemophiliacs, as well as two different partial deletions of the gene. Our survey of 92 haemophiliacs indicates no firm correlation between antibody (inhibitor) production and gross gene defects.

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Year:  1985        PMID: 2987704     DOI: 10.1038/315427a0

Source DB:  PubMed          Journal:  Nature        ISSN: 0028-0836            Impact factor:   49.962


  59 in total

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3.  Sex ratio of the mutation frequencies in haemophilia A: coagulation assays and RFLP analysis.

Authors:  A H Bröcker-Vriends; F R Rosendaal; J C van Houwelingen; E Bakker; G J van Ommen; J J van de Kamp; E Briët
Journal:  J Med Genet       Date:  1991-10       Impact factor: 6.318

4.  Defining DNA diagnostic tests appropriate or standard clinical care.

Authors:  R V Lebo; G Cunningham; M J Simons; L J Shapiro
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5.  Molecular analysis of hemophilia A mutations in the Finnish population.

Authors:  B Levinson; A E Lehesjoki; A de la Chapelle; J Gitschier
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6.  TaqI polymorphism in the LDL receptor gene and a TaqI 1.5-kb band associated with familial hypercholesterolemia.

Authors:  K Yamakawa; T Okafuji; Y Iwamura; K Yuzawa; J Satoh; N Hattori; Y Yamanouchi; H Yanagi; K Kawai; S Tsuchiya
Journal:  Hum Genet       Date:  1988-09       Impact factor: 4.132

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8.  Mutations and a polymorphism in the factor VIII gene discovered by denaturing gradient gel electrophoresis.

Authors:  S Kogan; J Gitschier
Journal:  Proc Natl Acad Sci U S A       Date:  1990-03       Impact factor: 11.205

9.  An intronic region within the human factor VIII gene is duplicated within Xq28 and is homologous to the polymorphic locus DXS115 (767).

Authors:  M Patterson; J Gitschier; J Bloomfield; M Bell; H Dorkins; U Froster-Iskenius; S Sommer; J Sobell; D Schaid; S Thibodeau
Journal:  Am J Hum Genet       Date:  1989-05       Impact factor: 11.025

10.  The molecular genetics of haemophilia A: screening for point mutations in the factor VIII gene using the restriction enzyme TaqI.

Authors:  D S Millar; B Zoll; U Martinowitz; V V Kakkar; D N Cooper
Journal:  Hum Genet       Date:  1991-09       Impact factor: 4.132

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