Variant von Willebrand's disease: characterization of two subtypes by analysis of multimeric composition of factor VIII/von Willebrand factor in plasma and platelets.

نویسندگان

  • Z M Ruggeri
  • T S Zimmerman
چکیده

We have examined the multimeric composition of factor VIII/von Willebrand factor in plasma and platelet lysates by means of sodium dodecyl sulfate agarose electrophoresis followed by staining with (125)I-labeled affinity-purified antibody. In normal plasma and platelet lysates, factor VIII/von Willebrand factor displayed 10 distinct multimers that ranged in apparent molecular weight from 0.86 to 9.9 x 10(6). The molecular weight difference between adjacent bands was 0.8-1.1 x 10(6). Larger material, not resolved into discrete bands, was also present with an average M(r) of 14.5 x 10(6). Though the dimer (apparent M(r) = 0.48 x 10(6)) and the monomer (apparent M(r) = 0.28 x 10(6)) generated by reduction of disulfide bonds were readily identified in this system, they were not detected in normal plasma or platelets. No differences were observed between fresh plasma prepared without anticoagulant and fresh or frozen plasma anticoagulated with either citrate or heparin. "Variant" (type II) von Willebrand's disease could be divided into two subtypes. In subtype IIA, factor VIII/von Willebrand factor in plasma consisted predominantly of the five smaller multimers with traces of the sixth and seventh (M(r) up to 4.5 x 10(6)). In subtype IIB, all these multimers were easily detected and, in addition, bands of intermediate size (M(r) = 8.5 x 10(6) and smaller) were present. In contrast, the multimeric composition of IIB platelet factor VIII/von Willebrand factor was identical to normal, whereas in subtype IIA the larger multimers were absent from platelets as well as from plasma. In subtype IIB, binding of factor VIII/von Willebrand factor to platelets occurred at lower concentrations of ristocetin than required for normal and multimers of smaller size than in normal bound. On the contrary, in subtype IIA, binding was minimal, as was true of normal factor VIII/von Willebrand factor of equivalent size. Thus, physical as well as functional differences in the two subtypes of variant von Willebrand's disease described suggest that different pathogenetic mechanisms underlie the factor VIII/von Willebrand factor abnormalities in these patients.

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

منابع مشابه

Multimeric composition of factor VIII/von Willebrand factor following administration of DDAVP: implications for pathophysiology and therapy of von Willebrand's disease subtypes.

We have studied the modifications in the multimeric composition of plasma factor VIII/von Willebrand factor and the bleeding time response following administration of 1-Deamino-[8-D-arginine]-Vasopressin (DDAVP) to patients with different subtypes of von Willebrand's disease. In type I, all multimers were present in plasma in the resting state, though they were decreased in concentration. Admin...

متن کامل

Effect of multimeric structure of the factor VIII/von Willebrand factor protein on binding to platelets.

The characteristics of the intact factor VIII/von Willebrand factor protein binding to human platelets was compared to 2-mercaptoethanol-treated factor VIII/von Willebrand factor protein and to fractions of plasma factor VIII/von Willebrand factor protein that elute after the void volume. These studies indicate that the factor VIII/von Willebrand factor protein larger size oligomers bind prefer...

متن کامل

The complex multimeric composition of factor VIII/von Willebrand factor.

We have analyzed the multimeric structure of factor VIII/von Willebrand factor in plasma by sodium dodecyl sulfate electrophoresis using gels of varying porosity and a discontinuous buffer system. Factor VIII/von Willebrand factor bands were identified by reaction with 125I-labeled affinity-purified antibody and subsequent autoradiography. In 1% agarose gels, normal plasma displayed a series of...

متن کامل

Variant von Willebrand's disease type B--revisited.

Results of investigations of the factor VIII (FVIII) of a patient with an unusual variant form of von Willebrand's disease (vWD) are presented. A two-peak crossed-immunoelectrophoresis (CIE) pattern was seen when fresh plasma was electrophoresed, but the CIE pattern became normal by incubating the plasma at 37 degree C for more than 72 hr. The two peaks on CIE were separated by cryoprecipitatio...

متن کامل

A new variant of dominant type II von Willebrand's disease with aberrant multimeric pattern of factor VIII-related antigen (type IID).

A new type II variant form of von Willebrand's disease has been recognized in a mother and daughter who have bleeding manifestations typical of von Willebrand's disease. Laboratory findings include consistently prolonged bleeding times, with normal levels of factor VIII procoagulant and antigen, but decreased ristocetin cofactor activity. Electrophoresis in SDS 1.5% agarose gel and reaction wit...

متن کامل

ذخیره در منابع من


  با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

عنوان ژورنال:
  • The Journal of clinical investigation

دوره 65 6  شماره 

صفحات  -

تاریخ انتشار 1980