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PMID: 6982283 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Aberrant multimeric structure of von Willebrand factor in a new variant of von Willebrand's disease (type IIC).

The Journal of clinical investigation ·Vol. 70 ·No. 5 ·1982-11-00 ·Pages 1124-7

Ruggeri ZM, Nilsson IM, Lombardi R, Holmberg L, Zimmerman TS

Abstract

A variant of von Willebrand's disease has been identified in which sodium dodecyl sulfate agarose electrophoresis provides evidence that the von Willebrand factor present is structurally abnormal. Rather than the repeating triplet seen in normal subjects and in patients with the IIA and IIB variants, a repeating doublet was present in the propositus. None of the bands had the same mobility as bands in normal subjects or previously described von Willebrand's disease patients. The larger multimers of von Willebrand factor were lacking both from plasma and platelets, and did not appear in the circulation after infusion of 1-deamino-[8-D-arginine]-vasopressin. There was a marked increase in the concentration of the smallest multimer in the propositus and his phenotypically normal children, indicating that this abnormality of von Willebrand factor is inherited in an autosomal-recessive manner.

MeSH Terms
Adult Blood Coagulation Factors/genetics Electrophoresis, Polyacrylamide Gel Female Humans Male von Willebrand Diseases/blood,genetics von Willebrand Factor/analysis,genetics
Chemicals
Blood Coagulation Factors von Willebrand Factor
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Ruggeri Z M
Nilsson I M
Lombardi R
Holmberg L
Zimmerman T S
References (8)
8 references, click to expand
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Article Info
Journal
The Journal of clinical investigation
Abbr.
J Clin Invest
ISSN
0021-9738
Published
1982-11-00
Pages
1124-7
Language
English
Region
United States
NLM ID
7802877
PMCID
PMC370325
Subset
IM
Grants
NHLBI NIH HHS · HL 15491 · United States
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