Document Detail


JAK2 V617F-positive essential thrombocythemia in a patient with Klinefelter syndrome: a case report.
MedLine Citation:
PMID:  20362232     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Klinefelter syndrome (KS) is the most commonly diagnosed X chromosome aneuploid syndrome among males. The association between hematologic malignancies such as non-Hodgkin lymphoma and leukemia and KS has been established recently on the basis of numerous case reports and a large cohort study. The risk of chronic myeloproliferative disease (MPD) as a hematologic malignancy in KS, however, has not been evaluated to date. Moreover, to our knowledge, there is only one case report that observed MPD in a patient with KS. Even though the Janus kinase 2 (JAK2) mutation was considered the primary cause in the pathogenesis of MPD, the mutation status was not described in that report. We found JAK2 V617F mutation-positive essential thromobocythemia (ET) in a patient with KS. To the best of our knowledge, this case represents the first case report of JAK2 V617F mutation in a patient with KS. Furthermore, we also suggest the hypothesis that X chromosome aneuploidy as a "pre-JAK2" cytogenetic change may be associated with the development of clonal hematopoiesis and carcinogenesis, because of the finding of higher oncogenic potential of extra X chromosome. In this study, we present the first report of JAK2 V617F-positive ET in a patient with KS, as well as a review of the relevant literature.
Authors:
Ho-Young Yhim; Hee Sun Kim; Ji-Youn Sohn; Min-Ju Song; Na-Ri Lee; Eun-Kee Song; Sam-Im Choi; Chang-Yeol Yim; Jae-Yong Kwak
Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Cancer genetics and cytogenetics     Volume:  198     ISSN:  1873-4456     ISO Abbreviation:  Cancer Genet. Cytogenet.     Publication Date:  2010 Apr 
Date Detail:
Created Date:  2010-04-05     Completed Date:  2010-04-26     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  7909240     Medline TA:  Cancer Genet Cytogenet     Country:  United States    
Other Details:
Languages:  eng     Pagination:  162-5     Citation Subset:  IM    
Copyright Information:
Copyright 2010 Elsevier Inc. All rights reserved.
Affiliation:
Division of Hematology/Oncology, Department of Internal Medicine, Chonbuk National University Medical School, Jeonju, 561-712 Korea.
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MeSH Terms
Descriptor/Qualifier:
Adult
Amino Acid Substitution / genetics
Chromosome Banding
Humans
Janus Kinase 2 / genetics*,  metabolism
Klinefelter Syndrome / complications,  genetics*
Male
Mutation, Missense / physiology
Phenylalanine / genetics
Thrombocythemia, Essential / complications,  genetics*
Valine / genetics
Chemical
Reg. No./Substance:
63-91-2/Phenylalanine; 7004-03-7/Valine; EC 2.7.1.112/JAK2 protein, human; EC 2.7.10.1/Janus Kinase 2

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