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B-cell lymphoma in a patient with complete interferon gamma receptor 1 deficiency.

Abstract：

:Immunosuppression-associated lymphoproliferative disorders can be related to primary as well as acquired immune disorders. Interferon gamma receptor (IFN-γR) deficiency is a rare primary immune disorder, characterized by increased susceptibility to mycobacterial infections. Here we report the first case of an Epstein Barr Virus (EBV) related B-cell lymphoma in a patient with complete IFN-γR1 deficiency. The patient was a 20-year-old man with homozygous 22Cdel in IFNGR1 resulting in complete absence of IFN-γR1 surface expression and complete lack of responsiveness to IFN-γ in vitro. He had disseminated refractory Mycobacterium avium complex and Mycobacterium abscessus infections. At age 18 he presented with new spiking fever and weight loss that was due to an EBV-positive B-cell non-Hodgkin lymphoma. Two years later he died of progressive lymphoma. IFN-γ plays an important role in tumor protection and rejection. Patients with IFN-γR deficiencies and other immune deficits predisposing to mycobacterial disease seem to have an increased risk of malignancies, especially those related to viral infections. As more of these patients survive their early infections, cancer awareness and tumor surveillance may need to become a more routine part of management.

journal_name

J Clin Immunol

journal_title

Journal of clinical immunology

authors

Bax HI,Freeman AF,Anderson VL,Vesterhus P,Laerum D,Pittaluga S,Wilson WH,Holland SM

doi

10.1007/s10875-013-9907-0

subject

Has Abstract

pub_date

2013-08-01 00:00:00

pages

1062-6

issue

6

eissn

0271-9142

issn

1573-2592

journal_volume

33

pub_type

杂志文章

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