Literature DB >> 198660

Variable phenotypic expression of an X-linked recessive lymphoproliferative syndrome.

D T Purtilo, D DeFlorio, L M Hutt, J Bhawan, J P Yang, R Otto, W Edwards.   

Abstract

Investigation of a family with cancer in boys revealed that at least 20 males had the X-linked recessive lymphoproliferative syndrome. A variety of phenotypes occurred: aproliferative phenotypes consisted of aplastic anemia, agranulocytosis or acquired hypogammaglobulinemia; and proliferative phenotypes of B cells included disorders associated with the Epstein-Barr virus, American Burkitt's lymphoma, immunoblastic sarcoma of B cells, fatal infectious mononucleosis or plasmacytoma. The lymphoproliferative disorders observed in males could have resulted from an immunodeficiency to Epstein-Barr virus. The variable phenotypic expression could have resulted from individual differences in the viral dose, duration of exposure and age at which the boys were exposed to the virus. Aproliferative phenotypes such as acquired hypogammaglobulinemia could have ensued from excessive suppressor-cell activity on B cells, whereas proliferative phenotypes such as Burkitt's lymphoma or fatal infectious mononucleosis could have resulted from infection by Epstein-Barr virus and failure to stop proliferation of B cells.

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Year:  1977        PMID: 198660     DOI: 10.1056/NEJM197711172972001

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  39 in total

1.  Severe neutropenia in infectious mononucleosis.

Authors:  W P Hammond; J M Harlan; S E Steinberg
Journal:  West J Med       Date:  1979-08

2.  B-cell lymphoproliferation and lymphomagenesis are associated with clonotypic intracellular terminal regions of the Epstein-Barr virus.

Authors:  N A Brown; C R Liu; Y F Wang; C R Garcia
Journal:  J Virol       Date:  1988-03       Impact factor: 5.103

Review 3.  Lymphocyte function and virus infections.

Authors:  A M Denman
Journal:  J Clin Pathol Suppl (R Coll Pathol)       Date:  1979

Review 4.  Genetics of neoplasia--impact of ecogenetics on oncogenesis. A review.

Authors:  D T Purtilo; L Paquin; T Gindhart
Journal:  Am J Pathol       Date:  1978-06       Impact factor: 4.307

5.  An Epstein-Barr-related herpesvirus from marmoset lymphomas.

Authors:  Y Cho; J Ramer; P Rivailler; C Quink; R L Garber; D R Beier; F Wang
Journal:  Proc Natl Acad Sci U S A       Date:  2001-01-30       Impact factor: 11.205

6.  Effects of nucleoside analogs on Epstein-Barr virus-induced transformation of human umbilical cord leukocytes and Epstein-Barr virus expression in transformed cells.

Authors:  E E Henderson; W K Long; R Ribecky
Journal:  Antimicrob Agents Chemother       Date:  1979-01       Impact factor: 5.191

Review 7.  X-linked lymphoproliferative disease (XLP) as a model of Epstein-Barr virus-induced immunopathology.

Authors:  D T Purtilo
Journal:  Springer Semin Immunopathol       Date:  1991

Review 8.  Lymphoma in patients with rheumatoid arthritis: what is the evidence of a link with methotrexate?

Authors:  L Georgescu; S A Paget
Journal:  Drug Saf       Date:  1999-06       Impact factor: 5.606

9.  Epstein-Barr virus nuclear antigen 2 transactivates latent membrane protein LMP1.

Authors:  F Wang; S F Tsang; M G Kurilla; J I Cohen; E Kieff
Journal:  J Virol       Date:  1990-07       Impact factor: 5.103

10.  Frequent mutations in SH2D1A (XLP) in males presenting with high-grade mature B-cell neoplasms.

Authors:  J T Sandlund; S A Shurtleff; M Onciu; E Horwitz; W Leung; V Howard; R Rencher; M E Conley
Journal:  Pediatr Blood Cancer       Date:  2013-04-15       Impact factor: 3.167

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