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- Title
Parvovirus B19-Induced Anemia as the Presenting Manifestation of X-Linked Hyper-IgM Syndrome.
- Authors
Seyama, Kuniaki; Kobayashi, Roger; Hasle, Henrik; Apter, Andrea J.; Rutledge, Joe C.; Rosen, David; Ochs, Hans D.
- Abstract
Parvovirus B19 (B19) can cause chronic anemia due to persistent infection in immunocompromised hosts who cannot produce neutralizing antibody necessary for clearing B19. Three patients with X-linked hyper-IgM syndrome (XHIM), who were all asymptomatic until they developed B19-induced chronic anemia at the ages of 8, 14, and 17 years, respectively, were found to have mutations of the CD40L gene, including a missense mutation (T254M), a nonsense mutation resulting in a new initiation codon and loss of the intracellular domain (R11X), and a splice site mutation (nt 309+2t→a). Antibody responses to the T cell-dependent antigen, bacteriophage φX174, were impaired, but neutralizing antibody titers were higher than in XHIM patients with classic phenotype. All 3 patients responded to intravenous immune globulin (IVIG) treatment. Certain mutations of the CD40L gene result in a mild XHIM phenotype that may become apparent following B19 infection in patients not on IVIG therapy and therefore not protected from B19 infection.
- Publication
Journal of Infectious Diseases, 1998, Vol 178, Issue 2, p318
- ISSN
0022-1899
- Publication type
Article
- DOI
10.1086/515633