Induction of tolerance to factor VIII in a child with a high-titer inhibitor: in vitro and in vivo observations
| Autor: | Jacques M. Lemire, Joseph A. Church, Rebecca Sakai, Stanley C. Jordan, Edward D. Gomperts |
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| Rok vydání: | 1984 |
| Předmět: |
Male
Clinical Trials as Topic Factor VIII business.industry Hemophilia A In vitro Antibodies Titer Regimen Immunoglobulin M In vivo hemic and lymphatic diseases Child Preschool Immunoglobulin G Pediatrics Perinatology and Child Health Immunology Immune Tolerance Medicine Humans Lymphocytes Antigens business Bypassing agent |
| Zdroj: | The Journal of pediatrics. 104(1) |
| ISSN: | 0022-3476 |
| Popis: | High dose factor VIII concentrate was infused over 12 months in a 3-year-old child with hemophilia A and a high-titer inhibitor. This regimen was successful in producing clinical tolerance to the factor VIII and was associated with a fall of inhibitor titer from 85 BU to less than 1 BU. Subsequently, a rise in inhibitor to peak at 3.5 BU correlated with a decrease in infusion dose. In vitro studies using a microdroplet assay for immunoglobulin synthesis in the presence of varying concentrations of factor VIII was carried out prior to the clinical trial. This system indicated that the patient's peripheral blood mononuclear cells were stimulated to release IgG by 0.01 U/ml factor VIII coagulant activity and inhibited at 0.9 U/ml FVIIIc. Lymphocyte subsets were monitored during the course of therapy. The ratio of Leu-3a+/Leu-2a+ (helper/suppressor) varied considerably, fluctuating between a peak of 4.4 and low point of 0.6, with the initial pretrial ratio being 1.2. |
| Databáze: | OpenAIRE |
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