Abstract
A four month old dizygotic triplet with pseudomonas sepsis and disseminated intravascular coagulopathy necessitating platelet tranfusions was subsequently found to have severe combined immunodeficiency (SCID) with thymic dysplasia and mild cutaneous graft-vs-host disease (GvHD). T lymphocytes from the mother and both platelet donors but not from the triplets were identified in the patient's blood by HLA typing. Non-T lymphocytes displayed the same maternal and paternal haplotypes as the other triplet siblings and absorption of lymphocytotoxic HLA antibodies by the patient's platelets confirmed the patient's HLA genotype.
GvHD resolved during ablative therapy with anti-thymocyte globulin and cyclophosphamide and did not recur following bone marrow transplantation (BMT) from an HLA identical triplet. One month after BMT, T lymphocytes from one platelet donor, but not from the mother, were still found in the patient's blood.
Since GvHD from non-irradiated blood transfusions in SCID patients is fatal without treatment, we hypothesize that intrauterine engraftment of maternal cells and/or mild GvH reaction induced by these cells suppressed the GvHD from the platelet donor's lymphocytes. An exacerbation of the GvHD after maternal cells disappeared may have been prevented by immune function from the BMT. Thus, intrauterine engraftment of maternal T lymphocytes, which usually causes limited GvHD, may modulate the GvHD due to postnatally transplanted T lymphocyes.
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Mamlok, R., Goldblum, R., Vaidya, S. et al. EVIDENCE FOR MODULATION OF GRAFT-VS-HOST DISEASE BY INTRAUTERINE ENGRAFTED MATERNAL T LYMPHOCYTES. Pediatr Res 21 (Suppl 4), 314 (1987). https://doi.org/10.1203/00006450-198704010-00881
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DOI: https://doi.org/10.1203/00006450-198704010-00881