Autoimmune polyglandular syndrome type II after bone marrow transplant: real transfer or acceleration of a programmed disease?
We report a case of autoimmune polyglandular syndrome type II that developed in an 11-year-old boy with homozygous sickle cell disease after allogeneic bone marrow transplant; the donor was his father, who was human leukocyte antigen identical and had vitiligo. On day 24 after transplant, the patient developed grade 1 acute graft-versus-host disease, which was controlled over a period of 3 months with corticosteroid-induced immunosuppression. Full donor engraftment was documented on day 31 after transplant, and this was further confirmed on days 59, 231, 321, 472, 549, and 720. Three months after transplant, the recipient developed adrenal insufficiency, and at 13 months, he developed vitiligo. Seventeen months after transplant, autoimmune thyroid disease, positive for thyroid peroxidase and thyroglobulin autoantibodies, was diagnosed. At the same time, we identified adrenal insufficiency in the donor. We analyzed a serum sample from the recipient for autoantibody markers for type 1 autoimmune diabetes mellitus. The sample was positive for antiglutamic acid decarboxylase. Antibody against 21-hydroxylase enzyme was also found (261 U/mL; normal value, < 1 U/mL). We conclude that the recipient developed autoimmune polyglandular syndrome type II after bone marrow transplant from his father, who was probably affected by the same syndrome.
Service d'Immuno-Hematologie Pediatrique, Centre National de Greffe de Moelle Osseuse, Centre National de Transfusion Sanguine, Tunis, Tunisia.
SourceExperimental and clinical transplantation : official journal of the Middle East Society for Organ Transplantation 10:1 2012 Feb pg 76-80
Anemia, Sickle Cell
Bone Marrow Transplantation
Genetic Predisposition to Disease
Pub Type(s)Case Reports