Unbound MEDLINE

Autoimmune polyglandular syndrome type II after bone marrow transplant: real transfer or acceleration of a programmed disease?

Abstract

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.

Links

  • Publisher Full Text
  • Authors

    Mellouli F, Ksouri H, Lakhal A, Torjmen L, Ladeb S, Ben Othman T, Hmida S, Bejaoui M

    Institution

    Service d'Immuno-Hematologie Pediatrique, Centre National de Greffe de Moelle Osseuse, Centre National de Transfusion Sanguine, Tunis, Tunisia.

    Source

    Experimental and clinical transplantation : official journal of the Middle East Society for Organ Transplantation 10:1 2012 Feb pg 76-80

    MeSH

    Addison Disease
    Adrenal Insufficiency
    Anemia, Sickle Cell
    Bone Marrow Transplantation
    Child
    Genetic Predisposition to Disease
    Humans
    Male
    Polyendocrinopathies, Autoimmune
    Tissue Donors
    Vitiligo

    Pub Type(s)

    Case Reports
    Journal Article

    Language

    eng

    PubMed ID

    22309426