Literature DB >> 22309426

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

Fethi Mellouli1, Habib Ksouri, Amel Lakhal, Lamia Torjmen, Saloua Ladeb, Tarek Ben Othman, Slama Hmida, Mohamed Bejaoui.   

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.

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Year:  2012        PMID: 22309426     DOI: 10.6002/ect.2011.0062

Source DB:  PubMed          Journal:  Exp Clin Transplant        ISSN: 1304-0855            Impact factor:   0.945


  2 in total

1.  Severe chronic psychosis after allogeneic SCT from a schizophrenic sibling.

Authors:  I E Sommer; D W van Bekkum; H Klein; R Yolken; Lot de Witte; G Talamo
Journal:  Bone Marrow Transplant       Date:  2014-10-06       Impact factor: 5.483

2.  Adrenal crisis in a 14-year-old boy 12 years after hematopoietic stem cell transplantation.

Authors:  Theresa Penger; Andrea Albrecht; Michaela Marx; Daniel Stachel; Markus Metzler; Helmuth G Dörr
Journal:  Endocrinol Diabetes Metab Case Rep       Date:  2018-06-06
  2 in total

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