title: Graves' orbitopathy in a patient with adrenoleukodystrophy after bone marrow transplantation.
abstract: OBJECTIVE
  For many years, the treatment of X-linked childhood cerebral adrenoleukodystrophy (XALD) consisted of hydrocortisone replacement and a mixture of short chain-fatty acids, known as 'Lorenzo's oil'. Recently, bone marrow transplantation (BMT) has also been used.
  
  
  METHODS
  We report the case of a patient affected by XALD who developed Graves' hyperthyroidism (GH) and Graves' orbitopathy (GO) after BMT and who we could follow-up for 6.5 years afterwards.
  
  
  RESULTS
  A boy affected by XALD was treated at the age of 6 years, with a whole BMT from his sister. One year after BMT, the transplanted patient presented TSH at the lower normal value and 3 years later he developed thyrotoxicosis. After a further 2 years, the patient developed GO, which showed clinical evidence of reactivation 5 years after its onset as a consequence of an attempt to treat thyrotoxicosis by means of I(131) (300 MBq). Seven years after BMT, the donor showed alterations of thyroid autoimmunity and 1 year thereafter she developed GH. She never presented GO during a subsequent 5 year follow-up.
  
  
  CONCLUSIONS
  This case illustrates that autoimmunity originating from a pre-symptomatic donor can be transferred into the host during allogeneic stem cell transplantation. In cases where autoimmune phenomena are recognized in the donor prior to donation, alternative donors or T-cell manipulation of the graft might be considered.
authors.affiliation: Department of Ophthalmology, Orbital Center, University of Amsterdam, Room D2-436, Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands.
authors.forename: Y
authors.initials: Y
authors.lastname: Vardizer
authors.forename: A
authors.initials: A
authors.lastname: Lupetti
authors.forename: S
authors.initials: S
authors.lastname: Vandelanotte
authors.forename: A C
authors.initials: AC
authors.lastname: Lankester
authors.forename: W M
authors.initials: WM
authors.lastname: Wiersinga
authors.forename: L
authors.initials: L
authors.lastname: Baldeschi
mesh_terms: D000326:Adrenoleukodystrophy / Q000276:immunology* / Q000601:surgery; D016026:Bone Marrow Transplantation / Q000009:adverse effects / Q000276:immunology*; D002648:Child; D005260:Female; D006111:Graves Disease / Q000209:etiology / Q000276:immunology*; D006801:Humans; D008297:Male; D013972:Thyrotropin / Q000097:blood
chemical_list: D013972:Thyrotropin