Graves' orbitopathy in a patient with adrenoleukodystrophy after bone marrow transplantation
| Autor: | W. M. Wiersinga, Antonella Lupetti, Y Vardizer, A C Lankester, S Vandelanotte, Lelio Baldeschi |
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| Přispěvatelé: | Endocrinology, Ophthalmology |
| Jazyk: | angličtina |
| Rok vydání: | 2009 |
| Předmět: |
Male
medicine.medical_specialty Endocrinology Diabetes and Metabolism Graves' disease Thyrotropin medicine.disease_cause Autoimmunity Endocrinology Internal medicine medicine Adrenal insufficiency Humans Adrenoleukodystrophy Child Bone Marrow Transplantation Autoimmune disease business.industry Leukodystrophy General Medicine medicine.disease Graves Disease Surgery Transplantation medicine.anatomical_structure surgical procedures operative Female Bone marrow business |
| Zdroj: | European journal of endocrinology / European Federation of Endocrine Societies, 161(2), 369-373. BioScientifica Ltd. |
| ISSN: | 0804-4643 |
| Popis: | ObjectiveFor 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.Case reportWe 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.Evidence synthesisA 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 I131 (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.ConclusionsThis 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. |
| Databáze: | OpenAIRE |
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