Sonwalkar S A, James R M, Ahmad T, Zhang L, Verbeke C S, Barnard D L, Jewell D P, Hull M A
Molecular Medicine Unit, University of Leeds, and Department of Gastroenterology, St James's University Hospital, Leeds, UK.
Gut. 2003 Oct;52(10):1518-21. doi: 10.1136/gut.52.10.1518.
We report a case of fulminant Crohn's colitis that occurred following non-myeloablative allogeneic stem cell transplantation for Hodgkin's lymphoma. Adoptive transfer of inflammatory bowel disease by haematopoietic cells is recognised in several animal models of inflammatory bowel disease and remission of Crohn's disease has been reported in patients who have received a bone marrow transplant. However, adoptive transfer of Crohn's disease susceptibility leading to phenotypic manifestation of the disease after transplantation has not been previously reported. Having ruled out an infective cause of a colitis in this case, we speculated that adoptive transfer of Crohn's disease may have occurred and performed a genetic analysis of known susceptibility loci for significant donor-recipient mismatches. The donor and recipient had several haplotype mismatches in HLA class III genes at the IBD3 locus. In addition, the donor (but not the recipient) had a polymorphism of the 5' UTR of NOD2/CARD15 that may be associated with Crohn's disease. This case highlights the question of whether adoptive transfer of Crohn's disease can occur between allogeneic stem cell transplant donor and recipient, in a similar fashion to that reported for other autoimmune diseases. This report should also stimulate debate regarding the need for stem cell transplant donor screening for inflammatory bowel disease.
我们报告了1例在接受非清髓性异基因干细胞移植治疗霍奇金淋巴瘤后发生暴发性克罗恩病性结肠炎的病例。在几种炎症性肠病动物模型中已认识到造血细胞可导致炎症性肠病的过继转移，并且有报道称接受骨髓移植的患者克罗恩病得以缓解。然而，此前尚未有过继转移克罗恩病易感性导致移植后疾病表型显现的报道。在排除了该病例中结肠炎的感染性病因后，我们推测可能发生了克罗恩病的过继转移，并对已知的易感性位点进行了基因分析，以查找供体 - 受体之间显著的错配情况。供体和受体在IBD3位点的HLA III类基因中有几个单倍型错配。此外，供体（而非受体）的NOD2/CARD15基因5'非翻译区存在一种可能与克罗恩病相关的多态性。该病例突出了一个问题，即克罗恩病是否会像其他自身免疫性疾病那样在异基因干细胞移植供体和受体之间发生过继转移。本报告还应引发关于是否需要对干细胞移植供体进行炎症性肠病筛查的讨论。
