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Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report.
BMC Nephrology ( IF 2.3 ) Pub Date : 2020-06-10 , DOI: 10.1186/s12882-020-01884-9
Emily K Glover 1, 2 , R Alexander Speight 1, 3 , Despina Televantou 4 , Stephanie Needham 4 , Neil S Sheerin 1, 2
Affiliation  

Post-transplant lymphoproliferative disease is a recognized complication following solid organ transplantation. This is usually a B cell disease and frequently associated with Epstein Barr virus infection, although T cell PTLD can occur. T cell PTLD is usually a monomorphic, lymphomatous disease associated with an adverse prognosis. We report a 52 year old male pre-emptive renal transplant recipient who developed severe diarrhea with weight loss following intensification of his immunosuppression due to antibody mediated rejection 3 years after transplantation. Duodenal biopsy demonstrated monoclonal CD8+ T cell duodenitis leading to increased intraepithlieal lymphocytes and sub-total villous atrophy mimicking coeliac disease. Coeliac disease was excluded by negative anti-tissue transglutaminase antibody, HLA-DQ2 and HLA-DQ8 testing. There was no evidence of lymphoma either on biopsy or CT enterography and no FDG avid disease on PET. Symptoms did not improve with reduction of immunosuppression, but resolved fully on complete withdrawal of treatment. The transplant failed and he was established on dialysis. The diagnosis was early PTLD. Oesophagogastroduodenoscopy with small bowel biopsies is a useful investigation for determining the cause of diarrhoea in renal transplant patients when more common causes have been excluded. This is the first report that we are aware of clonal T cell PTLD mimicking coeliac disease which only resolved after complete withdrawal of immunosuppression. As treatments for lymphoma are aggressive they are only initiated in the malignant phase and management of early stage PTLD is to minimise risk of progression by reducing immunosuppression. Any plans to retransplant will have to take into consideration the possibility that PTLD will recur.

中文翻译:

免疫抑制诱导的克隆性 T 细胞淋巴组织增生性疾病导致严重腹泻,模仿肾移植后的乳糜泻:病例报告。

移植后淋巴组织增生性疾病是实体器官移植后公认的并发症。这通常是一种 B 细胞疾病,经常与 Epstein Barr 病毒感染有关,尽管 T 细胞 PTLD 也可能发生。T 细胞 PTLD 通常是一种与不良预后相关的单形性淋巴瘤疾病。我们报告了一名 52 岁男性先发制人肾移植受者,他在移植后 3 年因抗体介导的排斥反应加剧免疫抑制后出现严重腹泻并伴有体重减轻。十二指肠活检显示单克隆 CD8+ T 细胞十二指肠炎导致上皮内淋巴细胞增加和次全绒毛萎缩,类似于乳糜泻。通过阴性抗组织转谷氨酰胺酶抗体、HLA-DQ2 和 HLA-DQ8 测试排除了乳糜泻。活检或 CT 小肠造影均未发现淋巴瘤的证据,PET 上也未发现 FDG 狂热病。随着免疫抑制的减少,症状没有改善,但在完全停止治疗后完全消失。移植失败,他接受了透析治疗。诊断为早期 PTLD。在排除了更常见的原因后,食管胃十二指肠镜检查和小肠活检对于确定肾移植患者腹泻的原因是一项有用的研究。这是我们知道克隆性 T 细胞 PTLD 模仿乳糜泻的第一份报告,该疾病仅在完全撤消免疫抑制后才能解决。由于淋巴瘤的治疗是积极的,它们仅在恶性阶段开始,早期 PTLD 的管理是通过减少免疫抑制来最小化进展的风险。
更新日期:2020-06-10
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