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克罗恩病吻合口裂开并发罕见T细胞淋巴瘤的诊治:一例报告

Management of an uncommon T-Cell lymphoma revealed by an anastomotic dehiscence in Crohn's disease: A case report.

作者信息

Haddad Anis, Ben Mahmoud Ahmed, Maghrebi Houcine, Chelly Baya, Jouini Mohamed, Kacem Montasser Jameleddine

机构信息

Department of Surgery A, Rabta Hospital, Tunis, Tunisia; Faculty of Medicine of Tunis, Tunis El Manar University, Tunis, Tunisia.

Department of Surgery A, Rabta Hospital, Tunis, Tunisia; Faculty of Medicine of Tunis, Tunis El Manar University, Tunis, Tunisia.

出版信息

Int J Surg Case Rep. 2021 Feb;79:53-57. doi: 10.1016/j.ijscr.2021.01.011. Epub 2021 Jan 6.

DOI:10.1016/j.ijscr.2021.01.011
PMID:33429357
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7809163/
Abstract

INTRODUCTION AND IMPORTANCE

T-cell lymphoma degeneration in pancolic crohn's disease is scarce. It is mostly related to long-standing inflammatory bowel disease in patients under immunosuppressants. We reviewed the clinical, endoscopic, radiological and histologic data of the patient as well as the literature dealing with T-cell lymphoma arising from pancolic crohn's disease.

CASE PRESENTATION

We describe in this paper an unusual case of a female young patient who underwent emergency surgery for per endoscopic perforation of the right colon while being under azathioprine. She had a subtotal colectomy with ileostomy and sigmoidostomy. After six months, we restored the digestive continuity through an ileorectal anastomosis. She was kept in remission on azathioprine. After one year, she presented with a pelvic abscess revealing a dehiscence of the ileorectal anastomosis leading to a surgical drainage and resection of the anastomosis associated with terminal ileostomy and closure of the rectal stump. Pathology examination revealed T cell lymphoma arising from the ileorectal anastomosis.

DISCUSSION

Patients with long-standing IBD have an increased risk of developing colorectal cancer. The onset of a malignant lymphoma during the course of the CD is scarce. Some studies haves failed to identify crohn's disease as a risk factor of lymphoma whereas other ones have succeeded to. Immunosuppressants are reported to have carcinogenic effect. Rarely, lymphoma degeneration can be revealed by intestinal complications such as perforation like in our case.

CONCLUSION

Many studies reported lymphoma degeneration of crohn's disease after long-term immunosuppressant therapy. However, rapid T-cell lymphoma degeneration revealed by anastomotic dehiscence in crohn's disease made our case unique and interesting.

摘要

引言与重要性

全结肠型克罗恩病中T细胞淋巴瘤退变情况罕见。它大多与长期炎症性肠病患者使用免疫抑制剂有关。我们回顾了该患者的临床、内镜、放射学和组织学数据以及有关全结肠型克罗恩病引发T细胞淋巴瘤的文献。

病例介绍

本文描述了一名年轻女性患者的罕见病例,该患者在使用硫唑嘌呤期间因内镜检查导致右结肠穿孔而接受急诊手术。她接受了次全结肠切除术并进行了回肠造口术和乙状结肠造口术。六个月后,我们通过回肠直肠吻合术恢复了消化连续性。她继续使用硫唑嘌呤并保持缓解状态。一年后,她出现盆腔脓肿,提示回肠直肠吻合口裂开,导致进行手术引流并切除吻合口,同时进行末端回肠造口术和直肠残端闭合术。病理检查显示回肠直肠吻合口出现T细胞淋巴瘤。

讨论

长期患有炎症性肠病的患者患结直肠癌的风险增加。在克罗恩病病程中发生恶性淋巴瘤的情况罕见。一些研究未能将克罗恩病确定为淋巴瘤风险因素,而其他研究则成功做到了这一点。据报道免疫抑制剂具有致癌作用。很少有淋巴瘤退变会像我们的病例那样通过肠道并发症如穿孔表现出来。

结论

许多研究报道了长期免疫抑制治疗后克罗恩病出现淋巴瘤退变。然而,克罗恩病中因吻合口裂开导致的快速T细胞淋巴瘤退变使我们的病例独特且有趣。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/5479850623ff/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/20eb36cfa34d/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/9ec5d0d13ae7/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/5479850623ff/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/20eb36cfa34d/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/9ec5d0d13ae7/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3db4/7809163/5479850623ff/gr3.jpg

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