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克罗恩病患者在短期使用免疫调节剂后发生医源性小肠卡波西肉瘤:病例报告及文献复习。

Iatrogenic Kaposi sarcoma of the small bowel in Crohn's disease following short-term use of immunomodulators: a case report and review of the literature.

机构信息

Division of Hepatogastroenterology, Department of Internal Medicine, Chi Mei Medical Center, No.901, Zhonghua Rd. Yongkang Dist., Tainan City, 71004, Taiwan, ROC.

Department of Pathology, Chi Mei Medical Center, Tainan, Taiwan, ROC.

出版信息

J Med Case Rep. 2022 Nov 7;16(1):424. doi: 10.1186/s13256-022-03637-8.

Abstract

BACKGROUND

Kaposi sarcoma is a vascular tumor highly related to human herpesvirus-8 and Kaposi sarcoma-associated herpesvirus. Kaposi sarcoma usually manifests as skin or mucosal lesions; involvement in visceral organs such as the gastrointestinal tract is rare. Kaposi sarcoma can occur in immunocompromised patients receiving immunosuppressive therapy, in which case it is known as iatrogenic Kaposi sarcoma or drug-induced Kaposi sarcoma. Intestinal Kaposi sarcoma in patients with inflammatory bowel disease is extremely rare.

CASE PRESENTATION

A 46-year-old East Asian male with recently diagnosed Crohn's disease was administered azathioprine and prednisolone; however, the patient complained of persistent abdominal pain and diarrhea following treatment. Endoscopy revealed small bowel Kaposi sarcoma. The patient was treated with systemic chemotherapy successfully without relapse.

CONCLUSIONS

This is the fifth case of Kaposi sarcoma developed over the small intestine in a patient with Crohn's disease following administration of immunomodulators. Additionally, this case indicated that even short-term immunomodulator use can induce Kaposi sarcoma in patients with inflammatory bowel disease. Thus, in patients with inflammatory bowel disease, if symptoms are aggravated or do not abate after immunomodulators prescription, and before intending to upgrade immunomodulators, endoscopy should be considered. Finally, chemotherapy can also be considered if both medication withdrawal and surgical intervention are not feasible.

摘要

背景

卡波西肉瘤是一种与人类疱疹病毒 8 型和卡波西肉瘤相关疱疹病毒高度相关的血管肿瘤。卡波西肉瘤通常表现为皮肤或黏膜病变;涉及胃肠道等内脏器官的情况较为罕见。卡波西肉瘤可发生在接受免疫抑制治疗的免疫功能低下患者中,在这种情况下,它被称为医源性卡波西肉瘤或药物诱导的卡波西肉瘤。炎症性肠病患者的肠道卡波西肉瘤极为罕见。

病例介绍

一名 46 岁的东亚男性,最近被诊断患有克罗恩病,接受了硫唑嘌呤和泼尼松龙治疗;然而,治疗后患者出现持续性腹痛和腹泻。内镜检查显示小肠卡波西肉瘤。该患者接受全身化疗成功,无复发。

结论

这是第五例在接受免疫调节剂治疗后克罗恩病患者出现小肠卡波西肉瘤的病例。此外,该病例表明,即使短期使用免疫调节剂也可能在炎症性肠病患者中引发卡波西肉瘤。因此,对于炎症性肠病患者,如果在免疫调节剂处方后症状加重或未缓解,并且在打算升级免疫调节剂之前,应考虑进行内镜检查。最后,如果药物撤出和手术干预都不可行,也可以考虑化疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ea24/9639291/f74fa7cc4230/13256_2022_3637_Fig1_HTML.jpg

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