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青少年皮肌炎中肠道穿孔的罕见病因。

Unusual cause for intestinal perforation in juvenile dermatomyositis.

作者信息

Bhaskaran Harikrishnan, Balan Suma

机构信息

Department of Clinical Immunology and Rheumatology, Amrita Institute of Medical Sciences and Research Centre, Ernakulam, Kerala, India.

出版信息

BMJ Case Rep. 2019 Aug 22;12(8):e229395. doi: 10.1136/bcr-2019-229395.

DOI:10.1136/bcr-2019-229395
PMID:31444261
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6721139/
Abstract

We report a case of juvenile dermatomyositis (JDM) with cytomegalovirus (CMV) colitis which was further complicated with perforation. The patient, a 6-year-old girl, was diagnosed with JDM 1 month prior to the current presentation. After 2 weeks of optimising her treatment with steroid, intravenous Ig and methotrexate, she was readmitted with diffuse abdominal pain. Erect abdominal X-ray revealed gas under diaphragm. An exploratory laparotomy showed perforation of the large intestine. A biopsy showed inclusion bodies of CMV with immunohistochemistry for CMV positive. Strong positive CMV DNA PCR from tissue specimen, positive IgG CMV and negative IgM CMV in blood suggested a reactivation of CMV. The treatment followed included surgery and strategic use of antiviral agents as well as immunomodulators. CMV enteritis with complications should also be suspected in optimally treated autoimmune disease patients, including JDM, when they present with abdominal symptoms.

摘要

我们报告一例青少年皮肌炎(JDM)合并巨细胞病毒(CMV)结肠炎且进一步并发穿孔的病例。该患者为一名6岁女孩,在本次就诊前1个月被诊断为JDM。在用类固醇、静脉注射免疫球蛋白和甲氨蝶呤优化治疗2周后,她因弥漫性腹痛再次入院。立位腹部X线片显示膈下游离气体。剖腹探查显示大肠穿孔。活检显示CMV包涵体,CMV免疫组化呈阳性。组织标本的CMV DNA PCR呈强阳性,血液中CMV IgG阳性而CMV IgM阴性,提示CMV再激活。后续治疗包括手术、合理使用抗病毒药物以及免疫调节剂。在接受最佳治疗的自身免疫性疾病患者,包括JDM患者出现腹部症状时,也应怀疑CMV肠炎并伴有并发症。

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