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复发性后部巩膜炎和眼眶肌炎作为克罗恩病的肠外表现:病例报告和系统文献复习。

Recurrent posterior scleritis and orbital myositis as extra-intestinal manifestations of Crohn's disease: Case report and systematic literature review.

机构信息

Department of Gastroenterology, John Radcliffe Hospital, Headley Way, Headington, Oxford, OX3 9DU, UK.

出版信息

J Crohns Colitis. 2008 Dec;2(4):337-42. doi: 10.1016/j.crohns.2008.06.002. Epub 2008 Oct 29.

DOI:10.1016/j.crohns.2008.06.002
PMID:21172235
Abstract

BACKGROUND

Ocular episcleritis and uveitis are well-recognised extra-intestinal manifestations of Crohn's disease. Orbital myositis is rare: to our knowledge it has been associated with Crohn's disease in thirteen cases. Posterior scleritis, orbital myositis and Crohn's disease have been reported as coexisting in only two cases.

METHODS AND RESULTS

We describe a third case, that of a 31-year old female with Crohn's colitis for 8 years, complicated by enteropathic arthritis and pyoderma gangrenosum. She presented with intense and intractable periorbital pain, particularly at night and worse on eye movements. B-scan ultrasonography confirmed posterior scleritis and treatment with high dose oral steroids (up to 60 mg prednisolone) was initially effective, but subsequently failed to control the inflammation. There was only a partial response to infliximab. Five months after presentation, diplopia developed, with failure of abduction of the left eye. MRI scan of the orbits confirmed orbital myositis involving the left lateral and medial rectus muscles. Pulsed intravenous methylprednisolone and six cycles of intravenous cyclophosphamide over a three month period resulted in complete resolution of inflammatory symptoms.

CONCLUSIONS

This case highlights a rare combination of ocular abnormality secondary to Crohn's disease and reports successful resolution with aggressive immunosuppressive therapy.

摘要

背景

眼弓状层炎和葡萄膜炎是克罗恩病的典型肠外表现。眶肌炎则较为罕见:据我们所知,其仅与 13 例克罗恩病相关。后巩膜炎、眶肌炎和克罗恩病同时存在的情况仅有两例报道。

方法和结果

我们描述了第三例患者,为一名 31 岁女性,患有克罗恩病结肠炎 8 年,并发肠炎性关节炎和坏疽性脓皮病。她出现剧烈且难以控制的眼眶疼痛,尤其是夜间和眼球运动时加重。B 型超声扫描证实为后巩膜炎,最初采用大剂量口服类固醇(高达 60mg 泼尼松龙)治疗有效,但随后炎症无法得到控制。英夫利昔单抗也仅有部分缓解。发病 5 个月后,出现复视,左眼外展功能丧失。眼眶 MRI 扫描证实左侧外直肌和内直肌眶肌炎。经过 3 个月的时间,给予脉冲式静脉内甲基泼尼松龙和 6 个周期的静脉内环磷酰胺治疗后,炎症症状完全缓解。

结论

本例突出了由克罗恩病引起的眼部异常的罕见组合,并报告了采用强化免疫抑制疗法成功缓解。

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