Lancet:急性严重溃疡性结肠炎导致双下肢无力—案例报道

2016-07-02 MedSci MedSci原创

一个49岁的肥胖男性,既往无疾病史,2014年8月后出现5周的血性腹泻,厌食和体重减轻。他报告说道在过去20天,每天大便都带血,伴夜间睡眠障碍,入院时有发热,心动过速,贫血和强烈的炎症反应。乙状结肠镜检查显示严重的融合性的结肠炎,组织病理学证实为首次出现的活动性溃疡性结肠炎(如图)。综合这些发现,我们开始静脉使用甲强龙30mg每天,持续7天,同时口服递减剂量的泼尼松龙(40mg每天,每周减少5mg

一个49岁的肥胖男性,既往无疾病史,2014年8月后出现5周的血性腹泻,厌食和体重减轻。他报告说道在过去20天,每天大便都带血,伴夜间睡眠障碍,入院时有发热,心动过速,贫血和强烈的炎症反应。乙状结肠镜检查显示严重的融合性的结肠炎,组织病理学证实为首次出现的活动性溃疡性结肠炎(如图)。综合这些发现,我们开始静脉使用甲强龙30mg每天,持续7天,同时口服递减剂量的泼尼松龙(40mg每天,每周减少5mg)。由于他对类固醇只有部分反应,我们在第五天开始使用环孢素,在第11天时大便频率,血清标志物和各项观察指标均恢复正常。



急性严重溃疡性结肠炎患者出现脊髓脂肪瘤病

(A)内窥镜显示乙状结肠处严重的溃疡性结肠炎
(B)MRI T1加权成像,显示颈椎和胸椎脊髓硬膜外脂肪增多伴T1-T9压缩(箭头所指)

患者1个月之后出现渐进性步态障碍,摔跤,腰痛,躯干有束带感。患者否认任何尿储留或大小便失禁或勃起功能障碍。无发热或脊柱创伤史,先前无神经系统症状。至此,他服用环孢素200mg每天两次,泼尼松龙15mg/天,总共用了45天的类固醇。结肠炎保持较好的临床和生化缓解状态。

经过检查,他有典型的满月脸和库欣综合征的症状,需要两个拐杖辅助行走。两腿肌张力增加,伴有对称性近端肌力减弱(MRC分级4+/5)。双下肢深部腱反射病理性活跃(膝反射3 +,踝反射4 +),无持续性踝阵挛。下肢远侧关节位置觉受损,巴氏征可疑阳性。T10以下轻微触觉降低,针刺觉降低,第10肋以下震动觉降低。脊柱检查无特殊。

血液检查结果在正常范围内,降低了内在脊髓病变原因的可能性,如感染,维他命B12或铜缺乏,无糖耐量受损的证据。脊柱MRI显示T1-T11硬膜外肿块,远端骶骨部位硬膜外沉积(如图)。肿块在短T1反转恢复序列(STIR)信号完全抑制,在TI和T2上显示均匀高信号。这些表现与脂肪一致,弥散分布显示只可能为脂肪增多症。

因为有大范围的脊髓实质受到压迫,不太可能进行肿块切除术 ,这可能需要接受T1-L2的减压性椎扳切除术,以及椎弓根螺钉固定和脂肪切除术,这伴随着极高的出血风险和并发症,可能会恶化腰椎背部疼痛症状。因此,我们选择了保守的资料方法:7天内快速减少类固醇,使用普瑞巴林治疗神经性疼痛,理疗师和营养师协助减轻体重。

5个月后MRI检查显示脊髓脂肪增多症状部分缓解,脊髓受压从T6-T9,无髓核信号异常。他的症状逐渐得到好转,到2015年10月,只残留脚趾感觉减退,尽管他仍使用拐杖走路,但全身肌力是正常的。

脊髓硬膜外脂肪增多症是脂肪组织在硬膜外的病理性过度增长,导致髓核受压。这是很不常见的,在炎症性肠病的报道中仅出现过两次。脊髓硬膜外脂肪增多症可能是长期使用皮质醇治疗的并发症(55.3%的病例中),但极罕见出现在急性或短期使用的患者中。24.5%的病例仅仅是因为肥胖导致,以及肥胖患者倾向于发生更加快速的皮质类固醇诱导的脊髓硬膜外脂肪增多。17%的病例无明确病因,我们也没有了解到之前有环孢素相关的脊髓硬膜外脂肪增多症的报道。

25%的与类固醇使用相关的脊髓硬膜外脂肪增多症都是结合去除外源性类固醇和减肥的保守治疗。手术椎扳切除术和肿块切除术作为一种可选择的治疗方式正在更加频繁的使用( 75%的案例),与保守治疗的反应率相似(77%)。没有报道像这个病例一样出现如此大范围的硬膜外脂肪增多,MRI上显示的病变范围和发病的速度使其成为值得注意的病例。

在很多情况下类固醇依旧是主要的治疗方法,在急性严重的溃疡性结肠炎中,是可以救命的。然而 ,考虑到逐渐增加的肥胖人群,脊髓硬膜外脂肪增多症是一种需要谨记的重要的——尽管罕见的并发症。

原始出处:

Ruairi W Lynch, MBa, †, Timothy Soane, PhDb, †, Rod Gibson,et al,Bilateral lower limb weakness in acute severe ulcerative colitis,lancet,doi:10.1016/S0140-6736(16)30857-1




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    2016-12-20 nymo
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    2016-08-03 howi
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    2016-07-05 oo902

    长见识了

    0

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    2016-07-04 lovetcm

    你非常罕见的并发症,背后可能有共同的,基因的作用原理

    0

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    2016-07-04 深海的鱼

    少见,谢谢分享

    0

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