Marques Inês Brás, Giovannoni Gavin, Marta Monica
Queen Mary University London, Blizard Institute, 4 Newark Street, London, E1 1AT, UK.
BMC Neurol. 2014 Dec 11;14:237. doi: 10.1186/s12883-014-0237-5.
Several disorders may present with mononeuritis multiplex and the etiological diagnosis can be challenging.
We report a 42 year-old female who presented with severe lower limb neuropathic pain, asymmetric weakness and sensory impairment and was diagnosed with mononeuritis multiplex. Biopsy showed a granulomatous vasculitic process with eosinophils, scarce granulomata and axonal neuropathy and granulomatosis with poliangiitis was assumed. Steroids, cyclophosphamide, alemtuzumab, azathioprine, mycophenolate mofetil and rituximab were used, all with transient and insufficient response. Skin biopsy performed in a further exacerbation allowed sarcoidosis diagnosis. Infliximab and, later, adalimumab induced good clinical and laboratorial response, but neutralizing antibodies developed to both drugs, so etanercept was tried with good clinical response.
To the best of our knowledge, this is the first report of sarcoidosis successfully treated with etanercept. This drug may be considered in refractory sarcoidosis after other TNF-α inhibitors failure, having the advantage of not being associated with neutralizing antibodies development.
多种疾病可能表现为多发性单神经炎,病因诊断具有挑战性。
我们报告一名42岁女性,她出现严重的下肢神经性疼痛、不对称性肌无力和感觉障碍,被诊断为多发性单神经炎。活检显示为伴有嗜酸性粒细胞的肉芽肿性血管炎过程,肉芽肿稀少且存在轴索性神经病,考虑为显微镜下多血管炎。使用了类固醇、环磷酰胺、阿仑单抗、硫唑嘌呤、霉酚酸酯和利妥昔单抗,所有这些药物的反应均短暂且不充分。在病情进一步加重时进行的皮肤活检确诊为结节病。英夫利昔单抗以及后来的阿达木单抗诱导了良好的临床和实验室反应,但对这两种药物均产生了中和抗体,因此尝试使用依那西普并获得了良好的临床反应。
据我们所知,这是首例用依那西普成功治疗结节病的报告。在其他肿瘤坏死因子-α抑制剂治疗失败后,对于难治性结节病可考虑使用该药,其优点是不会产生中和抗体。