Background

Tumor necrosis factor (TNF) alpha is critical in the development of granulomas and multiple recent reports have highlighted the role of infliximab, an infused TNF alpha inhibitor, in the treatment of neurosarcoidosis. As a self-injected TNF alpha inhibitor, adalimumab has certain advantages over infused medications, including greater patient freedom and autonomy. Experience with adalimumab is not well reported in the literature.

Objective

To report clinical experience with adalimumab in the treatment of central nervous system (CNS) sarcoidosis by combining observations in our center with those that have been reported in the literature.

Methods

Patients were identified from the Mass General Brigham Research Patient Data Registry and in the literature by searching PubMed. Patients with CNS manifestations of sarcoidosis treated with adalimumab were included for retrospective review and analyzed for baseline characteristics, treatment indications, outcomes, and adverse effects.

Results

Adalimumab was commonly started after failure of or intolerance to infliximab and methotrexate. Of those with adequate follow-up, 5/10 ultimately improved, remission was maintained in 3/10, and 2/10 with active disease remained stable without further worsening. One patient suffered a relapse, likely multifactorial in etiology, but has remained relapse free on adalimumab for 10 months subsequently. Three patients ultimately discontinued adalimumab.

Conclusions

Preliminary evidence suggests that adalimumab may be a reasonable therapeutic option for patients with neurosarcoidosis affecting the CNS, including those with medically refractory disease.

中文翻译：

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阿达木单抗治疗中枢神经系统结节病：单中心经验和文献回顾

背景

肿瘤坏死因子 (TNF) α 在肉芽肿的发展中至关重要，最近的多份报告强调了英夫利昔单抗（一种注入的 TNF α 抑制剂）在治疗神经结节病中的作用。作为一种自我注射的 TNF α 抑制剂，阿达木单抗与输注药物相比具有一定的优势，包括更大的患者自由度和自主权。文献中没有很好地报道使用阿达木单抗的经验。

客观的

通过将我们中心的观察结果与文献报道的结果相结合，报告阿达木单抗治疗中枢神经系统（CNS）结节病的临床经验。

方法

通过搜索 PubMed 从 Mass General Brigham Research Patient Data Registry 和文献中确定患者。对接受阿达木单抗治疗的有中枢神经系统结节病表现的患者进行回顾性分析，并分析其基线特征、治疗适应症、结局和不良反应。

结果

阿达木单抗通常在英夫利昔单抗和甲氨蝶呤治疗失败或不耐受后开始使用。在进行充分随访的患者中，5/10 最终改善，3/10 维持缓解，2/10 有活动性疾病的患者保持稳定，没有进一步恶化。一名患者复发，病因可能是多因素的，但随后在阿达木单抗治疗 10 个月内保持无复发。三名患者最终停用阿达木单抗。

结论

初步证据表明，对于累及 CNS 的神经结节病患者，包括药物难治性疾病患者，阿达木单抗可能是一种合理的治疗选择。