Department of Basic Medical Sciences, Neurosciences and Sense Organs, University of Bari "Aldo Moro", Bari, Italy.
Brain Behav. 2021 Jan;11(1):e01930. doi: 10.1002/brb3.1930. Epub 2020 Dec 16.
Interferon beta (IFNβ) is a well-established first-line therapy for relapsing-remitting multiple sclerosis (RRMS) patients and remains the most widely prescribed agent. Atypical hemolytic uremic syndrome (aHUS) represents a rare but severe adverse effect (AE) that could occur even after many years from the beginning of IFNβ therapy. Eculizumab is currently approved for treatment of aHUS and recently for neuromyelitis optica spectrum disorder (NMOSD) with aquaporin-4 antibodies (AQP4-IgG). In this article, we report the case of the latest onset of IFNβ-related aHUS experienced by an MS patient and we briefly review the literature on this topic.
We performed a systematic review of the literature using PubMed, and we performed a retrospective analysis of RRMS patients that received IFNβ-1a in our center and developed thrombotic microangiopathy (TMA). From this search, we identified only one patient.
In the published literature, we identified 24 MS patients who received IFNβ as disease-modifying treatment (DMT) and then developed thrombotic microangiopathy with kidney injury. The aHUS has been diagnosed in 6, all received IFNβ-1a and the latest onset was after 15 years. We report a case of a 39-year-old man affected by RRMS who assumed IFNβ-1a since 1999. In July 2018, he developed an IFNβ-related aHUS. After the failure of plasma exchange, he underwent eculizumab, with an improvement of glomerular filtration rate and without new signs of MS activity.
To our knowledge, this case represents the latest onset of IFNβ-related aHUS in MS patients. Up to now, there are not literary reports about the possibility to reintroduce a DMT as add-on therapy to eculizumab.
干扰素β（IFNβ）是治疗复发缓解型多发性硬化症（RRMS）患者的一种成熟的一线治疗药物，也是应用最广泛的药物。非典型溶血尿毒综合征（aHUS）是一种罕见但严重的不良反应（AE），即使在开始 IFNβ 治疗多年后也可能发生。依库珠单抗目前被批准用于治疗 aHUS，最近还被批准用于水通道蛋白 4 抗体（AQP4-IgG）阳性的视神经脊髓炎谱系疾病（NMOSD）。本文报告了一例 MS 患者出现最新发病的 IFNβ 相关性 aHUS，并简要回顾了这一主题的文献。
我们使用 PubMed 进行了文献系统综述，并对在我们中心接受 IFNβ-1a 治疗且发生血栓性微血管病（TMA）的 RRMS 患者进行了回顾性分析。从这项搜索中，我们只确定了一名患者。
在已发表的文献中，我们确定了 24 例接受 IFNβ 作为疾病修正治疗（DMT）后发生肾损伤性血栓性微血管病的 MS 患者。其中 6 例被诊断为 aHUS，均接受 IFNβ-1a 治疗，发病最晚时间为 15 年后。我们报告了一例 39 岁男性 RRMS 患者，自 1999 年开始使用 IFNβ-1a。2018 年 7 月，他发生了 IFNβ 相关性 aHUS。血浆置换失败后，他接受了依库珠单抗治疗，肾小球滤过率有所改善，且无 MS 活动的新迹象。
据我们所知，本例是 MS 患者中 IFNβ 相关性 aHUS 发病最晚的病例。迄今为止，尚无关于将 DMT 作为依库珠单抗的附加治疗重新引入的可能性的文献报道。
