Puthenparampil Marco, Gaggiola Marta, Miscioscia Alessandro, Mauceri Valentina Annamaria, De Napoli Federica, Zanotelli Giovanni, Anglani Mariagiulia, Nosadini Margherita, Sartori Stefano, Perini Paola, Rinaldi Francesca, Gallo Paolo
Multiple Sclerosis Centre, Clinica Neurologica, Dipartimento di Neuroscienze, Università degli Studi di Padova, Via Giustiniani 5, 35128 Padova, Italy.
Multiple Sclerosis Centre, Azienda Ospedaliera di Padova, Padova, Italy.
Ther Adv Neurol Disord. 2023 Oct 6;16:17562864231177196. doi: 10.1177/17562864231177196. eCollection 2023.
Paediatric-onset multiple sclerosis (POMS) therapeutic approach derives from of adult-onset multiple sclerosis (AOMS) tailored algorithms.
To evaluate in a common clinical scenario the efficacy and safety of alemtuzumab (ALZ) in POMS and AOMS.
All patients switching from natalizumab (NTZ) to ALZ for safety concerns (high anti-John Cunningham Virus Antibody Index value, anti-JCV Index) were enrolled in this single-centre, retrospective, case-control open-label study.
Ten POMS and 27 AOMS were followed up for 51.3 months. After month 12, we found a lower risk of clinical or radiological relapses among AOMS patients and among patients with older age at ALZ (both < 0.05). Survival analysis revealed an increased risk of relapse in POMS compared with AOMS (logrank = 0.00498) and patients starting ALZ before age 22.75 years than the elder ones (logrank = 0.0018). Survival analysis did not disclose any difference between AOMS and POMS (logrank = 0.27) in terms of progression independent of any relapse activity (PIRA). In addition, no evidence of relapse-associated worsening was observed. Autoimmune events were reported by 5 AOMS and no POMS (29.4% 0.0%, = 0.057), and survival analysis was not significant (logrank = 0.0786).
ALZ seems more effective in AOMS than in POMS following NTZ. These findings underrate ALZ effectiveness when shifting from NTZ in POMS.
儿童期起病的多发性硬化症(POMS)的治疗方法源自针对成人期起病的多发性硬化症(AOMS)定制的算法。
在常见临床场景中评估阿仑单抗(ALZ)治疗POMS和AOMS的疗效与安全性。
所有因安全问题(高抗约翰·坎宁安病毒抗体指数值、抗JCV指数)从那他珠单抗(NTZ)换用ALZ的患者均纳入这项单中心、回顾性、病例对照开放标签研究。
10例POMS患者和27例AOMS患者接受了51.3个月的随访。在第12个月后,我们发现AOMS患者以及使用ALZ时年龄较大的患者发生临床或影像学复发的风险较低(均P<0.05)。生存分析显示,与AOMS相比,POMS复发风险增加(对数秩检验=0.00498),且开始使用ALZ时年龄小于22.75岁的患者比年龄较大者复发风险增加(对数秩检验=0.0018)。生存分析未发现AOMS和POMS在与任何复发活动无关的疾病进展(PIRA)方面存在差异(对数秩检验=0.27)。此外,未观察到与复发相关的病情恶化迹象。5例AOMS患者报告了自身免疫事件,POMS患者未报告(29.4%对0.0%,P=0.057),生存分析无显著性差异(对数秩检验=0.0786)。
在NTZ治疗后,ALZ在AOMS中似乎比在POMS中更有效。这些发现低估了POMS患者从NTZ换用ALZ时ALZ的有效性。