免疫检查点抑制剂相关的小脑毒性:临床特征及与副肿瘤性小脑共济失调的比较。

IF 2.7 3区 医学 Q3 NEUROSCIENCES Cerebellum Pub Date : 2024-12-01 Epub Date: 2024-08-17 DOI:10.1007/s12311-024-01727-5
Marta Dentoni, Irene Florean, Antonio Farina, Bastien Joubert, Le-Duy Do, Jérôme Honnorat, Valentina Damato, Martina Fabris, Gian Luigi Gigli, Mariarosaria Valente, Alberto Vogrig
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引用次数: 0

摘要

免疫检查点抑制剂(ICIs)给癌症治疗带来了革命性的变化,其与免疫相关不良事件(irAEs)的关系已得到证实。然而,小脑irAEs的定义尚不明确,它们与副肿瘤性疾病的关系也不清楚。我们的目的是:(i) 描述小脑irAE的特征;(ii) 将其与副肿瘤性小脑共济失调(PCA)进行比较。我们进行了一项多中心、回顾性、队列研究,研究对象是服用 ICI 后出现新发、免疫介导、孤立/主要小脑功能障碍的患者。此外,还按照PRISMA指南进行了系统性回顾。小脑irAE病例与PCA患者的连续队列进行了比较。共纳入 35 例患者,其中 12 例为原始病例(男性:25/35(71%),中位年龄:65 [范围:20-82])。最常见的肿瘤是非小细胞肺癌(12/35,34%)。19/35(54%)的患者采用了抗-PD1疗法。症状出现于 ICI 发病后中位 11 周。15/31(48%)名受检患者检测到神经元抗体。25/30(83%)名患者的脑脊液呈炎性。磁共振成像显示,8/35(23%)的患者出现小脑过度强化。33/35(94%)例患者接受了免疫治疗,大多数患者的病情有所好转,但仍有残障(16/35,46%)。与一系列PCA(n = 15)相比,小脑irAE组与男性、肺癌(而非妇科/乳腺癌)、孤立性共济失调和较好的预后明显相关。我们提供了小脑虹膜异位症的详细特征。与 PCA 相比,两者在肿瘤关联、临床特征和预后方面存在差异。ICI 组的临床表现-抗体-肿瘤三联征仅部分反映了副肿瘤性疾病的相关性。
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Immune Checkpoint Inhibitor-Related Cerebellar Toxicity: Clinical Features and Comparison with Paraneoplastic Cerebellar Ataxia.

Immune checkpoint inhibitors (ICIs) have revolutionized cancer therapy, and the association with immune-related adverse events (irAEs) is well-established. However, cerebellar irAEs are poorly defined and their relationship with paraneoplastic disorders remains unclear. Our aim was (i) to characterize cerebellar irAE; (ii) to compare it with paraneoplastic cerebellar ataxia (PCA). We performed a multicenter, retrospective, cohort study of patients developing new-onset, immune-mediated, isolated/predominant cerebellar dysfunction after ICI administration. In addition, a systematic review following PRISMA guidelines was performed. Cerebellar irAE cases were compared with a consecutive cohort of patients with PCA. Overall, 35 patients were included, of whom 12 were original cases (males: 25/35 (71%), median age: 65 [range: 20-82]). The most frequent tumor was non-small cell lung cancer (12/35, 34%). Anti-PD1 were adopted in 19/35 (54%). Symptoms developed at a median of 11 weeks after ICI onset. Neuronal antibodies were detected in 15/31 patients tested (48%). Cerebrospinal fluid was inflammatory in 25/30 (83%). Magnetic resonance imaging showed cerebellar hyperintensities in 8/35 (23%). Immunotherapy was applied in 33/35 cases (94%), and most patients improved with residual disability (16/35, 46%). When compared with a series of PCA (n = 15), the cerebellar irAE group was significantly more associated with male sex, lung cancer (rather than gynecological/breast cancers), isolated ataxia, and a better outcome. We provide a detailed characterization of cerebellar irAE. Compared to PCA, differences exist in terms of tumor association, clinical features, and outcome. Clinical presentation-antibody-tumor triad in the ICI group only partially reflects the associations described in paraneoplastic disorders.

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来源期刊
Cerebellum
Cerebellum 医学-神经科学
CiteScore
6.40
自引率
14.30%
发文量
150
审稿时长
4-8 weeks
期刊介绍: Official publication of the Society for Research on the Cerebellum devoted to genetics of cerebellar ataxias, role of cerebellum in motor control and cognitive function, and amid an ageing population, diseases associated with cerebellar dysfunction. The Cerebellum is a central source for the latest developments in fundamental neurosciences including molecular and cellular biology; behavioural neurosciences and neurochemistry; genetics; fundamental and clinical neurophysiology; neurology and neuropathology; cognition and neuroimaging. The Cerebellum benefits neuroscientists in molecular and cellular biology; neurophysiologists; researchers in neurotransmission; neurologists; radiologists; paediatricians; neuropsychologists; students of neurology and psychiatry and others.
期刊最新文献
Correction: Systematic Review and Meta-Analysis of the Diagnostic Accuracy of a Graded Gait and Truncal Instability Rating in Acutely Dizzy and Ataxic Patients. Correction: Long-Term Follow-Up Before and During Riluzole Treatment in Six Patients from Two Families with Spinocerebellar Ataxia Type 7. Correction: Silica Nanoparticles from Melon Seed Husk Abrogated Binary Metal(loid) Mediated Cerebellar Dysfunction by Attenuation of Oxido-inflammatory Response and Upregulation of Neurotrophic Factors in Male Albino Rats. Clinical Heterogeneity of Essential Tremor: Understanding Neural Substrates of Action Tremor Subtypes. The Neuroimmune System and the Cerebellum.
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