Response of Vestibular Schwannomas in NF2-Related Schwannomatosis (NF2-SWN) to Losartan: A Case Series.

IF 1.7 3区 医学 Q3 CLINICAL NEUROLOGY
Sophie Jabban, Spencer Frome, Kara Anstett, Maya Hatley, Kaleb Yohay, J Thomas Roland, Devorah Segal
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引用次数: 0

Abstract

Objective: Identify response rates of vestibular schwannomas in NF2-related Schwanomatosis to treatment with losartan alone or combined with other treatments.

Study design: Single-institution retrospective case series.

Setting: Tertiary referral center.

Patients: Twenty patients with NF2-related Schwannomatosis.

Interventions: Losartan alone, Losartan plus other therapy (bevacizumab, brigatinib, axitinib).

Main outcome measures: Volumetric progression, defined as a change in VS size >20%, in accordance with the Response Evaluation in Neurofibromatosis and Schwannomatosis (REiNS) criteria. Audiometric progression is defined as a change in hearing measures, either a 10 dB or greater increase in PTA or a 10% or greater decrease in WRS.

Results: This study included 20 patients, with 33 VSs, treated at our institution from 2014 to 2023. Losartan treatment duration ranged from 8.09 to 153.04 months (mean: 53.8, SD: 37.32). Twelve VSs were concurrently managed with antineoplastic therapy. Among 21 VSs treated with losartan alone, volumetric regression, progression, and stability were seen in 23.8%, 47.6%, and 28.6% of tumors, respectively. Among 5 VSs treated with combination losartan and bevacizumab, 40% of tumors regressed, 20% progressed, and 40% remained stable during the combination period. One tumor that progressed during bevacizumab monotherapy treatment achieved volumetric stability following combination therapy. Volumetric growth rates during bevacizumab alone did not significantly differ from growth rates following combination therapy.

Conclusions: This case series describes response rates among NF2-SWN patients treated with losartan. The potential role of losartan in modulating tumor size, either alone or in combination with bevacizumab, should be further studied through well-controlled studies. The study is limited by a small sample size.

前庭神经鞘瘤在nf2相关神经鞘瘤病(NF2-SWN)中对氯沙坦的反应:一个病例系列
目的:确定nf2相关神经鞘瘤患者前庭神经鞘瘤对氯沙坦单用或联用其他治疗的有效率。研究设计:单机构回顾性病例系列。单位:三级转诊中心。患者:20例nf2相关神经鞘瘤病。干预措施:氯沙坦单独,氯沙坦加其他治疗(贝伐单抗,布加替尼,阿西替尼)。主要结局指标:根据神经纤维瘤病和神经鞘瘤病(REiNS)反应评估标准,体积进展,定义为VS大小变化>20%。听力测量进展被定义为听力测量的变化,PTA增加10 dB或更多,或WRS下降10%或更多。结果:本研究纳入了2014年至2023年在我院治疗的20例患者,33例VSs。氯沙坦治疗时间8.09 ~ 153.04个月(平均:53.8个月,标准差:37.32)。12例VSs患者同时接受抗肿瘤治疗。在单独使用氯沙坦治疗的21例VSs中,分别有23.8%、47.6%和28.6%的肿瘤出现体积消退、进展和稳定性。在联合氯沙坦和贝伐单抗治疗的5例VSs中,40%的肿瘤消退,20%的肿瘤进展,40%的肿瘤在联合期间保持稳定。在贝伐单抗单药治疗期间进展的一个肿瘤在联合治疗后达到了体积稳定性。贝伐单抗单独治疗期间的体积增长率与联合治疗后的增长率没有显著差异。结论:本病例系列描述了氯沙坦治疗的NF2-SWN患者的缓解率。氯沙坦在调节肿瘤大小方面的潜在作用,无论是单独使用还是与贝伐单抗联合使用,都应通过良好的对照研究进一步研究。这项研究受到样本量小的限制。
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来源期刊
Otology & Neurotology
Otology & Neurotology 医学-耳鼻喉科学
CiteScore
3.80
自引率
14.30%
发文量
509
审稿时长
3-6 weeks
期刊介绍: ​​​​​Otology & Neurotology publishes original articles relating to both clinical and basic science aspects of otology, neurotology, and cranial base surgery. As the foremost journal in its field, it has become the favored place for publishing the best of new science relating to the human ear and its diseases. The broadly international character of its contributing authors, editorial board, and readership provides the Journal its decidedly global perspective.
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