Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University, Baltimore, Maryland, U.S.A.
Tufts University School of Medicine, Boston, Massachusetts, U.S.A.
Laryngoscope. 2021 May;131(5):1138-1146. doi: 10.1002/lary.29084. Epub 2020 Sep 22.
Juvenile onset recurrent respiratory papillomatosis (JORRP) can cause severe or disseminated disease. Surgical treatment may be inadequate. Systemic bevacizumab has shown initial success for severe JORRP. The objective of this systematic review was to assess usage, effectiveness, and safety of this treatment.
We searched PubMed, Embase, and Web of Science for studies of humans with JORRP treated with systemic bevacizumab. Two researchers independently reviewed the studies to determine inclusion and aggregate data on patient characteristics, dosing protocols, treatment response, adverse events, and level of evidence.
Of 80 identified articles, 12 studies were included detailing 20 distinct cases. At a mean age of 12.8 years (range = 1-43 years) patients received initial dosing of 5 to 10 mg/kg of bevacizumab followed by ongoing doses at a mean 3-week intervals (range = 2-5 weeks). All patients had clinically significant disease reduction with reduced need for surgery. Six patients (30%) had complete response in at least one involved anatomic site. Eleven (55%) required no surgery after initiating treatment. There was recurrence in all four patients whose treatment was stopped, but had rapid improvement with treatment resumption. Six (30%) experienced mild or moderate adverse events.
Marked improvement in severe JORRP has been reported from systemic bevacizumab. Treatment protocols vary, and treatment discontinuation was not feasible in any reported patient. Based on currently available data, systemic bevacizumab can be considered for severe JORRP as it appears to be well tolerated and effective. A clinical trial could enhance the understanding of its safety and efficacy for this indication. Laryngoscope, 131:1138-1146, 2021.
青少年复发性呼吸道乳头瘤病(JORRP)可导致严重或播散性疾病。手术治疗可能效果不足。系统使用贝伐珠单抗已显示出对严重 JORRP 的初步成功。本系统评价的目的是评估该治疗方法的使用、疗效和安全性。
我们在 PubMed、Embase 和 Web of Science 中搜索了接受系统贝伐珠单抗治疗的 JORRP 人类患者的研究。两名研究人员独立审查了这些研究,以确定纳入标准,并汇总患者特征、剂量方案、治疗反应、不良事件和证据水平的数据。
在 80 篇确定的文章中,有 12 项研究被纳入,详细描述了 20 个不同的病例。患者的平均年龄为 12.8 岁(范围=1-43 岁),初始剂量为 5 至 10mg/kg 贝伐珠单抗,然后以平均 3 周的间隔(范围=2-5 周)持续用药。所有患者的疾病均有明显改善,减少了手术需求。6 名患者(30%)至少在一个受累解剖部位有完全缓解。11 名患者(55%)在开始治疗后无需手术。所有停止治疗的患者均有复发,但恢复治疗后迅速改善。6 名患者(30%)出现轻度或中度不良反应。
系统使用贝伐珠单抗可显著改善严重 JORRP。治疗方案各不相同,在任何报告的患者中均无法停止治疗。根据目前可用的数据,系统使用贝伐珠单抗可用于严重 JORRP,因为它似乎耐受性良好且有效。一项临床试验可以增强对其用于该适应症的安全性和疗效的理解。《喉镜》,131:1138-1146,2021。