Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study

Abstract

Backgrounds: Bevacizumab (BVZ) is used as a subsequent line of treatment for pediatric optic pathway glioma (OPG) in the case of progression. Data on the treatment effect concerning tumor progression and visual function are scarce and nationwide studies are lacking. Methods: We performed a retrospective, nationwide, multicentre cohort study including all pediatric patients with OPG treated with BVZ in the Netherlands (2009–2021). Progression-free survival, change in visual acuity and visual field, MRI-based radiologic response, and toxicity were evaluated. Results: In total, 33 pediatric patients with OPG were treated with BVZ (median 12 months). Visual acuity improved in 20.5%, remained stable in 74.4%, and decreased in 5.1% of 39 of all analysed eyes. The monocular visual field improved in 73.1%, remained stable in 15.4%, and decreased in 7.7% of 25 analysed eyes. Radiologic response at the end of therapy showed a partial response in 7 patients (21.9%), minor response in 7 (21.9%), stable disease in 15 (46.9%), and progressive disease in 3 (9.3%). Progression-free survival at 18 and 36 months after the start of BVZ reduced from 70.9% to 38.0%. Toxicity (≥grade 3 CTCAE) during treatment was observed in five patients (15.2%). Conclusion: Treatment of BVZ in pediatric patients with OPG revealed stabilisation in the majority of patients, but was followed by progression at a later time point in more than 60% of patients. This profile seems relatively acceptable given the benefits of visual field improvement in more than 70% of analysed eyes and visual acuity improvement in more than 20% of eyes at the cessation of BVZ.

Evaluation of Neurofibromatosis Type 1 Associated Optic Pathway Gliomas

“…[20] Also, targeted treatment with bevacizumab led to treatment response after progression in some studies. [21] In our study two patients who were being evaluated for malnutrition, were diagnosed with OPG in MRI. Diencephalic syndrome was observed in these two patients.…”

A telerehabilitation program to improve visual perception in children and adolescents with hemianopia consecutive to a brain tumour: a single-arm feasibility and proof-of-concept trial

“…Case studies have shown that static, spatially and temporally correlated audiovisual stimuli displayed on computer screens can also improve perception in the blind field in adults/older adults with hemianopia after stroke, but require frequent clinic visits 49,50 . In addition, recent retrospective studies reported variable benefits of active Bevacizumab treatment on visual acuity and visual field in children with optic pathway glioma, although these improvements were often lost after discontinuation of the treatment [9][10][11][12] . The mechanisms underlying these temporary improvements are unknown.…”

“…Sparse evidence shows that visual acuity and visual field deficits can be improved with Bevacizumab, an inhibitor of vascular endothelial growth factor, in children with optic nerve gliomas. However, the benefits are not sustained after discontinuation of the drug and there is potential toxicity [9][10][11][12] . In our previous case series 13,14 , we used immersive virtual-reality (IVR) in the Oculus Go headset to deliver a personalized, remotely controlled, dynamic audiovisual stimulation in young adult patients with stable hemianopia (> 18 months) consecutive to a paediatric brain tumour.…”

“…Sparse evidence shows that visual acuity and visual field deficits can be improved with Bevacizumab, an inhibitor of vascular endothelial growth factor, in children with optic nerve gliomas. However, the benefits are not sustained after discontinuation of the drug and there is potential toxicity [9][10][11][12] . In our All rights reserved.…”

Safety of Off-Label Use of Anticancer Drugs in Oncology

“…Средняя продолжительность использования бевацизумаба составила 169 дней [4]. В ряде публикаций продемонстрирована эффективность и безопасность указанного агента при рецидивирующей медуллобластоме и глиоме у детей [6][7][8][9][10][11], что, вероятно, объясняет высокую распространенность его приме-…”