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

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 retrospec...

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Main Authors: Carlien A. M. Bennebroek, Judith van Zwol, Giorgio L. Porro, Rianne Oostenbrink, Anne T. M. Dittrich, Annabel L. W. Groot, Jan W. Pott, Etienne J. M. Janssen, Noël J. Bauer, Maria M. van Genderen, Peerooz Saeed, Maarten H. Lequin, Pim de Graaf, Antoinette Y. N. Schouten-van Meeteren
Format: Article
Language:English
Published: MDPI AG 2022-12-01
Series:Cancers
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Online Access:https://www.mdpi.com/2072-6694/14/24/6087
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author Carlien A. M. Bennebroek
Judith van Zwol
Giorgio L. Porro
Rianne Oostenbrink
Anne T. M. Dittrich
Annabel L. W. Groot
Jan W. Pott
Etienne J. M. Janssen
Noël J. Bauer
Maria M. van Genderen
Peerooz Saeed
Maarten H. Lequin
Pim de Graaf
Antoinette Y. N. Schouten-van Meeteren
author_facet Carlien A. M. Bennebroek
Judith van Zwol
Giorgio L. Porro
Rianne Oostenbrink
Anne T. M. Dittrich
Annabel L. W. Groot
Jan W. Pott
Etienne J. M. Janssen
Noël J. Bauer
Maria M. van Genderen
Peerooz Saeed
Maarten H. Lequin
Pim de Graaf
Antoinette Y. N. Schouten-van Meeteren
author_sort Carlien A. M. Bennebroek
collection DOAJ
description 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.
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spelling doaj.art-a44b8d4eee9e411fa42d754b99cee93b2023-11-24T13:45:56ZengMDPI AGCancers2072-66942022-12-011424608710.3390/cancers14246087Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre StudyCarlien A. M. Bennebroek0Judith van Zwol1Giorgio L. Porro2Rianne Oostenbrink3Anne T. M. Dittrich4Annabel L. W. Groot5Jan W. Pott6Etienne J. M. Janssen7Noël J. Bauer8Maria M. van Genderen9Peerooz Saeed10Maarten H. Lequin11Pim de Graaf12Antoinette Y. N. Schouten-van Meeteren13Department of Ophthalmology, Amsterdam UMC Location University of Amsterdam, 1053 VE Amsterdam, The NetherlandsDepartment of Ophthalmology, Amsterdam UMC Location University of Amsterdam, 1053 VE Amsterdam, The NetherlandsDepartment of Ophthalmology Utrecht UMC, 3584 CX Utrecht, The NetherlandsENCORE-NF1 Center, Department of General Pediatrics, Erasmus MC, 3015 GD Rotterdam, The NetherlandsDepartment of Pediatrics, Radboud University Medical Center, Amalia Children’s Hospital, 6525 GA Nijmegen, The NetherlandsDepartment of Ophthalmology, Radboud University Medical Center, 6525 GA Nijmegen, The NetherlandsDepartment of Ophthalmology, University Medical Center Groningen, University of Groningen, 9713 GZ Groningen, The NetherlandsDepartment of Neurology, Maastricht University Medical Center, 6221 CZ Maastricht, The NetherlandsDepartment of Ophthalmology, Maastricht University Medical Center, 6229 HX Maastricht, The NetherlandsDepartment of Ophthalmology Utrecht UMC, 3584 CX Utrecht, The NetherlandsDepartment of Ophthalmology, Amsterdam UMC Location University of Amsterdam, 1053 VE Amsterdam, The NetherlandsDepartment of Radiology, Princess Máxima Center for Pediatric Oncology, 3584 CS Utrecht, The NetherlandsDepartment of Radiology and Nuclear Medicine, Amsterdam UMC Location Vrije Universiteit Amsterdam, 1081 HV Amsterdam, The NetherlandsDepartment of Neuro-Oncology, Princess Máxima Center for Pediatric Oncology, 3584 CS Utrecht, The NetherlandsBackgrounds: 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.https://www.mdpi.com/2072-6694/14/24/6087bevacizumaboptic pathway gliomalow grade gliomachildsystemic anticancer therapyvisual function
spellingShingle Carlien A. M. Bennebroek
Judith van Zwol
Giorgio L. Porro
Rianne Oostenbrink
Anne T. M. Dittrich
Annabel L. W. Groot
Jan W. Pott
Etienne J. M. Janssen
Noël J. Bauer
Maria M. van Genderen
Peerooz Saeed
Maarten H. Lequin
Pim de Graaf
Antoinette Y. N. Schouten-van Meeteren
Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study
Cancers
bevacizumab
optic pathway glioma
low grade glioma
child
systemic anticancer therapy
visual function
title Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study
title_full Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study
title_fullStr Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study
title_full_unstemmed Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study
title_short Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study
title_sort impact of bevacizumab on visual function tumor size and toxicity in pediatric progressive optic pathway glioma a retrospective nationwide multicentre study
topic bevacizumab
optic pathway glioma
low grade glioma
child
systemic anticancer therapy
visual function
url https://www.mdpi.com/2072-6694/14/24/6087
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