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

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

doi:10.3390/cancers14246087

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

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

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

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.

Original language	English
Article number	6087
Number of pages	15
Journal	Cancers
Volume	14
Issue number	24
DOIs	https://doi.org/10.3390/cancers14246087
Publication status	Published - 10 Dec 2022

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10.3390/cancers14246087Licence: CC BY

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Bennebroek, C. A. M., van Zwol, J., Porro, G. L., Oostenbrink, R., Dittrich, A. T. M., Groot, A. L. W., Pott, J. W., Janssen, E. J. M., Bauer, N. J., van Genderen, M. M., Saeed, P., Lequin, M. H., de Graaf, P., & Schouten-van Meeteren, A. Y. N. (2022). Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study. Cancers, 14(24), Article 6087. https://doi.org/10.3390/cancers14246087

@article{3feae7df5a7f4185904fe07fab04a35b,

title = "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.",

author = "Bennebroek, {Carlien A M} and {van Zwol}, Judith and Porro, {Giorgio L} and Rianne Oostenbrink and Dittrich, {Anne T M} and Groot, {Annabel L W} and Pott, {Jan W} and Janssen, {Etienne J M} and Bauer, {No{\"e}l J} and {van Genderen}, {Maria M} and Peerooz Saeed and Lequin, {Maarten H} and {de Graaf}, Pim and {Schouten-van Meeteren}, {Antoinette Y N}",

note = "Funding Information: This study was funded by ODAS foundation: grant number: 2019–01. Publisher Copyright: {\textcopyright} 2022 by the authors.",

year = "2022",

month = dec,

day = "10",

doi = "10.3390/cancers14246087",

language = "English",

volume = "14",

journal = "Cancers",

issn = "2072-6694",

publisher = "Multidisciplinary Digital Publishing Institute (MDPI)",

number = "24",

}

Bennebroek, CAM, van Zwol, J, Porro, GL, Oostenbrink, R, Dittrich, ATM, Groot, ALW, Pott, JW, Janssen, EJM , Bauer, NJ, van Genderen, MM, Saeed, P, Lequin, MH, de Graaf, P & Schouten-van Meeteren, AYN 2022, 'Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma: A Retrospective Nationwide Multicentre Study', Cancers, vol. 14, no. 24, 6087. https://doi.org/10.3390/cancers14246087

TY - JOUR

T1 - Impact of Bevacizumab on Visual Function, Tumor Size, and Toxicity in Pediatric Progressive Optic Pathway Glioma

T2 - A Retrospective Nationwide Multicentre Study

AU - Bennebroek, Carlien A M

AU - van Zwol, Judith

AU - Porro, Giorgio L

AU - Oostenbrink, Rianne

AU - Dittrich, Anne T M

AU - Groot, Annabel L W

AU - Pott, Jan W

AU - Janssen, Etienne J M

AU - Bauer, Noël J

AU - van Genderen, Maria M

AU - Saeed, Peerooz

AU - Lequin, Maarten H

AU - de Graaf, Pim

AU - Schouten-van Meeteren, Antoinette Y N

PY - 2022/12/10

Y1 - 2022/12/10

N2 - 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.

AB - 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.

U2 - 10.3390/cancers14246087

DO - 10.3390/cancers14246087

M3 - Article

C2 - 36551572

SN - 2072-6694

VL - 14

JO - Cancers

JF - Cancers

IS - 24

M1 - 6087

ER -