Visual field outcomes in children treated for neurofibromatosis type 1–associated optic pathway gliomas: a multicenter retrospective study

Heidary, Gena; Fisher, Michael J.; Liu, Grant T.; Ferner, Rosalie E.; Gutmann, David H.; Listernick, Robert; Kapur, Kush; Loguidice, Michael J.; Ardern‐Holmes, Simone; Avery, Robert A.; Hammond, Christopher J; Hoffman, Robert O.; Hummel, Trent R.; Kuo, Annie; Reginald, Arun; Ullrich, Nicole J.

doi:10.1016/j.jaapos.2020.07.013

Cited by 8 publications

(9 citation statements)

References 12 publications

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“…Chiasmal and optic tract glioma mostly cause a combined decrease of BCVA and VF, but studies on VF as the outcome parameter for treatment of OPG are limited. Fisher et al presented VF evaluation in 30 eyes after first order SAT, with stabilisation or improvement in 63% [ 31 ]. Fangusaro et al reported on 19 patients treated with selumetinib, 26% of whom showed improvement and 74% of whom showed stabilisation of binocular VF [ 32 ].…”

Section: Discussionmentioning

confidence: 99%

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

Bennebroek

Zwol

Porro³

et al. 2022

Cancers

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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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Section: Discussionmentioning

confidence: 99%

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

Bennebroek

Zwol

Porro³

et al. 2022

Cancers

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“…Clinical evaluations with confrontation alone (without formal perimetry) were excluded. Clinically significant change in VF was defined as change between (1) full field and any field loss, (2) quadrantic and hemifield loss, or (3) generalized constriction or expansion of VF 4 . Radiographic response was assessed from clinical notes and radiology reports at baseline and at the end of BBT.…”

Section: Methodsmentioning

confidence: 99%

“…Radiographic response to treatment is poorly correlated with visual outcome, leading visual acuity (VA) to be included as an outcome in modern clinical trials 3 . Patients with OPG are also vulnerable to visual field (VF) deficits, and there is emerging evidence that VF deficits can be present even in the absence of VA impairment 4 . Biologic therapies have emerged as viable alternatives to traditional chemotherapy for the treatment of OPG.…”

Section: Introductionmentioning

confidence: 99%

Visual outcomes after bevacizumab-based therapy for optic pathway glioma

Siegel

Nelson

et al. 2023

Preprint

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In optic pathway glioma (OPG), bevacizumab-based therapy (BBT) has promising effects on radiographic tumor burden, but impact on vision is less clear. This single-institution study characterized visual acuity (VA) and visual field (VF) outcomes in 17 pediatric OPG patients treated with BBT. VA was stable or improved in 14 patients. Nine patients had evaluable VF data, 6 of whom experienced stability or improvement. Among 6 patients with vision deterioration as a treatment indication, stable or improved was observed for both VA and VF in 5 patients. In summary, BBT was associated with favorable visual outcomes in most patients with OPG.

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“…3 Patients with OPG are also vulnerable to visual field (VF) deficits, and there is evidence that VF deficits can be present even in the absence of VA impairment. 4 Biological therapies have emerged as viable alternatives to traditional chemotherapy for the treatment of OPG. Bevacizumab is a Abbreviations: BBT, bevacizumab-based therapy; NF1, neurofibromatosis type 1; OPG, optic pathway glioma; VA, visual acuity; VF, visual fields.…”

Section: Introductionmentioning

confidence: 99%

“…Radiographic response to treatment is poorly correlated with visual outcome, leading visual acuity (VA) to be included as an outcome in modern clinical trials 3 . Patients with OPG are also vulnerable to visual field (VF) deficits, and there is evidence that VF deficits can be present even in the absence of VA impairment 4 …”

Section: Introductionmentioning

confidence: 99%

Visual outcomes after bevacizumab‐based therapy for optic pathway glioma

Siegel,

Nelson,

Peragallo

et al. 2023

Pediatric Blood & Cancer

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In optic pathway glioma (OPG), bevacizumab‐based therapy (BBT) has promising effects on radiographic tumor burden, but the impact on vision is less clear. This single‐institution study characterized visual acuity (VA) and visual field (VF) outcomes in 17 pediatric OPG patients treated with BBT. VA was stable or improved in 14 patients. Nine patients had evaluable VF data, six of whom experienced stability or improvement. Among six patients with vision deterioration as a treatment indication, stable or improved was observed for both VA and VF in five patients. In summary, BBT was associated with favorable visual outcomes in this cohort of patients with OPG.

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Visual field outcomes in children treated for neurofibromatosis type 1–associated optic pathway gliomas: a multicenter retrospective study

Cited by 8 publications

References 12 publications

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

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

Visual outcomes after bevacizumab-based therapy for optic pathway glioma

Visual outcomes after bevacizumab‐based therapy for optic pathway glioma

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