Visual acuity of children treated with chemotherapy for optic pathway gliomas

Kalin-Hajdu, Evan; Décarie, Jean‐Claude; Marzouki, Monia; Carret, Anne‐Sophie; Ospina, Luis H.

doi:10.1002/pbc.24726

Cited by 43 publications

(42 citation statements)

References 20 publications

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“…This is in keeping with our own previous experience . Other publications have found similar rates, with the most common outcome after chemotherapy being stability of vision …”

Section: Introductionsupporting

confidence: 92%

Long‐term visual outcome after chemotherapy for optic pathway glioma in children: Site and age are strongly predictive

Dodgshun

Elder

Hansford

et al. 2015

Cancer

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BACKGROUND: Optic pathway gliomas (OPGs) are commonly noted in pediatric oncology services. Radiotherapy is effective at controlling tumors, but has many undesirable late effects, especially in patients with neurofibromatosis. Chemotherapy is commonly used to preserve vision and delay or eliminate the need for radiotherapy. Despite visual threat being a common reason to initiate chemotherapy in patients with OPG, reports of visual outcome after chemotherapy are not common and reports of long-term visual outcome are even scarcer. METHODS: In a single institution, all patients with OPG who had received chemotherapy or radiotherapy between 1996 and 2013 were identified from hospital databases. Visual, treatment, and radiological data were recorded. Categorized visual acuity was the primary outcome measure. RESULTS: Of 43 patients identified, visual data were available for 42 patients. Approximately 14% of patients experienced an improvement in visual acuity during therapy, 9% of patients experienced a deterioration, and the remainder were stable. At a mean follow-up of 78 months, 26% of patients were legally blind. Children aged <2 years and patients with a chiasmatic/hypothalamic tumor site were overrepresented in this category. An intraconal location was predictive of poor visual outcome for that eye but was unilateral with normal vision in the contralateral eye. CONCLUSIONS: Risk factors for long-term visual deterioration are young age, chiasmatic/hypothalamic tumor site, and intraconal tumor site for the involved eye. The most common visual outcome for children with OPG after treatment with chemotherapy is stability. This stability is maintained over the long term for >90% of children without these risk factors. Cancer 2015;121:4190-6.

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“…This is in keeping with our own previous experience . Other publications have found similar rates, with the most common outcome after chemotherapy being stability of vision …”

Section: Introductionsupporting

confidence: 92%

Long‐term visual outcome after chemotherapy for optic pathway glioma in children: Site and age are strongly predictive

Dodgshun

Elder

Hansford

et al. 2015

Cancer

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“…The overall rate of progression in the study was high at 74 %, adding to the accumulating body of evidence that sporadic OPGs have a greater risk of progression compared to NF1-associated tumors [6,[19][20][21][22][23][24]. At present, there is no universally accepted definition of progression and existing studies have used variable radiological and clinical criteria to define progression [16,25]. One thing that has become clear is that radiological changes and changes in visual function are not always correlated [16,[26][27][28].…”

Section: Chemotherapymentioning

confidence: 97%

Long-term visual outcomes of optic pathway gliomas in pediatric patients without neurofibromatosis type 1

et al. 2016

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Sporadic optic pathway gliomas (OPGs) have been reported to cause more vision loss than OPGs associated with neurofibromatosis type-1, but long-term visual outcome data are limited. The purpose of this study was to report the visual outcomes of a cohort of pediatric patients with sporadic OPGs. This was a retrospective, cohort study at a tertiary care pediatric hospital and cancer institute. The study included all patients with sporadic OPGs evaluated from 1990 to 2014. The primary outcome was visual acuity at final follow-up. Secondary outcomes were risk factors for a poor visual outcome and the rate of progression. There were 59 pediatric patients included in the study. Median age at presentation was 2.5 years old and median follow-up was 5.2 years. In the worse eye at final follow-up, 16 patients (27 %) were 20/30 or better, 9 patients (15 %) were between 20/40 and 20/80, and 34 patients (58 %) were 20/100 or worse. In the better eye at final follow-up, 33 patients (56 %) were 20/30 or better, 11 patients (19 %) were between 20/40 and 20/80, and 15 patients (25 %) were 20/100 or worse. Risk factors for a poor visual outcome included younger age at presentation, optic nerve pallor, and tumor extent. Of the 54 patients (92 %) who received treatment, 40 (74 %) experienced disease progression during or after treatment. A majority of pediatric patients with sporadic OPGs had significant long-term visual impairment. In spite of treatment, tumor progression is common. Serial ophthalmic examinations with quantitative vision measurements are essential in the management of sporadic OPGs.

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“…There is a report of four cases of refractory OPG (two sporadic and two NF1-associated OPG) that showed marked improvement in VA following treatment with bevacizumab [34]. These agents rarely restore the premorbid visual acuity and the aim of treatment is usually to stabilize disease and prevent further worsening [35,36]. Radiotherapy is usually avoided in NF1-associated OPG for concern of secondary tumors [37] and moya moya syndrome [38] Surgical excision of OPG is not feasible due to the tumor location and is usually reserved for instances of complete loss of vision, severe proptosis, or hydrocephalus.…”

Section: Nf1-associated Gliomasmentioning

confidence: 99%

Therapeutic Development in Neurofibromatosis

Lobbous¹,

Korf²

2020

Neurofibromatosis - Current Trends and Future Directions

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Although neurofibromatosis (NF) was initially recognized in the nineteenth century, only in the past two decades we have witnessed a paradigm shift in therapeutics. This progress is driven by the increasing understanding of the natural history of the NF-associated tumors and understanding of the molecular landscape of these disorders. Multiple clinical trials have been launched evaluating non-surgical treatment modalities and more studies are in the pipeline. Recently, the NF community has adopted standardized endpoints recommended by the Response Evaluation in Neurofibromatosis and Schwannomatosis (REiNS) International Collaboration established in 2011. Such collaborations among academic, regulatory and supporting communities are crucial for providing the infrastructure needed for advancing the therapeutic development in the field of NF.

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Visual acuity of children treated with chemotherapy for optic pathway gliomas

Abstract: In both NF1 mutant and sporadic OPGs, VA deteriorated directly following chemotherapy as well as at long-term follow-up. Despite chemotherapy, eyes with severe functional impairment gradually increased over time.

Cited by 43 publications

References 20 publications

Long‐term visual outcome after chemotherapy for optic pathway glioma in children: Site and age are strongly predictive

Long‐term visual outcome after chemotherapy for optic pathway glioma in children: Site and age are strongly predictive

Long-term visual outcomes of optic pathway gliomas in pediatric patients without neurofibromatosis type 1

Therapeutic Development in Neurofibromatosis

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