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Visual outcomes after chemotherapy for optic pathway glioma in children with and without neurofibromatosis type 1: results of the International Society of Paediatric Oncology (SIOP) Low-Grade Glioma 2004 trial UK cohort
  1. Kevin Falzon1,2,
  2. Evangelos Drimtzias1,
  3. Susan Picton1,
  4. Ian Simmons1
    1. 1 Leeds Teaching Hospitals NHS Trust, Leeds, UK
    2. 2 Department of Ophthalmology, York Teaching Hospital NHS Foundation Trust, York, UK
    1. Correspondence to Dr Kevin Falzon, Department of Ophthalmology, York Teaching Hospital NHS Foundation Trust, York YO31 8HE, UK; kevin.falzon{at}hotmail.com

    Abstract

    Aims To report visual acuity (VA) outcomes following chemotherapy for optic pathway glioma (OPG) in children with or without neurofibromatosis type-1 (NF1) and to analyse associated risk factors.

    Methods A prospective, multicentre, cohort study involving 155 children treated between September 2004 and December 2012. Initial and final VA was used for per-eye and per-subject analysis. Correlation tests were performed to determine whether initial VA predicted final VA. Logistic regression was used to determine whether age and tumour location were associated risk factors.

    Results 90 children had complete ophthalmological data. At initiation of chemotherapy, 26% and 49% of eyes with NF1-OPG and sporadic OPG, respectively, had VA of ≥0.7 log of the minimum angle of resolution (logMAR). At final visit, per eye, 49% had ≤0.2, 23% had 0.30–0.60 and 28% had VA≥0.70 logMAR in the NF1-OPG group. In the sporadic OPG group, per eye, 32% had ≤0.2, 11% had VA 0.30–0.60 and 57% had ≥0.70 logMAR. Children with sporadic OPG, per eye, were significantly less likely to have VA outcomes ≤0.60 logMAR compared with children with NF1-OPG (OR=0.30; 95% CI 0.16 to 0.56; P<0.0001). Per subject, VA improved in 24%, remained stable in 35% and worsened in 41% of children with NF1-OPG and improved in 18%, remained stable in 43% and worsened in 39% of children with sporadic OPG.

    Conclusions Children with and without NF1 demonstrated the same rate of VA improvement, stabilisation or worsening; however, children with sporadic OPG had a poorer VA outcome. Better initial VA, older age, absence of postchiasm tumour and presence of NF1 were associated with improved or stable VA outcomes.

    • optic nerve
    • visual pathway
    • child health (paediatrics)

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    Footnotes

    • KF and ED contributed equally.

    • Contributors KF and ED contributed towards the acquisition, analysis and interpretation of data for the work; drafting the work; revising it critically for important intellectual content; final approval of the version to be published; and agreed to be accountable for all aspects of the work. SP contributed towards the conception and design of the work and agreed to be accountable for all aspects of the work. IS contributed towards the conception and design of the work; revising it critically for important intellectual content; and agreed to be accountable for all aspects of the work.

    • Competing interests None declared.

    • Ethics approval MREC/03/4/108 East Midlands – Derby Research Ethics Committee (previously Trent Multi-centre Research Ethics Committees) The Old Chapel, Royal Standard Place Nottingham NG1 6FS.

    • Provenance and peer review Not commissioned; externally peer reviewed.

    • Collaborators Jack Gormley (Leeds Teaching Hospitals); Olwyn Nelson (Leeds Teaching Hospitals); Janice Hoole (Leeds Teaching Hospitals); Danielle Guy (Leeds Teaching Hospitals); Martha Campos (Cancer Research UK Clinical Trials Unit, Birmingham); Safoora Baig (Cancer Research UK Clinical Trials Unit, Birmingham).

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