Green, Katherine;
(2024)
Development of a multi-factorial prognostic model to optimise treatment decision making and outcomes in paediatric low-grade glioma: a mixed methods study.
Doctoral thesis (M.D(Res)), UCL (University College London).
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Abstract
Background: PLGG is a histologically benign and generally survivable CNS tumour of childhood. Varying morbidities from heterogeneous patient, tumour and treatment factors can afflict life-long functional and quality of life (QOL) impairments. Whilst prognostication of survival outcomes is established, there has been limited assessment of long-term PLGG QOL and functional outcomes, and no attempts to prognosticate for functional outcomes. / Aims: To assess long-term functional and QOL outcomes of the GOSH PLGG cohort, and to analyse associations between prognostic variables and overall survival (OS), progression-free survival (PFS), functional, & QOL outcomes. / Methods: A mixed-methods study comprising three work-packages to evaluate a large PLGG cohort alongside individual PLGG survivor narratives. Patients diagnosed with PLGG at GOSH 1980-2021 were retrospectively evaluated for patient, tumour and treatment variables, and OS, PFS & functional outcomes from medical records. Eligible survivors were invited to complete prospective QOL (PedsQL Core) and functional (PESAT-PT) questionnaires, with these outcomes evaluated descriptively and prognostically with patient, tumour and treatment variables. Ten long-term PLGG survivors completed semi-structured interviews to elicit PLGG survivor narratives. / Results: 814 patients with PLGG were included retrospectively, and 122 prospectively. Age at diagnosis, NF1, tumour location, surgical resection, and radiotherapy were prognostic indicators for functional and QOL outcomes on multivariate logistic regression analysis. 87% survivors reported long-term functional impairment. Average PedsQL scores of PLGG survivors at a median of 21 years from diagnosis were significantly lower than healthy adult reference populations, and comparable to adults with chronic illnesses. / Conclusions: PLGG is a chronic disease. Long-term functional and QOL outcomes of survivors were heterogeneous, but not infrequently impacted, within our large PLGG cohort. Prognostication of long-term PLGG functional and QOL outcomes is possible, but complex. Combining a large PLGG cohort dataset with individual PLGG survivor narratives provided enriched meaning and context for data interpretation.
| Type: | Thesis (Doctoral) |
|---|---|
| Qualification: | M.D(Res) |
| Title: | Development of a multi-factorial prognostic model to optimise treatment decision making and outcomes in paediatric low-grade glioma: a mixed methods study |
| Open access status: | An open access version is available from UCL Discovery |
| Language: | English |
| Additional information: | Copyright © The Author 2024. Original content in this thesis is licensed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International (CC BY-NC 4.0) Licence (https://creativecommons.org/licenses/by-nc/4.0/). Any third-party copyright material present remains the property of its respective owner(s) and is licensed under its existing terms. Access may initially be restricted at the author’s request. |
| Keywords: | Paediatric Low-Grade Glioma, Quality of Life, Functional Outcomes |
| UCL classification: | UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health UCL |
| URI: | https://discovery.ucl.ac.uk/id/eprint/10193669 |
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