This thesis aimed to provide a second validation phase of a recently developed measure: the Parental Understanding of Neurodisability Questionnaire (PUN-Q) (Moran et al., submitted). The PUN-Q is a thirteen item self-report questionnaire measuring parents' understanding of their child's neurodisability. This thesis prospectively validated the PUN-Q over three time points, prior to and following a child's attendance at a Tier-Four paediatric diagnostic assessment, for queries regarding social communication. Four main aims were investigated: 1) to establish prospective Construct Validity by comparing the PUN-Q to two other parent-report measures (perceived self-efficacy and parenting stress); 2) to examine test-retest reliability of the PUN-Q by comparing two pre-assessment time points; 3) to examine whether the PUN-Q is sensitive measuring potential pre-and-post assessment changes to parental understanding; 4) to explore the relationship between the PUN-Q and child emotional, behavioural and social communication difficulties. These objectives were addressed using data collected from 37 parents, due to time constraints the study was underpowered at Times two and three (n=26, n=11, respectively); bootstrapping confidence intervals were therefore estimated for non-parametric data. Evidence was provided for Construct Validity at Time 1, but not at Time 3. Test-retest reliability was suggested for the PUN-Q between two non-intervention time points. Results suggested that the PUN-Q is responsive to changes over time, and that the clinic's diagnostic assessment is effective in enhancing parental understanding. The PUN-Q was not shown to be related to child-related outcomes. These preliminary results suggest that the PUN-Q is an important measure that can reliably and conveniently measure parental understanding of their child's neurodisability symptoms. This study suggests a role for parental understanding within a wider model of parenting stress and coping with disability. Further validation is needed to allow dissemination to the wider neurodisability service, and to less complex symptom presentations.

Injuries to the brain are the leading cause of permanent disability and death. Survivors of acquired brain injury (ABI) experience cognitive impairments and emotional problems. These often persist into community rehabilitation and are among the most significant needs for those in chronic stages of rehabilitation. There is a dearth of research providing evidence of music therapy addressing cognitive deficits and emotional needs in a holistic approach. This research answers the question how can brief group music therapy address cognitive functional gains and emotional needs of people with acquired brain injury. A mixed methods design was used to investigate the effect of 16 sessions of weekly group music therapy on attention and memory impairments, and emotional needs of ten ABI survivors in community rehabilitation. Quantitative data were collected to determine the effect of treatment on attention and memory functioning, mood state, and the satisfaction of emotional needs. Qualitative data were collected to reveal survivors’ experiences of brain injury and brief group music therapy. Analysis of the data showed that the intervention improved sustained attention (p<.05, r=.80) and immediate memory recall (p>.05, r=.46), and that the effect of treatment increased with dosage. Overall, the intervention was more effective than standard care, and cognitive functional gains continued after treatment for some ABI survivors. The intervention addressed emotional needs of feeling confident (p<.05, d=.88), feeling part of a group (p<.05, d=.74), feeling productive/useful (p<.05, d=.90), feeling supportive (p<.05, d=.75), feeling valued (p<.05, d=.74), and enjoyment (p<.05, d=.34). Improvements in these domains were observed in the immediate term and over the course of therapy. Music therapy enabled emotional adjustment through the development of selfawareness and insight. This study offers a music therapy method to deliver a holistic approach in rehabilitation. It demonstrates that music therapy can provide a cost effective, holistic treatment for ABI survivors.

L’objet de cette thèse est de réaliser une analyse économique du champ de l’aide informelle des personnes adultes jeunes vivant à domicile et atteintes de maladie neurologique grave. La question de recherche posée concerne l’articulation entre l’organisation de l’aide informelle et la cinétique de la pathologie neurologique. La méthodologie repose sur la construction de quatre modèles neuro-pathologiques et économiques d’une part, et sur la constitution de quatre cohortes représentatives, comportant chacune une centaine de binômes « aidant-aidés » d’autre part. Ainsi, les modèles de la pathologie brutale avec handicap résiduel stabilisé, de la pathologie progressive avec handicap croissant, de la pathologie à cinétique déficitaire rapide, de la pathologie dégénérative héréditaire sont-ils respectivement illustrés par le traumatisme crânien, la sclérose en plaques, la tumeur cérébrale et la maladie de Huntington. Nos travaux (1) mettent en évidence les caractéristiques sur le plan économique des aidants informels (conjoints jeunes) qui sont fortement impliqués dans la production du soin, experts d’un accompagnement complexe et déstabilisés dans leur trajectoire professionnelle (2) font la démonstration de la prédétermination forte de la cinétique de la pathologie neurologique sur les comportements d’aide informelle à travers différents indicateurs temporels dont la prise en compte s’avère incontournable pour l’analyse économique (3) montrent la nécessité d’avoir recours à une mesure bidimensionnelle (subjective et objective) dans l’analyse du fardeau des aidants informels. Une telle mesure souligne d’une part l’insuffisance du recours à l’aide professionnelle publique et d’autre part l’impact sur les aidants des troubles cognitivo-comportementaux (handicap invisible) et de la phase neuro-palliative à domicile d’une pathologie neurologique grave. Ces résultats ouvrent des perspectives pour la mise en place de mesures d’action publiques en France dans le champ complexe du handicap neurologique. / The purpose of this thesis is to achieve an economic analysis of informal caregiving of young adults living at home and suffering from a severe neurological disease. The research questions the relationship between the organization of informal care and kinetics of neurological pathology. The methodology is based on the construction of four neuro-pathological and economic models on the one hand, and on the constitution of four representative cohorts, each with a hundred pairs of "patients-caregivers" on the other. Thus, models of brutal disease stabilized with residual disability, progressive disease with increasing disability, fast kinetics disease and neuro-degenerative hereditary disease are respectively illustrated by traumatic brain injury, multiple sclerosis, malignant brain tumor and Huntington's Disease. Our work (1) highlights the economic characteristics of informal caregivers (young spouses) who are highly involved in the production of care, expert of complex care and therapeutic pathways and destabilized in their professional careers (2) demonstrates that the kinetics of neurological disease predicts the economic behavior of informal caregivers : taking account of different time indicators is crucial for economic analysis in neurodisability (3) shows that a two-dimensional subjective and objective outcome measure is necessary in the analysis of the burden of informal caregivers. Such a double indicator first stresses the inadequate use of publicly funded professional care ; it also points out the impact of cognitive-behavioral disorders (so-called “invisible disability”) and of the home neuro-palliative phase on caregivers in case of a severe neurological disease. These results open perspectives for the development of public action measures in France in the complex field of neurological disability.