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Title: Capturing health-related quality of life in adults with Charcot-Marie-Tooth disease : a digital photovoice investigation
Author: Walklet, Elaine
ISNI:       0000 0004 7425 6885
Awarding Body: University of the West of England
Current Institution: University of the West of England, Bristol
Date of Award: 2018
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Rationale: CMT is a progressive and debilitating neuromuscular condition which has previously been linked with reduced HRQoL. However, little is known about what adults with CMT think about their HRQoL and the various factors that influence it. An in-depth understanding of experience is essential for valid assessment of HRQoL and appropriate intervention. Aim: The aim of this study was to explore how adults living with CMT understand HRQoL and the factors which influence this. Method: A digital photovoice study was conducted. Eleven participants with varying types of CMT completed an online journal over a 2-week period. Participant were asked to photograph anything which they felt influenced their quality of life (positively, negatively, or neutrally) and to write an accompanying reflective narrative. On average, participants posted 8.36 journal entries (including at least one photo and a narrative reflection) during the 2-week study duration. Findings: Thematic analysis of the textual and visual data identified 6 main themes: “The challenge of the every-day”, “Maintaining independence: adaption, adjustment and support”, “Loss of control and future-orientated anxiety”, “CMT Awareness”, “Threats to identity and self-esteem” and “Resilience and positive growth”. Overall, HRQoL emerged as a complex, individual and multi-faceted construct which is influenced by a range of internal and external factors. Conclusions: Whilst the physical aspects of CMT are inherently challenging, threats to HRQoL also arise from the physical and social environment and the psychological impact of living with a progressive, hereditary condition. Despite much adversity, all participants demonstrated substantial resilience and positive growth which helped them to manage the condition and enhanced their HRQoL. A CMT-specific measure of HRQoL and targeted interventions are now urgently required.
Supervisor: Not available Sponsor: Not available
Qualification Name: Thesis (D.Health Psych.) Qualification Level: Doctoral
EThOS ID:  DOI: Not available
Keywords: CMT ; quality of life ; Charcot-Marie-Tooth ; photovoice