Preliminary development of proxy-rated quality-of-life scales for children and adults with Niemann-Pick type C

Aston, Lydia; Shaw, Rachel; Knibb, Rebecca

doi:10.1007/s11136-019-02234-5

Cited by 2 publications

(1 citation statement)

References 25 publications

(31 reference statements)

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“…Our findings present novel conceptual insights to MS carers' QoL which can be used to generate new theoretical models or adapt existing ones to explain QoL in the MS caregiving context, and suggest that a more holistic conceptualization of QoL is needed to guide empirical research and practical efforts to better support MS carers. The impact of caregiving on QoL may be considered within the conceptual definitions of lifeworld-led care (Todres, Galvin, & Dahlberg, 2006), a model previously used in the context of caregiving (Ashworth, 2006) and understanding QoL (Aston, Shaw, & Knibb, 2019). The lifeworld is a holistic outlook in which individuals make sense of the world in relation to time, space, body, others, and mood (Todres et al, 2006).…”

Section: Discussionmentioning

confidence: 99%

Informal carers’ experiences of caring for someone with Multiple Sclerosis: A photovoice investigation

Topcu

Buchanan

Aubeeluck

et al. 2020

British J Health Psychol

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Objectives. This study explores the lived experiences of carers of people with Multiple Sclerosis (MS), specifically in relation to their quality of life (QoL), through the use of images and narratives, with the aim of gaining a nuanced insight into the complex nature of QoL in the MS caregiving context. Design. Real-time qualitative design using the photovoice method. Methods. Twelve MS carers (aged 30-73 years) took photographs of objects/places/ events that represented enhancement or compromise to their QoL and composed written narratives for each photograph based on their experiences of caregiving. In total, 126 photographs and their corresponding narratives were analysed using content analysis. Results. Seven interrelated themes were identified. MS caregiving-related challenges, sense of loss (e.g., loss of activities), emotional impact (e.g., feeling lonely), urge to escape, and sense of anxiety over the unpredictability of MS carer role were discussed in relation to the negative experiences that compromised their QoL. The themes precious moments (e.g., time spent with loved ones or hobbies) and helpful support (e.g., family and pets) encompassed participants' positive experiences that enhanced their QoL. Conclusions. Findings demonstrated the multi-faceted and complex nature of MS caregiver's QoL and highlighted that although the experiences of MS carers were mostly negative, there were also some positive aspects to caregiving, that helped enhance carers' QoL by ameliorating these negative experiences. These findings can be used to inform support programmes and enhance service provision for MS carers. This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.

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Section: Discussionmentioning

confidence: 99%

Informal carers’ experiences of caring for someone with Multiple Sclerosis: A photovoice investigation

Topcu

Buchanan

Aubeeluck

et al. 2020

British J Health Psychol

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Measuring health related quality of life (HRQoL) in Lysosomal Storage Disorders (LSDs): a rapid scoping review of available tools and domains

McDool,

Powell,

Carlton

2024

Orphanet J Rare Dis

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Background Lysosomal storage diseases (LSDs) are a group of rare inherited metabolic disorders, consisting of over 70 diseases that are characterised by lysosomal dysfunction. Due to their varied and progressive symptoms, LSDs have a continual impact on patients’ health-related quality of life (HRQoL). Several recently published studies have provided insight into the HRQoL of individuals with LSDs. However, it is challenging to meaningfully synthesise this evidence, since studies often focus upon a particular type of LSD and / or utilise different self-report questionnaires or patient-reported outcome measures (PROMs) to assess HRQoL. Aims The aim of this study was to review the published literature in LSDs, to identify the PROMs which have been used to assess HRQoL and generate a conceptual map of HRQoL domains measured in individuals diagnosed with LSDs. Methods Three electronic databases were searched in March 2022. Primary studies of any design which utilised multi-item PROMs to assess at least one aspect of HRQoL in individuals with LSDs since 2017 were identified. Data were extracted to assess both the characteristics of each study and of the PROMs utilised within each study. The extraction of HRQoL domains and synthesis were informed by an a priori framework, inductively modified to reflect data emerging from the identified literature. Selection and extraction was undertaken independently by two reviewers; discrepancies were ratified by a third reviewer. Results Sixty nine studies were identified which were published 2017-2022, with a combined total of 52 PROMs (71 variants) used to assess HRQoL in individuals with LSDs. The final extracted HRQoL framework included 7 domains (Activities; Physical sensations; Autonomy; Cognition; Feelings and emotions; Self-identity; Relationships), characterised by 37 sub-domains. Conclusions This review highlights the breadth and variety of HRQoL domains assessed in individuals with LSDs, across three broad domains of physical, psychological and social functioning. The resultant framework and mapped PROMs will aid researchers and clinicians in the selection of PROMs to assess aspects of HRQoL in people living with LSDs, based on their conceptual coverage.

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Preliminary development of proxy-rated quality-of-life scales for children and adults with Niemann-Pick type C

Cited by 2 publications

References 25 publications

Informal carers’ experiences of caring for someone with Multiple Sclerosis: A photovoice investigation

Informal carers’ experiences of caring for someone with Multiple Sclerosis: A photovoice investigation

Measuring health related quality of life (HRQoL) in Lysosomal Storage Disorders (LSDs): a rapid scoping review of available tools and domains

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