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The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis / Sean Aspinall

Swansea University Author: Sean Aspinall

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DOI (Published version): 10.23889/SUthesis.62705

Abstract

Cystic fibrosis (CF) is a lethal autosomal recessive disease caused by mutation in the cystic fibrosis transmembrane conductance regulator (CFTR), resulting in a build-up of viscous mucus and multi-organ dysfunction. Advances in treatment have decreased the burden of CF and increased survival age. H...

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Published: Swansea 2023
Institution: Swansea University
Degree level: Doctoral
Degree name: Ph.D
Supervisor: McNarry, Melitta ; Mackintosh, Kelly
URI: https://cronfa.swan.ac.uk/Record/cronfa62705
first_indexed 2023-02-21T12:54:49Z
last_indexed 2023-02-22T04:16:21Z
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spelling 2023-02-21T13:08:49.8125488 v2 62705 2023-02-21 The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis ca10b63acd93e4ef1daa85566a37ddbc Sean Aspinall Sean Aspinall true false 2023-02-21 Cystic fibrosis (CF) is a lethal autosomal recessive disease caused by mutation in the cystic fibrosis transmembrane conductance regulator (CFTR), resulting in a build-up of viscous mucus and multi-organ dysfunction. Advances in treatment have decreased the burden of CF and increased survival age. However, clinicians and researchers must continue to look for areas that contribute to improved quantity and quality of life for individuals with CF. The aim of this thesis was to explore the effects of nutritional status, aerobic capacity, and modulator therapy on clinical outcomes within CF patients. Study 1 demonstrated the minimal effective dose of exercise needed to elicit change in aerobic capacity. Safety and efficacy of this protocol was evidenced with increases in aerobic capacity akin to that seen in the general population. Study 2 observed the longitudinal effect nutritional status had on pulmonary function. Female pulmonary function evidenced an increased sensitivity to change in body mass index when compared to males. Study 3 evaluated the lived experience of CFTR treatment, highlighting despite clinically relevant benefits, benefits do not come in the absence of negative physical and psychological challenges. Study 4 demonstrated increased incidence of overweight/obesity following implementation of CFTR treatment Introduction of CFTR treatment has rapidly changed the outlook of CF care. As landscape of the disease changes there is a need to adapt to new individual needs in what is a familiar disease with a new face. Whilst diet and exercise represented significant cornerstones of care in the past 30 years, as disease burden decreases, there is a need to view diet and exercise as means of enhancing physical and psychological health, rather than tools to manage the symptoms of the disease. Whilst this thesis is unable to quantify the effect of CFTR therapy on health, the future has never looked brighter for CF patients. E-Thesis Swansea Cystic Fibrosis, modulator therapy, qualitative analysis, exercise physiology 16 2 2023 2023-02-16 10.23889/SUthesis.62705 COLLEGE NANME COLLEGE CODE Swansea University McNarry, Melitta ; Mackintosh, Kelly Doctoral Ph.D KESS II 2023-02-21T13:08:49.8125488 2023-02-21T11:58:02.4515815 Faculty of Science and Engineering School of Engineering and Applied Sciences - Sport and Exercise Sciences Sean Aspinall 1 62705__26647__758d5bdafab0483c968ee6907838345b.pdf Aspinall_Sean_P_PhD_Thesis_Final_Redacted_Signature.pdf 2023-02-21T13:07:09.7218663 Output 7782054 application/pdf E-Thesis – open access true Copyright: The author, Sean P. Aspinall, 2023. true eng
title The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
spellingShingle The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
Sean Aspinall
title_short The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
title_full The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
title_fullStr The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
title_full_unstemmed The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
title_sort The effect of therapeutic and non-therapeutic interventions on the health trajectory of individuals with cystic fibrosis
author_id_str_mv ca10b63acd93e4ef1daa85566a37ddbc
author_id_fullname_str_mv ca10b63acd93e4ef1daa85566a37ddbc_***_Sean Aspinall
author Sean Aspinall
author2 Sean Aspinall
format E-Thesis
publishDate 2023
institution Swansea University
doi_str_mv 10.23889/SUthesis.62705
college_str Faculty of Science and Engineering
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hierarchy_top_id facultyofscienceandengineering
hierarchy_top_title Faculty of Science and Engineering
hierarchy_parent_id facultyofscienceandengineering
hierarchy_parent_title Faculty of Science and Engineering
department_str School of Engineering and Applied Sciences - Sport and Exercise Sciences{{{_:::_}}}Faculty of Science and Engineering{{{_:::_}}}School of Engineering and Applied Sciences - Sport and Exercise Sciences
document_store_str 1
active_str 0
description Cystic fibrosis (CF) is a lethal autosomal recessive disease caused by mutation in the cystic fibrosis transmembrane conductance regulator (CFTR), resulting in a build-up of viscous mucus and multi-organ dysfunction. Advances in treatment have decreased the burden of CF and increased survival age. However, clinicians and researchers must continue to look for areas that contribute to improved quantity and quality of life for individuals with CF. The aim of this thesis was to explore the effects of nutritional status, aerobic capacity, and modulator therapy on clinical outcomes within CF patients. Study 1 demonstrated the minimal effective dose of exercise needed to elicit change in aerobic capacity. Safety and efficacy of this protocol was evidenced with increases in aerobic capacity akin to that seen in the general population. Study 2 observed the longitudinal effect nutritional status had on pulmonary function. Female pulmonary function evidenced an increased sensitivity to change in body mass index when compared to males. Study 3 evaluated the lived experience of CFTR treatment, highlighting despite clinically relevant benefits, benefits do not come in the absence of negative physical and psychological challenges. Study 4 demonstrated increased incidence of overweight/obesity following implementation of CFTR treatment Introduction of CFTR treatment has rapidly changed the outlook of CF care. As landscape of the disease changes there is a need to adapt to new individual needs in what is a familiar disease with a new face. Whilst diet and exercise represented significant cornerstones of care in the past 30 years, as disease burden decreases, there is a need to view diet and exercise as means of enhancing physical and psychological health, rather than tools to manage the symptoms of the disease. Whilst this thesis is unable to quantify the effect of CFTR therapy on health, the future has never looked brighter for CF patients.
published_date 2023-02-16T08:19:25Z
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score 11.04748

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