2026-09-25T09:13:23Zhttps://bura.brunel.ac.uk/oai/request

oai:bura.brunel.ac.uk:2438/235052021-11-14T03:03:24Zcom_2438_58col_2438_3672

Norris, P De Souza, L Elyoussfi, Samia 2021-11-13T18:44:00Z 2021-11-13T18:44:00Z 2021 http://bura.brunel.ac.uk/handle/2438/23505 This thesis was submitted for the degree of Doctor of Philosophy and awarded by Brunel University Individuals with rare genetic endocrine syndromes, such as von-Hippel Lindau, Multiple Endocrine Neoplasia and mutations in the succinate dehydrogenase complex genes can develop diverse and unpredictable new tumour formations. Regular screening is recommended as best practice for early detection and management of tumours in those deemed at risk. However, little is known about screening attendance in gene carriers and the experiences of screening which may influence attendance. The aim of this thesis was to address this gap by gaining a deeper understanding of how individuals carrying genes for rare endocrine tumour syndromes comprehend and use health services generally and, in particular, the service provided by the Barts endocrine screening clinics. This was a multimethod three staged study in the context of a national specialist clinic. Study 1 was a retrospective cohort study to determine screening attendance rates over a three-year period and examine the relationship between patient demographic characteristics and attendance. Study 2 examined the experience of attending appointments through in-depth interviews with 12 participants. Study 3 was designed to enhance engagement with initial screening through the co-production of an information resource using focus groups. Integration of qualitative findings explored the relationship between the themes. Study 1 identified an 83.27% attendance rate. Attendance showed no significant association with patient demographic characteristics. Study 2 interviews illuminated how the complexities of living with an incurable diagnosis interacted with the anticipation and attendance at screening. Study 3 resulted in a simple leaflet with signposting, noting the importance of reassurance and availability of family clinics. Issues regarding data quality and recruitment were encountered. These studies contribute to understanding of and engagement with patients with rare syndromes and screening. Considerations of data collection, life-course, diagnosis familiarity, family dynamics, practical accessibility and navigating the system are specific and novel insights that should inform future service delivery. Submitted by SATYESH SHAH (satyesh.shah@brunel.ac.uk) on 2021-11-13T18:43:25Z No. of bitstreams: 1 FulltextThesis.pdf: 20567216 bytes, checksum: 3ce2906a184508dcb41520b815b494f6 (MD5) Approved for entry into archive by SATYESH SHAH (satyesh.shah@brunel.ac.uk) on 2021-11-13T18:43:59Z (GMT) No. of bitstreams: 1 FulltextThesis.pdf: 20567216 bytes, checksum: 3ce2906a184508dcb41520b815b494f6 (MD5) Made available in DSpace on 2021-11-13T18:44:00Z (GMT). No. of bitstreams: 1 FulltextThesis.pdf: 20567216 bytes, checksum: 3ce2906a184508dcb41520b815b494f6 (MD5) Previous issue date: 2021 Barts Charity en Brunel University London http://bura.brunel.ac.uk/handle/2438/23505 Multimethod research Retrospective cohort study Qualitative Patient experience Co-produced information resource Understanding patient engagement: exploring factors around utilisation of screening and surveillance services in individuals with or at risk of rare endocrine syndromes Understanding screening attendance in rare endocrine syndromes Thesis