DSpace Welsh Repository Network Partner WRN Partners
  
 

Glamorgan DSpace >
University of Glamorgan >
Theses >
PhD theses from the University of Glamorgan >

Please use this identifier to cite or link to this item: http://hdl.handle.net/10265/563

Title: Identifying the benefits and disbenefits of clinical genetics services : a framework for economic evaluation
Authors: Pithara, Christalla
Keywords: Medical genetics
Issue Date: 16-May-2012
Citation: Pithara, C. (2011)'Identifying the benefits and disbenefits of clinical genetics services : a framework for economic evaluation'. Unpublished Ph.D. thesis. University of Glamorgan.
Abstract: A number of methodological considerations have been discussed in the area of economic evaluation of Clinical Genetic Services (CGSs) including the limited knowledge of psychosocial consequences of these services. This study aims to address this gap by identifying tangible and intangible benefits and disbenefits of CGSs and presenting these within a framework to assist in the design of a comprehensive welfarist economic evaluation. Mixed methods of data collection were adopted and a UK medical genetics service was used as a case study. Face-to-face interviews with genetic service providers were undertaken in Phase One to explore patient pathways and the perceived role of the service. Focus groups and face-to-face interviews with service users explored the perceived benefits and disbenefits of the CGS in Phase Two. Phase Three comprised a pilot study of using Audience Response Systems (ARSs) for exploring stakeholder preferences and tackled issues of respondent validation and transferability. Both process-related attributes and psychosocial outcomes emerged as utility-bearing for service users. Patient pathways i.e. patient experience, were found to be influenced by factors associated with the genetic condition and with individual patient/family characteristics and needs. The overall (dis)benefits of the service however were found to be common across conditions. The concept of Perceived Familial Control is proposed as a suitable outcome which encompasses the psychosocial dimension of CGSs. This study has demonstrated the use of qualitative methods in the context of health economics and economic evaluation. It has specifically demonstrated the use of various levels of qualitative analysis for obtaining attributes and outcomes of CGSs and has incorporated these within a framework directed towards the design of a welfarist economic evaluation. The use of ARSs was also tested for their usefulness as a method of establishing preferences and exploring the opinions of CGS stakeholders. Further research is required to establish whether the emerging (dis)benefits represent the experiences of users of other UK clinical genetic centres. Subsequently, user preferences for the identified (dis)benefits could be explored as a step towards the design of a welfarist economic evaluation. Further research is also required to develop the concept of Perceived Familial Control into an appropriate outcome measure for CGSs.
URI: http://hdl.handle.net/10265/563
Appears in Collections:PhD theses from the University of Glamorgan

Files in This Item:

File Description SizeFormat
THESIS-FINAL_BINDED.pdf6.61 MBAdobe PDFView/Open


Items in DSpace are protected by copyright, with all rights reserved, unless otherwise indicated.

 

Valid XHTML 1.0! DSpace Software Copyright © 2002-2010  Duraspace - Feedback