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Title: Psychosocial outcomes of bone marrow transplant for individuals affected by MPS I Hurler Disease
Author: Pitt, Cheryl Anne
ISNI:       0000 0004 2683 3625
Awarding Body: Brunel University
Current Institution: Bucks New University
Date of Award: 2009
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RATIONALE: A theoretical model was used to examine the impact of risk and resistance factors on the psychosocial adjustment of children and young people affected by Mucopolysaccharidosis Type I Hurler Disease post-bone marrow transplant. METHOD: A sequential exploratory mixed methods design was employed and the study carried out in two phases. In the initial phase qualitative methods were employed in an in-depth study of a small sample of parents of individuals affected by this condition (n=10). They were administered semi-structured interviews, which were analysed using Interpretative Phenomenological Analysis. The themes highlighted were used to inform the design of the second phase, which employed quantitative methods. In this phase forty-four families affected by MPS IH post-BMT participated (44 mothers, 36 fathers, 44 patient participants). This comprised almost the entire population of this patient group in the UK, whose ages ranged from 16 months to 25 years. A face-to-face survey method was used with the mothers, telephone-survey with the fathers and psychometric testing of the patient participants. The measures included risk factors (e.g. physical, cognitive, and adaptive functioning), resistance factors (intrapersonal, stress processing, and socialecological), and adjustment for both parent and patient participants. RESULTS: Data from the qualitative phase revealed a number of themes, highlighting numerous issues pertinent to parents of children affected by this condition. These included perceptions of child vulnerability, uncertainty, perceptions of stress and sources of support, and feeling that others do not understand their situation. Data from the second phase illustrate how stress processing and social-ecological factors make significant contributions to parent adjustment. They also illustrate how intrapersonal, socialecological, and maternal stress processing factors contribute significantly to patient adjustment. A theoretical path of the determinants of patient psychosocial outcomes was created to illustrate these relationships. CONCLUSION: The findings are indicative of how resistance factors moderate the effects that disease- and disability-related risk factors have on parent and patient adjustment as predicted by the model. The dynamic interplay between disease-related risk factors and intrapersonal, socialecological, and stress processing factors are discussed in relation to parent and patient adjustment outcomes, as are the implications these relationships pose for patient and family support.
Supervisor: Not available Sponsor: Buckinghamshire New University
Qualification Name: Thesis (Ph.D.) Qualification Level: Doctoral
EThOS ID:  DOI: Not available