Qualitative Perspectives on Stress and Coping from Parents of Children with Marshall-Smith Syndrome Público
Taylor, Mackenzie (Spring 2020)
Abstract
Up to 30 million people living in the United States are affected by a rare disease. Though the specific diseases are rare, the broader impacts of rare disease affect up to 10 percent of the United States population. Marshall-Smith Syndrome (MSS) is one rare disease that is infrequently described in the literature. The people with MSS require lifelong care which often makes their parents long-term caregivers. Previous research has demonstrated some of the challenges of caregiving for children with medically complex and rare diseases, but no research has specifically focused on stress and coping for parents of children with MSS. The purpose of this study was to understand more about the experiences of parents of children with MSS, the significance of being a part of a rare disease community, and major stressors and coping strategies. Guided by the Transactional Model of Stress and Coping (TMSC), qualitative data were collected from nine parents of children with MSS (from eight families) through individual semi-structured interviews in August and September of 2019. The parents were recruited from a Facebook support group for MSS families and the interviews focused on the usefulness of online social support for this population. Inductive and deductive codes were used for thematic analysis of the data and three key themes emerged: 1) Fear of the unknown, 2) Social connection, and 3) Information gathering. These themes illuminated experiences of stress and coping for parents of children with MSS and the ways that online social support impacts the rare disease community. The findings fit within the previous literature on rare diseases and inform the importance of disease-specific social support and care. Understand more about the experiences of disease specific communities can inform practice of genetic counseling and online interventions for families with rare disease.
Table of Contents
Chapter 1: Introduction 1
Background 1
Problem 3
Purpose 3
Research Questions 4
Rationale 4
Theoretical Framework 4
Chapter 2: Literature Review 6
Significance of Rare Disease 6
History of Marshall-Smith Syndrome 7
Genetics information on Marshall-Smith Syndrome 10
Development and Behavior of Marshall-Smith Syndrome 11
Information of Care for Individuals with Marshall-Smith Syndrome 12
Parent Caregivers of Children with Chronic Disease 13
Caring for a Child with a Rare Disease 14
Family-Engaged Research and Care 16
Online Social Support 17
The Transactional Model of Stress and Coping 19
Summary of the Current Problem and Study Relevance 23
Chapter 3: Methods 24
Project Conceptualization 24
Reflexivity 24
Research Design 25
Population 25
Stakeholder Engagement 25
Eligibility 26
Recruitment 26
Instrument 28
Data Collection 28
Data Management 29
Data Analysis 30
Chapter 4: Manuscript 32
Abstract 33
Keywords 33
Introduction 34
Theoretical Framework 36
Methods 37
Stakeholder Engagement 38
Recruitment and Eligibility 38
Instrument 39
Data Collection and Management 39
Data Analysis 40
Results 42
Theme 1: Fear of the Unknown 42
Theme 2: Information Gathering 45
Theme 3: Social Connections 48
Relationship Between Stress and Coping 50
Assessment of Model Fit in the Data 52
Discussion 53
Implications for Theory and Practice 54
Strengths and Limitations 55
Future Directions 56
Conclusions 57
References 58
Tables 63
Figures 64
Chapter 5: Public Health Implications 65
Implications for Theory 66
Implications for Practice 67
Strengths and Limitations 68
Future Directions 69
Conclusions 70
References 71
Appendix A: Interview Guide 76
Appendix B: Codebook 79
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