Date Presented 04/04/19
This qualitative research examined the experiences of families living with FXS through an occupation-centered lens. We investigated family occupations via their own voice, through semistructured, recorded interviews. By analyzing occupations, including routines, behaviors, thoughts, interactions, and contexts, OTs can create individualized interventions and promote further research. Results are presented in a conceptual model: Occupations of FXS: Dynamic Interactions of Nested Contexts.
Primary Author and Speaker: Laura Hess
Additional Authors and Speakers: Katelyn Harden, Raechel Jacala, Amanda Lee, Holly Snyder
PURPOSE: Individuals living with FXS experience difficulties across the lifespan with social interaction, communication, and sensory processing abilities (Brady et al., 2006). Given this complex presentation, children with FXS often receive specialized services, such as occupational, physical, and speech therapies as well as behavioral interventions and special education. The current literature on FXS is informative and rich in genetic and medical studies, yet, FXS has not been researched from an occupation centered lens. By interviewing parents and listening to their stories, occupational therapists can acknowledge the personal meaning that parents attach to their overall experience and occupations while living with FXS. Therefore this study examined the need for an occupation-centered lens within current FXS research and employed semi-structured interviews and qualitative analysis. The research question was - What are the complexities of daily life, experiences and family meaning associated with various manifestations of FXS?
DESIGN: Participants in this phenomenological study were families of children diagnosed with full mutation FXS, ages two through six years old who may have a co-diagnosis of autism. Enrolled families participated in a larger study conducted at the University of California, Davis Medical Investigation of Neurodevelopmental Disorders (MIND) Institute (Hess et al., 2016) with original funding from HRSA # R40MC22641. This research study accessed that database, specifically the audio recorded interviews of 20 randomly selected families. The study was approved by the Dominican University Institutional Review Board for the Protection of Human Participants - IRB #10532.
METHOD: Qualitative data analysis was conducted using the constant comparison method originated by Corbin and Strauss (1990). Qualitative analysis of interviews coded for common themes, which were continuously refined throughout the data coding process (Dye, Schatz, Rosenberg, & Coleman, 2000). Coding of five interviews, were completed by consensus to 100% agreement by all four of the researchers. Relationships among the codes were schematically mapped out to further understand familial experiences and values.
RESULTS: Qualitative analysis revealed themes and contexts that families reported as impacting their daily occupations. The major contexts included (1) Immediate Family, (2) Schools and Services, and (3) Community and Social Participation. Additional themes also revealed phenotypic characteristics of FXS that impact a child’s engagement in daily contexts and occupations, which were, (1) Communication Barriers, (2) Sensory Issues, and (3) Behavioral Impacts. Our results are presented in a conceptual model entitled, Occupations of FXS: Dynamic Interactions of Nested Contexts.
CONCLUSION: This study emphasized the need to look at children and families with FXS holistically and the importance of moving beyond solely a phenotypic deficit model. The impact of this research is significant for occupational therapy practice as very often, therapists feel limited to treating a set of deficits. The impact of this research is significant to occupational therapy research, as rarely are qualitative or occupation centered methods applied to FXS or other conditions which are often studied solely via a medical model. This research lends a needed occupation based voice to this body of literature.
References
Brady, N., Skinner, D., Roberts, J., & Hennon, E. (2006). Communication in young children with fragile x syndrome: a qualitative study of mothers’ perspectives. American Journal Of Speech-Language Pathology, 15(4), 353-364.
Hess, L. G., Fitzpatrick, S. E., Nguyen, D. V., Chen, Y., Gaul, K. N., Schneider, A., ... & Rivera, S. (2016). A randomized, double-blind, placebo-controlled trial of low-dose sertraline in young children with fragile X syndrome. Journal of Developmental & Behavioral Pediatrics, 37(8), 619-628.
Dye, J.F., Schatz, I.M., Rosenberg, B.A., & Colemon, S.T. (2000). Constant comparison method: A kaleidoscope of data. The qualitative report, 4(1), 1-10.
