CHARACTERIZING PLAY SKILLS AND IDENTIFYING THEIR ASSOCIATION WITH EXPRESSIVE AND RECEPTIVE LANGUAGE SKILLS IN YOUNG BOYS WITH FRAGILE X SYNDROME A Thesis Presented to the faculty of the Department of Speech Pathology and Audiology California State University, Sacramento Submitted in partial satisfaction of the requirements for the degree of MASTER OF SCIENCE in Speech Pathology and Audiology by Alex Stewart FALL 2013 CHARACTERIZING PLAY SKILLS AND IDENTIFYING THEIR ASSOCIATION WITH EXPRESSIVE AND RECEPTIVE LANGUAGE SKILLS IN YOUNG BOYS WITH FRAGILE X SYNDROME A Thesis by Alex Stewart Approved by: __________________________________, Committee Chair Robert A. Pieretti, Ph.D. __________________________________, Second Reader Ann Blanton, Ph.D. ____________________________ Date ii Student: Alex Stewart I certify that this student has met the requirements for format contained in the University format manual, and that this thesis is suitable for shelving in the Library and credit is to be awarded for the thesis. _______________________________ ___________________ Ann Blanton, Ph.D. Date Department of Speech Pathology and Audiology iii Abstract of CHARACTERIZING PLAY SKILLS AND IDENTIFYING THEIR ASSOCIATION WITH EXPRESSIVE AND RECEPTIVE LANGUAGE SKILLS IN YOUNG BOYS WITH FRAGILE X SYNDROME by Alex Stewart Little is known about the play skills of children with FXS and the relationship of play and language acquisition in this population. There are no published studies examining play skills in young males with FXS, even though play skills have been shown to be directly related to the development of receptive and expressive language. Research that attempts to identify more efficient and naturalistic language intervention approaches for children with FXS is needed. This study aimed to provide a descriptive characterization of play for 11 boys with FXS between the ages of 27 and 51 months. This study was designed to answer the following research questions: (1) What are the concurrent correlations between play skills, receptive and expressive language, and cognitive abilities of young males affected by fragile X syndrome? (2) How can the play of young males affected by fragile X syndrome be described and characterized? A series of one-tailed Spearman rank-order correlation (rho) was performed to provide quantitative data to answer the research questions. A significant correlation was iv found between the Mullen Visual Reception raw scores and frequency of Discriminative Play Actions (r = .667, p = .025, one-tailed) and a marginally significant correlation was found between the Mullen Visual Reception raw scores and frequency of Total Play Actions (r= .588, p= .057). The correlations with the Mullen Receptive Language raw scores were hovering for Object Interest (r= .529, p= .094), frequency of Discriminative Play Actions (r= .557, p= .075), and frequency of Total Play Actions (r= .538, p= .088). There was also a hovering negative correlation between the ADOS Autism Severity Score and Diversity of Play (r= -.559, p= .093). It should also be noted that no correlations were found between duration of the play sample and any of the play variables. Using item analysis of the PLS-5 and level of mastery results from the DPA, a language and play development chart (see Table 4-1) was created to describe and characterize the play of young boys with FXS ages 1-5. The information from this study will provide clinicians with information specific to the developmental trajectory of young boys with FXS, which may also provide insight into both assessment and therapy standards. This should improve the overall service delivery to these clients. ____________________________, Committee Chair Robert A. Pieretti, Ph.D. ____________________________ Date v ACKNOWLEDGEMENTS The research included in this thesis could not have been performed if not for the assistance, patience, and support of many individuals. I would like to extend my gratitude first and foremost to my thesis advisor, Dr. Robert Pieretti, for mentoring me over the course of my graduate studies. His insight led to a more refined and descriptive research objectives. His perspective has helped me to thoroughly express my results and observations to allow for better understanding for my readers. I sincerely thank him for his confidence in me and his help over the course of the analysis and the writing of this thesis. I would additionally like to thank Dr. Ann Blanton for the support she has provided me throughout my undergraduate and graduate studies. Her background in research has only benefitted me, especially throughout the revision stages of this paper. I cannot thank her enough for the example she has set for me as both a clinician and a researcher. This study would not have been possible without the help, guidance, and support of Dr. Leonard Abbeduto, Dr. Andrea McDuffie, Dr. Angela John Thurman, and Ashley Oakes. To Dr. Abbeduto and Dr. Andrea McDuffie, I cannot thank you enough for providing me with the wonderful opportunity to participate in such an innovative research study. Your wisdom and guidance helped me throughout the course of this study, as well as during the writing process. Dr. Angela John Thurman, I want to thank you for your constant support and insight into the research field, including your assistance during the data analysis and interpretation process. To Ashley Oakes, you led by example and taught vi me to not be afraid of the research process, but rather enjoy the ride that it takes you on. And to my all my coworkers at the UC Davis M.I.N.D. Institute, it was a pleasure collaborating with you all throughout the course of this study. I am forever grateful for this experience. This research would not have been possible without the cooperation and participation of the children and families involved in this study. I cannot express how much I appreciate the knowledge you have given me and the help you have provided for my research. The larger study, in which this pilot study was associated with, was supported by funding from the National Fragile X Foundation. I would like to thank this wonderful association for the support they provide to the families of those affected by fragile X syndrome and their involvement in innovative research that will further support these families. Finally I would like to extend my deepest gratitude to my parents, Teri and David Hinz, and my husband, Scott Stewart, without whose love, support and understanding I could never have completed this master’s degree. To my mom and dad, you have constantly shown me that hard work and dedication pay off. To my loving husband, you have given me confidence when I needed it, help when I asked for it, and stability though I never requested it. I could not have made it through this process without you all. vii TABLE OF CONTENTS Page Acknowledgements ..................................................................................................... vi List of Tables ................................................................................................................ x Chapter 1. REVIEW OF THE LITERATURE ....................................................................... 1 Introduction ....................................................................................................... 1 Causes of Fragile X Syndrome ............................................................. 1 Prevalence of FXS ................................................................................ 2 Cognitive and Developmental Characteristics of Individuals with FXS ................................................................................................3 Physical Characteristics of Individuals with FXS ................................ 4 Language Development in Individuals with FXS ................................. 5 The Relationship Between Play and Language Development .............. 8 Play and Typically-Developing Children ............................................. 9 Play and Children with Developmental Delays .................................. 15 Statement of the Problem ................................................................................ 16 2. METHODOLOGY ............................................................................................... 18 Design. ............................................................................................................ 18 Participants ...................................................................................................... 18 Evaluation Measures ....................................................................................... 20 Variables Analyzed ......................................................................................... 26 viii 3. RESULTS ............................................................................................................. 28 Individual Characteristics: Developmental Play Assessment ......................... 28 Individual Characteristics: Measures of Language, Cognition, and Autism Symptom Severity ........................................................................................... 31 Concurrent Correlational Analysis ................................................................. 37 4. DISCUSSION ....................................................................................................... 41 5. IMPLICATIONS, LIMITATIONS, AND FUTURE DIRECTIONS .................. 49 Implications....................................................................................................