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©Copyright 2017 Mallory Dawson ©Copyright 2017 Mallory Dawson Do Perceptual Subgroups Exist in Ataxic Dysarthria? Mallory Dawson A thesis submitted in partial fulfillment of the requirements for the degree of Master of Science University of Washington 2017 Committee: Kristie Spencer, Chair Edythe Strand Kathryn M. Yorkston Program Authorized to Offer Degree: Speech and Hearing Sciences 2 University of Washington Abstract Do Perceptual Subgroups Exist in Ataxic Dysarthria? Mallory Dawson Chair of the Supervisory Committee: Associate Professor Kristie Spencer Speech and Hearing Sciences When the cerebellar circuit is damaged, the resulting speech disturbance is known as ataxic dysarthria. Features of this disorder include irregular articulatory breakdowns, distorted vowels, excess and equal stress, prolonged phonemes, and abnormal pitch/loudness variation. However, heterogeneity within this disorder has been documented by clinicians and researchers for decades, suggesting that subgroups of ataxic dysarthria may exist. The purpose of this study was to extend this line of research to determine a feasible, reliable perceptual method for identifying and differentiating these proposed subgroups. Two hypotheses were tested based on existing theoretical frameworks: (1) speakers be classified into instability/inflexibility (or mixed presentation) subgroups, and/or (2) speakers be classified into subgroups based on differential speech subsystems involvement. Four dysarthria experts listened to speech samples of adults with ataxic dysarthria and completed rating forms of the speech features. Results suggested that five speakers fit the pattern of instability, two speakers aligned with inflexibility, and one speaker had a mixed presentation. Patterns did not emerge according to differential subsystem involvement. The emergence of subgroups has implications for clinical and research practice; additional research is warranted. 3 Table of Contents Abstract……………………………………………………………………………………………3 List of Figures……………………………………………………………………………………..5 List of Tables……………………………………………………………………………………...6 Introduction………………………………………………………………………………….…….8 Clusters of deviant speech characteristics (subsystem hypothesis)………………………10 Instability/inflexibility hypothesis……………………………………………………….12 Testing the instability/inflexibility hypothesis: A pilot study……………………………16 Rationale for current study……………………………………………………………….20 Methods…………………………………………………………………………………………..23 Participants……………………………………………………………………………….23 Tasks……………………………………………………………………………………..25 Perceptual ratings of habitual speech tasks ………………………………………………26 Measuring response to instruction………………………………………………………..31 Analysis…………………………………………………………………………..………………31 Reliability……………………………………………………………………...…………32 Results……………………………………………………………………………………………32 Reliability………………………………………………………………………………...32 Intra-rater reliability – VAS ratings………………………………………………32 Intra-rater reliability – Mayo scale ratings……………………………………….34 Inter-rater reliability – VAS ratings………………………………………………35 Inter-rater reliability – Mayo scale ratings……………………………………….36 Testing the hypotheses…………………………………………………………………...37 Instability/Inflexibility Hypothesis: VAS ratings………………………………..37 Differential Subsystem Hypothesis (Mayo scale ratings)………………………..41 Measuring Response to Instruction………………………………………………………43 Comparing standard, slow, and fast DDKs………………………………………43 Perceptual rating of clear speech monologue……………………………………..43 Discussion………………………………………………………………………………………..44 Instability/Inflexibility hypothesis (Visual Analog Scale ratings)……………………….45 Subsystem hypothesis (Mayo Clinic Scale ratings)………………………………………49 Response to instruction…………………………………………………………………..50 Clinical Applications……………………………………………………………………..51 Limitations and Future Directions………………………………………………………..53 References………………………………………………………………………………………..56 Appendix A: Participant Protocol……..…………………………………………………………59 Appendix A: Intake Form…………..……………………………………………………………61 Appendix C: Audio file order of presentation for each expert rater………………………..……62 Appendix D: Elaboration of differences in perceptual ratings between the two connected speech conditions…………………………………………………...……………………………………63 4 List of Figures Figure 1: Rating sheet for perceptual features per speech task with color coding to reflect hypothetical subgroups of Instability [red] and Inflexibility [green] (France, 2014)……………18 Figure 2: Sample of speakers who fit criteria for the Instability subgroup [n=5]………………..19 Figure 3: Speaker fitting the criteria for the Inflexibility profile [n=1]…………………………..19 Figure 4: Sample of speakers who fit criteria for the Mixed subgroup [n=4]……………………..19 Figure 5: Perceptual rating form specific to the diadochokinetic task (AMRs/SMRs)………….28 Figure 6: Perceptual ratings specific to sustained phonation task……………………………….29 Figure 7: Perceptual ratings specific to connected speech task………………………………30-31 Figure 8: Percent intra-rater agreement for VAS ratings across speaking tasks per individual raters (R1 – R4)…………………………………………………………………………………..33 Figure 9: Percent intra-rater agreement for Mayo scale ratings across individual raters, collapsed across speaking tasks………………………………………………………………….35 Figure 10: Percent inter-rater agreement for VAS ratings, by speaking task…………………....35 Figure 11: Percent inter-rater agreement for Mayo scale ratings, by speaking task……………..37 Figure 12: Percent of VAS ratings reflecting instability (<45mm) and inflexibility (>55mm) for each speaker, by speaking task…………………………………….......……………………..38 Figures 13-17: Percent of VAS ratings falling on the either side of midline, collapsed across four raters, for speakers fitting the “instability” profile……………………….........………..38-39 Figures 18-19: Percent of VAS ratings falling on the either side of midline, collapsed across four raters, for speakers fitting the “inflexibility” profile…………………………………….…40 Figure 20: Percent of VAS ratings falling on the either side of midline, across four raters, for speakers fitting the “mixed instability/inflexibility” profile…………………………………40 Figure 21: Average severity ratings by speaker for DDK task, according to speech subsystem, based on Mayo scale………………………………………………………………...41 Figure 22: Average Mayo scale ratings by speaker and subsystem, for connected speech……...42 5 List of Tables Table 1: Summary of salient speech features that are consistent with patterns of instability versus inflexibility (France, 2014)……………………………………………………………….15 Table 2: Proposed null hypothesis and three alternate hypotheses, with related interpretations……………………………………………………………………………………23 Table 3: Participant demographic information………………………………………………….24 Table 4: Primary neurologic symptoms reports by each participant…………………………….24 Table 5: Participant screening measures and mean intelligibility scores………………………..25 Table 6: Intraclass correlation coefficients and confidence intervals by rater, for VAS ratings……….................................................................................................................................33 Table 7: Percent intra-rater agreement across tasks and raters, for VAS ratings………………..33 Table 8: Intra-rater agreement by rater across speaking tasks for Mayo scale severity ratings…………………………………………………………………………………………….34 Table 9: Percent intra-rater agreement across tasks and raters, for 5-point Mayo scale ratings…………………………………………………………………………………………….34 Table 10: Inter-rater agreement (Spearman’s rank correlation coefficient) by rater, collapsed across speaking tasks, for Mayo scale severity ratings………………………………..36 Table 11: Perceptual ratings of habitual monologue compared to monologue with “clear speech” instruction……………………………………………………………………………….44 6 Acknowledgments I would like to express my deepest gratitude to my advisor and mentor, Dr. Kristie Spencer, for her endless patience and unwavering support throughout this process. Working on this project for the past 15 months has been my most challenging undertaking, and her expert direction and confidence in my abilities motivated me at times when I felt like giving up. I am incredibly grateful to have had this experience, and I will miss hashing out the details in her office each week. I would like to thank the members of my committee, Dr. Edythe Strand and Dr. Kathryn Yorkston, for their contributions and unique insight. Their guidance pushed me to keep clinical relevance in my mind at all times. I would also like to thank my fourth expert listener, Dr. Robert Miller for graciously donating his time and expertise. I would like to thank the members of the Motor Speech Lab, especially Taylor Ellerbrock, for helping with this research project. I would like to thank the participants, who generously and selflessly allowed me to come into their homes and record their speech. It was a joy to get to know each one of you. Your time and willingness to participate were greatly valued. I would also like to thank my friends and family, for their kindness and encouragement throughout the past year. Thank you for hearing my complaints and worries, and motivating me to push through the late nights. Finally, I would like to thank my parents, for their eternal love and support. My drive to make you proud has pushed me to accomplish more than I ever could have done on my own. 7 Introduction Ataxic dysarthria refers to speech disturbances caused by disorders of the cerebellum. Decades of acoustic and perceptual studies have attempted to describe the features associated with this motor speech disorder, which span across multiple speech subsystems, including
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