Institute of Neurology University of London Respiratory Therapy for Speech in Multiple Sclerosis Ph.D. Thesis Kimberley Anne Mathieson Bradley April 1997 ProQuest Number: 10055444 All rights reserved INFORMATION TO ALL USERS The quality of this reproduction is dependent upon the quality of the copy submitted. In the unlikely event that the author did not send a complete manuscript and there are missing pages, these will be noted. Also, if material had to be removed, a note will indicate the deletion. uest. ProQuest 10055444 Published by ProQuest LLC(2016). Copyright of the Dissertation is held by the Author. All rights reserved. This work is protected against unauthorized copying under Title 17, United States Code. Microform Edition © ProQuest LLC. ProQuest LLC 789 East Eisenhower Parkway P.O. Box 1346 Ann Arbor, Ml 48106-1346 ABSTRACT Respiratory Support for Speech In Multiple Sclerosis The dysarthria of multiple sclerosis is known to worsen as the disease progresses (Darley, Brown and Goldstein 1972). Thus as activities of daily life and opportunities for activity for the person with multiple sclerosis are curtailed by increasing disability, the capacity of their respiratory system is also diminished both by disease and lack of demand on the system (Olgiati, Hofstetter and Bailey 1988). It is a hypothesis of this paper and others that disuse creates a discrepancy between the functional ability that is neurologically available and that which is characteristically used (Olgiati et al., 1988; Olgiati et al. 1986). It is this functional overlay that may be the target of speech therapy (Farmakides and Boone 1960). Five subjects with MS and dysarthria affecting intelligibility were involved in a multiple baseline therapy study to establish the efficacy of respiratory exercises in improving functional speech performance. Intervention effects were demonstrated by introducing the therapy to different subjects at successive points in time. Therapy exercises targeted the respiratory system alone with no phonatory or articulatory components. Various measures including laryngographic analysis, clinical motor speech tasks and standardised dysarthria profiles were found to be unsuitable as baseline or repeat measures to show improvement in speech. Intelligibility was chosen as a global and objective repeat measure of functional speech performance and was established for each subject using the Yorkston Beukelman Test of the Intelligibility of Dysarthric Speech. Findings cautiously suggest that certain subjects can benefit from respiratory muscle exercises that improve speech performance as measured by intelligibility. A component of the dysarthria of MS may not be due to neuro-motor dysfunction but to atrophy based on fatigue and disuse of the system. This study suggests that this atrophy may be reversible. The objective measurement of intelligibility and the clinical use of a multiple baseline research format are also discussed. THE N/ for Paul, with thanks ACKNOWLEDGEMENTS To the five subjects in this study who gave unstintingly of their time, enthusiasm and concern I am greatly indebted. As well to the members of the North Surrey Group of the MS Society of Great Britain who welcomed me into their homes and let me tape their voices I am most grateful. I thank Dr. Keith Andrews of the Royal Hospital for Neuro-Disability, Putney and the members of the Speech Therapy Department at RHND in 1992, Julie Lepatourel, Joan Ellams, and Fiona Sugden for their assistance, suggestions and office space. I thank Sherwood Medical Supplies, Great Britain for supplying the Voldyne Inspiratory Spirometers used in the study and Sandra Robertson and Vincent McDonnell of the Speech Pathology Department of Manchester Metropolitan University for allowing me the use of the Respiratory Drive Indicator. Drs. Maggie Snowling, Frances McCurtain and Rosemary Morgan-Berry at the (then) National Hospitals College of Speech Sciences I thank for their input in the early stages of this project. I am grateful to Dr. Bill Wells of the Department of Human Communication Science, University College for his input into Round Two. To Dr. Jane Maxim, also of UCL, I am especially grateful for seeing the project to completion. Drs. Jamie Mathieson and John Lloyd of the Curry School of Education, University of Virginia I thank for their conflicting but helpful guide through the world of statistics and single case studies. I thank my parents who have always believed in me whatever I have attempted. I thank Charlotte and Georgia for being such sweethearts about letting Mum do her “fesis”. And finally I owe to my husband, Paul, infinite thanks for the emotional, financial, personal and practical support that he has always given me. I would not have done this without him. TABLE OF CONTENTS Page Abstract 2 Dedication 3 Acknowledgements 4 Table of Contents 5 Table of Figures 7 1 BACKGROUND TO THE RESEARCH QUESTION 9 1.1 Incidence Of Dysarthria In Multiple Sclerosis 11 1.2 Mixed Dysarthria Of Multiple Sclerosis As Distinct From 14 Paroxysmal Dysarthria 1.3 Language And Other Communication Problems In Multiple 17 Sclerosis 1.4 Acoustic Analysis Of Speech 23 1.5 Perceptual Analysis Of Speech 29 1.6 Treatment Of The Dysarthria In Multiple Sclerosis 36 1.7 Respiratory Function In Multiple Sclerosis 41 1.8 Respiratory Therapy In Multiple Sclerosis 48 1.9 Conclusion 49 2 METHODOLOGY 51 2.1 The Research Design 51 2.2 Subjects 52 2.2.1 Subjects - General 52 2.2.2 Subjects - Dysarthria 56 2.3 Research Procedure 60 2.3.2 Therapy 64 2.3.3 Judging 66 3 BACKGROUND TO THE METHODOLOGY 70 3.1 Case Study Methodology 70 3.2 Choice Of Repeat Measure 75 3.2.1 The Potential Of The Laryngograph As A Repeat Measure 79 3.2.2 Intelligibility Testing As A Repeat Measure 106 3.3 Respiration And Speech Therapy 124 3.4 Variables Controlled For In The Study 135 3.4.1 Diurnal Effects 135 3.4.2 Cyclical Effects 139 3.4.3 Location Of Session 140 3.4.4 Posture 142 3.5 Procedural Reliability 142 4 RESULTS 146 4.1 Multiple Baseline Study 146 4.2 Individual Responses To Therapy 148 4.3 Statistical Analysis 160 4.4 Reliability Testing 167 4.5 Error Analysis 172 4.6 Therapy Results 184 5 DISCUSSION 189 5.1 Statistical Analysis 189 5.2 Conclusions — The Research Question 194 5.3 Conclusions — Case Study Methodology 209 5.4 Conclusions — Measurement Of Intelligibility 215 6 RECOMMENDATIONS FOR FURTHER RESEARCH 220 7 BIBLIOGRAPHY 225 APPENDICES 243 A Upper And Lower Extremity Function Scales 244 B Dysarthria And Case History Assessment Form 246 C Standardised Reading Passages 253 D Multiple Baseline Therapy Results 255 E Consent Form 259 F Procedural Reliability Protocols 261 G Forms For The Yorkston Beukelman Assessment Of The 269 Intelligibility Of Dysarthric Speech H Diary Sheets 274 I Error Analysis Subjects VG And GF 278 J Subject Frenchay Dysarthria Assessment Profiles 299 TABLE OF FIGURES Figure 1 : Specific Speech Deviations Noted in a Group of Subjects with Multiple Sclerosis 31 Figure 2: Distribution of Speech, Breathing and Oral Diadochokinetic Rate Measures in Six Neurologic Groups of Subjects with Multiple Sclerosis* 33 Figure 3: Subject Details 55 Figure 4: Intelligibility Testing: Subjects, Settings and Procedures 63 Figure 5: Therapy Goals for Subjects 66 Figure 6: Normal Glottic Cycle 79 Figure 7: Laryngograph Study Participants Profile 82 Figure 8: Hillel’s Ranking of Speech in ALS (Hillel et al. 1989) 83 Figure 9: Laryngograph Study: Subjects’ Intelligibility Scores and Awareness of Speech Problem. 89 Figure 10: Laryngograph Study: Spontaneous Speaking Rate 91 Figure 11 : Laryngograph Study: Motor Speech Tasks Targeting Respiration 92 Figure 12: Laryngograph Study: Motor Speech Tasks Targeting Phonation 93 Figure 13: Laryngograph Study: Articulation Task - Diadochokinesis Data 95 Figure 14: Laryngograph Study: Articulation Tasks: Diadochokinesis 95 Figure 15: Laryngograph Study: L(jc) Traces 97 Figure 16: Laryngograph Study: Subject’s Report of Dysphagia 98 Figure 17: Laryngograph Study: Motor Speech Tasks Targeting Resonance and Multiple Levels in Speech Process 100 Figure 18: Laryngograph Study: Mean Results by Group 102 Figure 19: Laryngograph Study: ANOVA Group Difference Results 103 Figure 20: Intelligibility plotted against Sound Pressure Level 111 Figure 21 : Sherwood Voldyne 5000 Spirometer 134 Figure 22: Subjects and Control Times 138 Figure 23: Subject Session Location 141 Figure 24: Subject Posture 142 Figure 25: Multiple Baseline Study - The Effect of Respiratory Exercises on Speech Intelligibility as Measured by the YB Intelligibility Test 147 Figure 26: Intelligibility Scores by Week, Date and Session 149 Figure 27: VG Performance on Motor Speech Tasks - Summary 151 Figure 28: VG Intelligibility Scores, Repeated Listening 152 Figure 29: VG Intelligibility Scores 152 Figure 30: HP’s Performance on Motor Speech Tasks - Summary 154 Figure 31: RP Intelligibility Scores 154 Figure 32:GF’s Performance on Motor Speech Tasks - Summary 156 Figure 33: GF Intelligibility Scores 156 Figure 34: GF Intelligibility Scores 158 Figure 35: WR Intelligibility Scores 159 Figure 36: EB Intelligibility Scores 160 Figure 37: VG t-Test Statistics 162 Figure 38: GF t-Test Statistics 163 Figure 39: Mann Test Calculations for Subject GF 163 Figure 40: Summary of C Statistic Calculations for Subject VG 165 Figure 41 : Summary of C Statistic Calculations for Subject RP 165 Figure 42: Summary of C Statistic Calculations for Subject GF 166 Figure 43: Summary of Z Scores Results for Tryon’s C Statistic 166 Figure 44: Intrajudge