The Nature of Acquired Dysgraphia: Patterns of Impairment and Rehabilitation Janna Geertruida (Trudy) Krajenbrink A thesis submitted in fulfillment of the requirements for the degree of Doctor of Philosophy ARC Centre of Excellence in Cognition and its Disorders (CCD) Department of Cognitive Science Macquarie University, Sydney, Australia April, 2015 Declaration The work in this thesis is my own original work. It has not been submitted for a higher degree in any other university or institution. All of the work reported in this thesis was undertaken during the time I was enrolled as a PhD student at Macquarie University, under the supervision of Prof Lyndsey Nickels and Dr Saskia Kohnen. Ethics approval for the studies reported in this thesis was obtained from Macquarie University’s Human Research Ethics Committee, Reference No. 5201200905. Signed: Trudy Krajenbrink 10 April 2015 ii Table of Contents Title Page i Declaration ii Table of Contents iii Acknowledgements v General Summary ix GENERAL INTRODUCTION 1 STUDY ONE 17 Graphemic Output Buffer Impairment with Fragment Errors: a Case of Rapid Decay of Activation from the Buffer Abstract 18 Introduction 19 Case Report 27 Writing to Dictation: Comprehensive Analysis 42 Fragment errors 60 Discussion 76 Conclusion 83 References 84 Appendix A 89 Appendix B 90 Appendix C 91 Appendix D 92 Appendix E 93 STUDY TWO 95 Patterns of Sub-Lexical Impairment in Aphasia Abstract 96 Introduction 97 Participants 104 Tests 107 Results 109 Overview of sub-lexical spelling performance: Controls and PWA 109 Detailed analysis of PGC spelling in people with aphasia 116 Effects of context on sub-lexical spelling 125 iii Discussion 129 Conclusion 133 References 134 Appendix A 136 Appendix B 137 STUDY THREE 139 Generalisation after Treatment of Acquired Spelling Impairments: A review Abstract 140 Introduction 141 Generalisation Effects: a Review of Treatment Studies 152 Discussion 191 Future Directions 197 Conclusion 199 References 201 STUDY FOUR 211 The Effect of Orthographic Neighbourhood on Treatment of Acquired Dysgraphia Abstract 212 Introduction 213 Case Reports 222 Treatment Program 230 Results 237 Discussion 250 Conclusion 258 Acknowledgements 260 References 261 Appendix A 267 Appendix B 269 Appendix C 271 Appendix D 272 Appendix E 273 GENERAL DISCUSSION 275 iv Acknowledgements During the time of my PhD I could not have wished for better guidance. I would like to express my deepest gratitude to Lyndsey Nickels, my primary supervisor, who convinced me to come back to Macquarie to start a PhD, and to my second supervisor, Saskia Kohnen. I have truly enjoyed learning from their wide range of expertise, and have been inspired by their energy and enthusiasm for research. Thank you for being so generous with your time and feedback, and for always reminding me to ask the right questions. Thank you to the directors from IDEALAB for their time and energy in setting up a wonderful and valuable international collaboration: Prof Roelien Bastiaanse, Prof Ria de Bleser, Prof David Howard, Prof Barbara Höhle, Prof Gabriele Miceli, and Prof Lyndsey Nickels. I would like to thank the participants in my research, in particular the people with aphasia. I admire the enthusiasm and energy with which they performed many tasks, that may have been challenging at times. In particular I would like to thank GEC and JOD and their partners. I am thankful for their hard work during the many sessions, and I have truly enjoyed the chats over tea and (many) biscuits. This research would not have been possible without their ongoing enthusiasm and commitment. The friendly and supportive environment at CCD (MACCS) have contributed to my enthusiasm for research, and have made my time as a PhD student so wonderful. I would like to thank Lesley, Katie, Teri, Marcus and Craig for helping out along the way. Over the years colleagues became friends, and have made it such a nice place to work. v In particular I would like to thank Anastasiya, Britta, Danielle, Huachen, Linda, Nathan, Rochelle, Serje, Thushara, and Yvette. Thank you also to my dear friends back home, showing that friendship can cover great distances: Nienke, Laura, Martine, Vera, Rimke, Suzanne, Jeanine, Mariska and Rinie. I have always felt greatly supported by my parents in my decision to start a PhD in Australia for which I am grateful. Pap en mam – ik heb mij altijd zeer gesteund gevoeld in mijn keuzes. Bedankt voor jullie brede interesse en het voortdurende vertrouwen in mijn werk. Also many thanks to my brother Henk, and Anne, for their valuable support and interest in my work. Finally, I am very grateful to Pete, for being so caring and supportive, and for always making me laugh. vi This PhD has been carried out as part of the Erasmus Mundus joint International Doctorate for Experimental Approaches to Language and Brain (IDEALAB) Macquarie University (Australia) University of Newcastle (UK) University of Groningen (the Netherlands) University of Potsdam (Germany) University of Trento (Italy) vii viii General Summary This thesis focuses on the processes involved in spelling, and in particular explores the nature of impairment and rehabilitation of acquired dysgraphia, using a cognitive neuropsychological approach. The first study investigates the spelling impairment of GEC, a man with acquired dysgraphia. GEC showed characteristics of graphemic output buffer impairment, together with lexical influences (e.g., frequency) on performance and a large number of deletion errors of multiple letters (fragment errors). A detailed error analysis provided evidence to support the hypothesis that fragment errors can be the result of rapid decay of activation from the graphemic output buffer. The study also concludes that lexical influences could be observed in graphemic output buffer impairment due to cascading of activation. The second study investigates the nature of acquired sub-lexical spelling impairments. Data from spelling sounds in isolation and spelling non-words are analysed to inform three issues: the relationship between performance on the two spelling tasks, the effects of phoneme-grapheme consistency and frequency on spelling, and the use of context when spelling a vowel in a non-word. Results indicated that people with aphasia show comparable difficulty spelling single sounds and sounds in initial position of the non-word. Furthermore, accuracy of individual PGCs was influenced by frequency and consistency of the non-word. Finally, no evidence was found for a loss context sensitive rules when spelling vowels. The final two studies focus on rehabilitation of acquired dysgraphia. Study three comprises a literature review on generalisation effects after treatment of acquired dysgraphia. This study summarises 40 treatment studies, investigating the link between type of impairment, method of treatment, and generalisation. Some treatment studies in the literature have found an improvement in untreated items, however it is unclear what predicts such generalisation of treatment effects. ix Study three highlights that a mechanism of interactive processing may play a role in treatment and generalisation. The final study therefore investigates this mechanism of interactivity within the spelling process in two treatment studies examining the role of orthographic neighbourhood size on the effects of treatment and generalisation in two individuals with acquired dysgraphia. Feedback between the orthographic lexicon and the graphemic output buffer predicts a target word will activate orthographically related words. However, while treatment improved spelling for treated items, there was no generalisation and no evidence for effects of orthographic neighbourhood size on treatment. It was hypothesised that severe impairment to the graphemic output buffer reduced the feedback within the spelling system. This thesis contributes to our understanding of the nature of spelling impairment and rehabilitation. Furthermore, the thesis highlights the value of cognitive neuropsychological methods in research of acquired dysgraphia. x
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