This Master's thesis explores how developmental dysarthria and communication disability effect the socialization and communication abilities of people with cerebral palsy. The thesis is divided into two primary sections: theoretical and empirical. The theoretical part is divided into four sections (cerebral palsy, disrupted communication abilities in people with cerebral palsy, dysarthria, communication and socialization in people with cerebral palsy). The next part of the work presents the analysis of dysarthria and disrupted communication abilities effect of the socialization and communication abilities in people with cerebral palsy. The empirical section is based on original research, primarily employing qualitative methods, namely semi-⁠structured interviews. I interviewed six individuals all of whom are affected by cerebral palsy and developmental dysarthria. Research goals included how disrupted communication skills, known as developmental dysarthria, affect the individuals' communication and socialization skills, how it affects them in daily situations, what the main hallmarks of these situations are, what these situations have in common and how developmental dysarthria affects the individuals' motivation towards socializing. The results showed that the degree to which disrupted...

Children with cerebral palsy in South Africa may face various challenges in their acquisition of literacy. In order to address these, an understanding of the nature of these challenges can be seen as the first step. Successful acquisition of literacy is not only determined by formal instruction. Studies have found that the development of literacy skills appear to be related to supportive home literacy environments and experiences. Specific aspects of home literacy experiences have been found to predict later literacy and language skills. The aim of this research is to describe the home literacy experiences of Zulu children with cerebral palsy aged four to six years and their peers without disabilities living in KwaZulu-Natal. Caregivers of 10 children with cerebral palsy and caregivers of 10 children without disabilities, matched for age and gender, were selected from various preschools and schools within KwaZulu-Natal to complete a questionnaire. The questionnaire was developed based on a previous study and is based on five domains of home literacy experiences which include: (1) the child’s literacy experiences and interest, (2) materials and caregiver activities for child literacy development, (3) shared storybook reading, (4) caregiver’s own literacy materials and activities, and, (5) caregiver’s expectations of their child’s literacy development. The results obtained indicated that, in general, the home literacy experiences of the two groups did not differ for most of the home literacy experience aspects. Both groups of caregivers engaged in literacy mediating activities with their children. There were similar trends in the frequency that the child and the caregivers in both groups engaged in literacy practices, which shows that both groups of children had literate role models to demonstrate literacy experiences. Both groups had relatively high expectations of their children’s literacy development. Statistically significant differences were found between the two groups regarding the reported level of active involved in some of the activities engaged in during shared storybook reading and in children’s interest in literacy activities. Children with cerebral palsy were reportedly less actively involved during certain shared storybook reading activities and less interested in literacy activities. The study highlights the nature of the home literacy experiences and some of the areas that need consideration in the literacy development of children with cerebral palsy. Suggestions for future research are provided. / Mini Dissertation (M(AAC))--University of Pretoria, 2017. / National Research Foundation (NRF) / Centre for Augmentative and Alternative Communication (CAAC) / M(AAC) / Unrestricted

Background: Children and adolescents with Cerebral Palsy [CP] are not as physically active as their typically developing peers, which contributes to reduced physical fitness. Previous studies about participating in physical activity [PA] and disability in general have identified barriers and facilitators. However, less research has been performed targeting persons with CP, the most common physical disability in childhood. Previous studies mainly focused on the parental perspectives of their child with CP. Aim: This systematic review aimed to identify and critically review the existing literature on barriers to and facilitators for children and adolescents with CP to participate in PA. Method: A literature search was carried out in four online databases for social and health sciences to select peer-reviewed qualitative articles, identifying barriers to and facilitators for children and adolescents with CP to participate in PA. Studies were identified following the pre-established selection criteria, resulting in eight qualitative studies.  Results: Various personal and environmental factors were identified working as either barriers or facilitators, and yielded into two sub-categories and codes for personal and environmental factors. Sub-categories of identified barriers and facilitators within personal factors were physical abilities to participate in physical activity and psychological factors, and acceptance and relationships and possibilities to participate in PA within environmental factors. Conclusion: Children and adolescents with CP are facing many personal and environmental barriers and facilitators to participate in PA. Further research is needed to develop an awareness program about CP to work with them, and to investigate the number of individuals required in a team to give this group the time and attention they need to master their skills. / Achtergrond: Kinderen en adolescenten met Cerebrale Parese [CP] zijn niet zo fysiek actief als hun typisch ontwikkelde leeftijdsgenoten, wat bijdraagt aan een verminderde fysieke conditie. Eerdere studies over deelname aan fysieke activiteiten [PA] en beperkingen hebben belemmeringen en mogelijkheden in kaart gebracht. Er is echter minder onderzoek gedaan naar CP, terwijl dit de meest voorkomende fysieke beperking in de kindertijd is. Eerdere studies richtten zich vooral op de ouderperspectieven van hun kind met CP. Doel: Het in kaart brengen en kritisch beschouwen van bestaande literatuur over belemmeringen en mogelijkheden om deel te nemen in PA voor kinderen met CP. Methode: Een literatuuronderzoek is uitgevoerd in vier online databases voor sociale en gezondheidswetenschappen om peer-reviewed kwalitatieve artikelen te selecteren die belemmeringen en mogelijkheden identificeren voor kinderen en adolescenten met CP om deel te nemen in een fysieke activiteit. Studies werden geïdentificeerd volgens de vooraf vastgestelde selectiecriteria, wat resulteerde in acht kwalitatieve studies.  Resultaten: Verschillende persoonlijke en omgevingsfactoren werden geïdentificeerd, werkend als belemmering of mogelijkheid, en resulteerden in twee subcategorieën en codes voor beide factoren. Belemmeringen en mogelijkheden binnen de persoonlijke factoren resulteerden in de subcategorieën fysieke capaciteiten om deel te nemen aan fysieke activiteiten psychologische factoren, en binnen de omgevingsfactoren in acceptatie en relaties en mogelijkheden om deel te nemen aan een fysieke activiteit. Conclusie: Kinderen en jongeren met CP worden geconfronteerd met vele persoonlijke en omgevingsbelemmeringen en mogelijkheden om deel te nemen aan PA. Er is verder onderzoek nodig om een bewustwordingsprogramma over CP te ontwikkelen om met deze doelgroep te werken en om te onderzoeken hoeveel individuen er nodig zijn om deze groep de tijd en aandacht te geven die nodig is om hun vaardigheden onder de knie te krijgen.

The thesis investigated objective neuroimaging biomarkers in parkinsonian syndromes, which could be applied to increase diagnostic accuracy. To find convergence of the literature concerning disease-specific patterns in Parkinson’s disease and progressive supranuclear palsy, we conducted meta-analyses. In Parkinson’s disease glucose hypometabolism was re- vealed in bilateral inferior parietal cortex and left caudate nucleus and focal gray matter atrophy in the middle occipital gyrus. In progressive supranu- clear palsy we identified gray matter atrophy in the midbrain and white mat- ter atrophy in the cerebral/cerebellar pedunculi and midbrain. In sum, in Parkinson’s disease hypometabolism outperforms atrophy and in progres- sive supranuclear palsy we validated pathognomonic markers as disease- specific. Our studies create a novel framework to investigate disease- specific regional alterations for use in clinical routine. Further, we inves- tigated neural correlates by voxel-based morphometry and discriminated disease and clinical syndrome by multivariate pattern recognition in sin- gle patients with corticobasal syndrome and corticobasal syndrome with a unique syndrome - alien/ anarchic limb phenomenon. We found gray matter volume differences between patients and controls in asymmetric frontotem- poral/ occipital regions, motor areas, and insulae. The frontoparietal gyrus including the supplementary motor area contralateral to the side of the af- fected limb was specific for alien/ anarchic limb phenomenon. The predic- tion of the disease among controls was 79.0% accurate. The prediction of the specific syndrome within a disease reached an accuracy of 81.3%. In conclusion, we reliably classified patients and controls by objective pattern recognition. Moreover, we were able to predict a specific clinical syndrome within a disease, paving the way to individualized disease prediction.:SELBSTSTÄNDIGKEITSERKLÄRUNG I ACKNOWLEDGMENTS II SUMMARY III ZUSAMMENFASSUNG VIII BIBLIOGRAPHISCHE DARSTELLUNG XIV CONTENTS XVI 1 GENERAL INTRODUCTION 1 1.1 ParkinsonianSyndromes .................... 2 1.2 Parkinson’sDisease ....................... 2 1.2.1 DiagnosticCriteria .................... 3 1.3 ProgressiveSupranuclearPalsy ................ 4 1.3.1 DiagnosticCriteria .................... 5 1.4 CorticobasalDegeneration ................... 5 1.4.1 DiagnosticCriteria .................... 7 1.5 ImagingBiomarkers ....................... 7 1.6 CurrentThesis .......................... 9 1.6.1 MotivationandFramework ............... 9 1.6.2 ResearchQuestions................... 9 2 GENERAL MATERIALS AND METHODS 12 2.1 MagneticResonanceImaging.................. 12 2.2 AnalyticalMethods........................ 13 2.2.1 Meta-Analysis ...................... 13 2.2.2 Voxel-BasedMorphometry ............... 14 2.2.3 Support-Vector Machine Classification . . . . . . . . . 15 2.3 Multi-CentricData ........................ 16 2.4 ClinicalAssessment ....................... 17 3 Study 1 4 Study 2 5 Study 3 6 Study 4 7 Study 5 8 DISCUSSION 73 8.1 MainFindings........................... 73 8.2 Statistical Approaches to Find Imaging Biomarker . . . . . . 76 8.3 Brain Alterations and their Utility as Imaging Biomarker . . . . 77 8.4 Limitations ............................ 78 8.5 Contributions of the Current Thesis and Future Directions . . 79 9 REFERENCES APPENDIX XVIII LIST OF AUTHORSHIP XXVII CURRICULUM VITÆ XXXVIII

Introduction: Cerebral palsy (CP) is a CNS related permanent disorder following a lesion on the developing brain. Spasticity and muscle weakness are the most commonly reported impairments. Aim: To describe spasticity measured instrumentally as the muscle resistance and muscle strength in children with CP and in typically developing children (TD), and explore the relationship between muscle resistance, muscle strength and functional mobility. Method: Measurements of the muscle resistance in plantarflexors with Neuroflexor®. Muscle strength measurements in the lower leg with a rig-fixed dynamometry in n= 4 children with CP and in n = 11 TD children. Timed Up and Go (TUG) test measured in seconds in 4 children with CP. Results: Mann- Whitney U test showed a significant increased muscle resistance in plantarflexors during a fast movement between children with CP and TD children (p = 0.044). It was significantly shown that the children with CP were weaker in their dorsiflexors compared to the group of TD children (p = 0,001). Analysis for muscle resistance and TUG was collected for 2 children with CP and therefore no correlation analysis could be made. Conclusion: This pilot study indicates that the children with CP were weaker in their dorsiflexors compared to TD children and that muscle resistance measured during a passive elongation was higher, but no statistically significant conclusion can be made due to few included numbers of participants.

Background & aims: Several systematic reviews have demonstrated that caring for a child with functional limitations leads to poor caregiver mental health outcomes. For instance, depression, anxiety and caregiver burden are endemic in informal (unpaid) caregivers of children with cerebral palsy (CP), a common paediatric disability. However, caregivers who receive an adequate amount of social support are likely to better adjust to the caregiving role. Given the increasing move towards family-centred care and evidence-based care, there is a need to evaluate caregivers’ mental health and to develop and implement context-specific caregiver well-being programs. The valid measurement of the impact of these programs is dependent on the availability of psychometrically robust patient-reported outcomes (PROMs). Unfortunately, most available PROMs have been developed in high-income settings, and their applicability in low-resource settings such as Zimbabwe may be questionable. The present study thus aimed to develop a context-specific caregiver strain outcome, to adapt, translate, and validate a social support outcome measure and finally to profile the mental health of caregivers of children with CP residing in Zimbabwe. Methods: The Zimbabwean Caregiver Challenges Scale (ZCCS) was developed through the amalgamation of findings from a systematic review, caregivers’ interviews and two rounds of content validation by a panel of experts. A systematic review was then undertaken to appraise the psychometric properties of the Multidimensional Scale of Perceived Social Support (MSPSS). The MSPSS was then adapted, translated into Shona, a Zimbabwean native language, and validated. In the final phase, further validation of the ZCCS and the MSPSS was done, and structural equation modelling was used to profile the mental health profile of caregivers. Results: The ZCCS yielded four factors which were ; physical & economic burden, concerns for the child, family relations and community participation. The ZCCS was found to be a reliable tool as it yielded excellent Internal Consistency (IC) ratings at both sub-scale [α range: 0.765- 0.841] and scale level [α=0.925]. The Interclass Correlation (ICC) (95% CI) for ZCCS scores at baseline and after four weeks was 0.880 (0.793: 0.930), demonstrating the stability of the ZCCS. We replicated the original 3-factor structure of the MSPSS through factor analysis. Further, dichotomisation of scoring options and the deletion of one item resulted in a parsimonious solution as the 11-items solution met Rasch modelling requirements. The MSPSS-Shona version is reliable; it yielded excellent IC ratings at both sub-scale [α range: 0.833-0.892] and scale levels[α=0.8685]. The ICC rating (95% CI) for MSPSS scores at baseline and after four weeks was 0.980 (0.959: 0.990) demonstrating the stability of the MSPSS, and the person separation index (PSI) was 0.731. Both the ZCCS and MSPSS displayed construct validity; higher caregiver burden was associated with greater psychiatric morbidity and lower health related quality of life. Caregivers who received a higher amount of social support had the best mental health outcomes. Further, structural equation modelling provided evidence of the multidimensionality of the caregiver burden. Contextual factors, such as increased child age, increased caregiving duration, lower child functional capacity/more severe CP, and lower socioeconomic status were associated with poor mental health functioning. Conclusion: The thesis contributes to the body of knowledge by documenting the validation of caregivers' mental health outcomes and determination of mental health functioning of caregivers residing in low-resource settings. The ZCSS and MSPSS were both found to be valid and reliable measures within the context of Shona speaking rural and urban Zimbabweans. It is therefore suggested that both measures can be used with confidence in these contexts. Efforts should be made to integrate patient-reported outcome measures (PROMs) in routine clinical care and research and in developing and implementation of tailor-made caregiver wellness programs, within the validation contexts.

The computational musculoskeletal models that are used to study muscle moment-generating capacities of persons with movement disorders and planning treatment options must be accurate, and take into account the inter-individual variability of musculoskeletal geometry. In Paper I the methods of creating the subject-specific musculoskeletal model of the lower extremities from magnetic resonance images (MRIs) were developed. The subject-specific model was used to analyze hip, knee and ankle muscle moment arms (MALs) and muscle-tendon lengths (MTLs) during gait in a subject with unilateral cerebral palsy (CP), and to evaluate the accuracy of widespread and commonly-used scaled generic model. It was found that the scaled generic model delivered accurate values for changes in MTLs in most muscles. However, the scaled generic and the subject-specific lower extremity musculoskeletal models showed substantial differences in MALs calculated during gait. In Paper II subject-specific musculoskeletal models of nine subjects with unilateral CP were created to study muscles volumes, MTLs and MALs; and to examine the accuracy of MALs calculated by the scaled generic models. It was shown that the scaled generic model significantly underestimated hip MALs discrepancies between the affected and the non-affected sides of the lower extremities. However, it significantly overestimated hip adduction/abduction of gluteus maximus, gluteus medius, gluteus minimus, tensor fascia latae and biceps femoris long head; and hip flexion of adductor longus and rectus femoris in the affected and the non-affected sides. It was also found that muscle volumes and hip abduction MALs in gluteus medius and gluteus minimus, hip flexion MALs in iliacus and hip rotation in gluteus maximus were smaller in the affected side of lower extremities. MTLs in the affected and the non-affected sides throughout the range of hip motion were similar. This thesis suggests the need for the subject-specific musculoskeletal models that can account for variability of muscle attachments and musculoskeletal geometry of persons with movement disorders. Based on inaccuracies of the scaled generic model reported here, the generic models that are used to guide treatment decisions must be tested, and interpreted with care. / QC 20110901

Patients with cerebral palsy account for great upper extremities deviationswhile walking. However, the number of studies assessing their upper bodygait kinematics are rare and no studies have been conducted interested inthe whole body kinematics during walking. In this study, we created awhole body index, the Body Profile Score made of modified existing kinematicindexes assessing the gait pattern of children with cerebral palsy. TheBody Profile Score (BPS) is an average of combination of the Gait ProfileScore (GPS), a modified Trunk Profile Score (TPS), a modified Arm PostureScore (APS) and a also new index called Head Profile Score (HPS), basedon a similar calculation. Dierent versions of the BPS were tested on threegroups: a control group, a CP group before botulinum toxin A treatmentand a CP group after botulinum toxin A treatment. The results showed apoor level of linear correlations between the dierent BPS versions and theGait Profile Score, indicating that lower body indexes such as the GPS orGait Deviation Index (GDI) and full body index such as the BPS do not renderthe same information. The BPS is the first index proposing a full bodykinematic analysis and aims at showing that such an analysis is needed ingait assessment of spastic children in order to have a realistic overview ofthe pathological walking condition.

Bakgrund: Cerebral pares (CP) är den vanligaste fysiska funktionsnedsättningen bland barn. Constraint-induced movement therapy (CIMT) syftar till att öka funktion i den nedsatta handen. Det finns en kunskapslucka kring föräldrars upplevelser av CIMT-träning i barnets vardag. Syfte: Syftet var att undersöka och beskriva upplevelser av CIMT via funktionell terapi bland föräldrar till barn med spastisk hemiplegisk cerebral pares, med fokus på föräldramedverkan, genomförande och följsamhet. Metod: Designen var en kvalitativ intervjustudie med semistrukturerade intervjuer. Metodansatsen var en induktiv kvalitativ innehållsanalys. Resultat: Nio föräldrar från sju familjer deltog i studien. Föräldrarna hade barn i åldrarna fyra till sju år med Gross Motor Function Classification System (GMFCS) 1. Studien resulterade i fyra huvudkategorier; Hinder som måste övervinnas, Draghjälp och stöd, Hantera förväntningar och Lösningar för att få träning att fungera i vardagskontexten, med tillhörande underkategorier. Resultatet visade att tiden är det största hindret och att inblandning av lek är den bästa lösningen att få till träningen i vardagen. Egna förväntningar på sig själv och vikten av yttre stöd beskrevs. Föräldramedverkan visades vara avgörande för genomförande av interventionen. Konklusion: Det är av vikt att ta vara på föräldrarnas upplevelser för bästa genomförande och för att gynna följsamhet vid CIMT. Resultatet kan användas för att utveckla interventionen inom aktuell region.

Young children with HCP experience difficulties performing age-appropriate daily activities such as self-care and play. Research in neuroplasticity has shown that intensive, task-specific training in early development is ideal to maximize functioning in children with HCP. The aim of this study was to analyze the feasibility of a new manualized clinical guideline, Family Activity Adaptation Model (FAAM), via Telehealth to coach families to develop daily activities and routines that facilitate functional bimanual skills in young children with hemiplegic cerebral palsy (HCP). Ten caregivers and children with HCP (3-7 years) received virtual training on how to embed bimanual intensive training (HABIT) into their everyday activities and routines on their own at home for 90 minutes a day, 5 days a week over 8 weeks. Caregivers and children received virtual coaching using the FAAM method 2 times per week. FAAM Activity Analysis was used to describe manual development. Daily activity logs were used to assess family adherence and home program feasibility. Bimanual functional goal performance and satisfaction was measured using the Canadian Occupational Performance Measure (COPM). Perceived changes in overall bimanual functional skills were evaluated using the Mini-Children’s Hand Use Experience Questionnaire (Mini-CHEQ). Caregiver stress, burden and program satisfaction was monitored using the Parenting Stress Index (PSI-4-SF), the Ease of Caregiving for Children measure and a caregiver satisfaction survey. The measures were assessed immediately prior to (pretest), midpoint (burden measures only, repeated measures ANOVA) and immediately after (posttest, Wilcoxon Sign Rank Test) the intervention. All families made statistically and clinically significant improvements in goal performance (p=.008) satisfaction (p=.007), and overall bimanual hand use (p=.035). All families completed daily logs and practiced strategies for an average of 81.37 (SD=7.069) minutes a day. Caregiver burden and stress remained consistent throughout the study and all caregivers reported satisfaction with the program. This is the first study to manualize a family centered, telehealth-delivered intensive motor training program. This study adds a continuum of care and access to services for children and families with limited resources. The study supports clinicians with home program design and meets the individual needs of families using resources in their natural environment.