Paediatrics (RCH) - Theses

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    Proximal femoral osteotomy in children and adolescents with cerebral palsy
    Zhou, Leena ( 2018)
    Background Cerebral Palsy (CP) is the most common cause of physical disability affecting children in developed countries. Approximately one third of children with CP may develop hip displacement. Non-ambulant children at Gross Motor Function Classification System (GMFCS) levels IV and V are at highest risk. Without early detection through surveillance programs, hip displacement can progress to hip dislocation, which is frequently painful and negatively impacts health-related quality of life (HRQoL). Injections of Botulinum Neurotoxin A (BoNT-A) have no role, and soft tissue surgery has a limited role in preventing hip displacement in non-ambulant children with CP. Bony hip reconstruction surgery such as a proximal femoral osteotomy (PFO) is effective in stabilising the hip and HRQoL. PFOs include Femoral Derotation Osteotomies (FDO) which aim to improve the gait of an ambulant child (GMFCS I-III), and Varus Derotation Osteotomies (VDRO) which aim to contain the hips in non-ambulant children (GMFCS IV-V). However, PFOs can carry high risks, especially in children with medical co-morbidities such as respiratory disease, nutritional deficiencies, hypertonia and osteopenia. Aim This thesis involved a series of three studies, which aimed to expand our knowledge of the trainee learning curve, outcomes and adverse events relating to PFO in children and adolescents with CP. Method and Results A new implant combining locking and cannulated technology (Locking Cannulated Blade Plate, LCBP) was recently developed for use in PFO. A pilot study was performed on the first 25 patients who had surgery with the LCBP, at the Royal Children’s Hospital (RCH), Melbourne. This study established safety for use in children as young as three, with weights as low as eleven kilograms. A further prospective, parallel cohort study of 90 consecutive children with CP was conducted to compare the LCBP against with existing non-cannulated, non-locking implant (Angled Blade Plate, ABP). Technical and radiological outcomes of surgery were similar between implants. However, the surgical technique was reported by trainees to be easier when using the LCBP, with less technical errors. Approximately 60 percent of the children experienced minor adverse events including: constipation, inadequate pain control, and respiratory compromise. However, a CP specific tool was not available to classify the severity of events. Study three was performed to clarify the Modified Clavien-Dindo (MCD) system for lower limb surgery in children with CP and test its’ reliability for classifying adverse events. Very good reliability was demonstrated amongst members within a multidisciplinary team. Conclusion Novel findings from these studies may help improve the safety and efficacy of the management of hip displacement in children with CP. Further research should address the long-term outcomes of PFO in children with CP, evaluate the validity of the MCD for children in CP and determine if the MCD can be embedded in the electronic medical records (EMR) as a routine tool for audit and clinical research.
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    Investigating and interpreting the use, costs, accessibility and organisation of healthcare services among children with cerebral palsy
    Meehan, Elaine ( 2016)
    Background and Rationale: Cerebral palsy is an umbrella term for a group of non-progressive disorders of movement and posture. Compared to their typically-developing peers, children with cerebral palsy are at an increased risk of multiple comorbidities and frequent acute illnesses that affect almost all of the organ systems, and as a consequence, have significantly greater medical care needs. There is growing interest internationally in investigating the patterns of medical service use among children with complex conditions such as cerebral palsy. It is hoped that a greater understanding of their healthcare needs could assist with decision making around health service delivery for this group. Aims: The aims of this thesis were to describe the patterns of medical service use among children with cerebral palsy, identify factors associated with the frequency and type of medical services used by this group, and compare their hospital admission and emergency department presentation characteristics with those of the general population. A secondary aim was to explore with parents the financial implications of having a child with additional medical care needs. Methods: Population cohorts of children with cerebral palsy and data linkage techniques, as well as survey methods, were used. The Victorian Cerebral Palsy Register was linked to the emergency department and inpatient databases of the Australian state of Victoria’s two paediatric hospitals to examine service use at these centres, and to the Victorian Admitted Episodes Dataset and Victorian Emergency Minimum Dataset to investigate statewide admissions and emergency department presentations respectively among population cerebral palsy cohorts. A purposefully-designed survey gathered information from parents on other medical services used, and on how they cope financially in light of their child’s medical care needs. Results: Of 2,183 children with cerebral palsy, 814 had at least one emergency department presentation and 1,443 had at least one admission at one of Victoria’s two paediatric hospitals over a five-year period, accounting for an average of 1,400 admissions and 700 emergency department presentations at the two hospitals each year. A higher than expected proportion of paediatric emergency department presentations were triaged as high urgency (71%) and resulted in a hospital admission (44%). Children with severe and/or complex cerebral palsy had more frequent admissions and emergency department presentations to the two hospitals, as well as higher re-admission rates. A similar pattern was observed when admissions and emergency department presentations at all hospitals in the state were investigated, those children with more severe and/or complex cerebral palsy having more hospital episodes each year. Compared to the general population of children, those with cerebral palsy had longer lengths of stay (median of 3 vs 2 nights; p<0.001), and more procedures (median of 2 vs 1 procedures; p<0.001) per multi-day admission, and more emergency department presentations that were preceded by an ambulance arrival (27% vs 8%; p<0.001), triaged as being of high urgency (66% vs 34%; p<0.001) and resulted in a hospital admission (38% vs 13%; p<0.001). Ultimately, they accrued higher hospital charges per episode. The types of paediatric specialists most often seen in outpatient settings were general or developmental paediatricians, orthopaedic surgeons, rehabilitation specialists, and neurologists. Most children received medical services from a number of different providers, in multiple different settings, and this continued into adolescence and early adulthood. Overall, the number and complexity of hospital episodes, the number of professionals involved in their medical management, and the proportion of families reporting financial difficulties all rose with increasing cerebral palsy severity and complexity. Conclusions and Significance: Through a series of data linkage studies and a purposefully-designed parent survey, this thesis provides new knowledge about medical service use among children with cerebral palsy. It provides, for the first time, objective population-level data on which future decisions around health service delivery for this patient group can be based, as well as highlighting some future research priorities. 
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    Scoliosis in children with cerebral palsy: a population based study
    Ang, Soon Ghee ( 2015)
    Introduction Scoliosis is the most common spinal deformity in children with cerebral palsy. Previous published studies have been based on institutionalised patients and not on a total population of individuals with cerebral palsy. Methods This study was based on both prospective and retrospective cross-section analysis of 292 children identified from the Victorian Cerebral Palsy Register. These children were spread across GMFCS levels I–V. The children were assessed during their transition clinic appointment prior to exit from the Royal Children’s Hospital. The research looked at three main sections: clinical review for scoliosis, radiographic assessment of scoliosis, and CHQ and CPCHILD questionnaires survey. Cobb angles were measured by two experienced observers. Results If a Cobb angle of more than 10° was used, then 40% of patients were classified with scoliosis. By changing the definition of scoliosis in cerebral palsy to a Cobb angle greater than 40°, the prevalence of “clinically important scoliosis” was 12.7%. The majority of the severe curves occurred in children at GMFCS levels IV and V. As the GMFCS level increased, the mean Cobb angle increased. The mean score for the questionnaires decreased as the GMFCS level increased. Conclusion The CP scoliosis and the scores for the questionnaire were closely related to the GMFCS levels. The prevalence of CP scoliosis is overestimated in other studies. Our study shows the prevalence of CP scoliosis is 12.7% using a Cobb angle of more than 40°. Non-ambulant children are at high risk of developing scoliosis and formal spine surveillance should be considered.
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    Speech and language abilities of children with cerebral palsy: a population-based study
    Mei, Cristina ( 2014)
    Although speech and language development are known to be vulnerable in children with cerebral palsy (CP), little is known about the prevalence and features of impairments in these areas during early childhood. Speech and language are known to influence the activity and participation of children with CP, yet no population-based study has examined the contribution of speech versus language. Activity and participation are recognised as key health outcomes, however, at the time of this research, no tool measuring these functional areas existed specifically for children with CP with communication impairment. This study consisted of three parts: (1) a qualitative study exploring the activity and participation of children with CP to provide the basis of a questionnaire measuring these constructs; (2) development and piloting of the questionnaire; and (3) a population-based study examining the speech and language outcomes of children with CP aged 5 and 6 years at an impairment and activity/participation level. In Part 1, activity and participation were explored by interviewing 13 parents of children with CP. Parents’ responses were thematically analysed to reveal key areas to include in a structured questionnaire of activity and participation. Children’s reported activities and participation were related to: (1) leisure, early learning and educational activities (e.g., reading, classroom activities); (2) communication (e.g., asking questions, expressing needs); (3) independence within the home (e.g., activities of daily living, household chores); and (4) engagement within the community (e.g., shopping, visiting family). Barriers affecting children’s activity and participation included aspects of parents’ own interactions with their child (e.g., not offering choices), unfamiliar people and settings, the negative attitudes of others, and the child’s frustration. Facilitators included the support received from the child’s family and school, being amongst children, having a familiar routine, and the child’s positive disposition (e.g., being determined). These qualitative findings informed the development of an activity and participation questionnaire (Part 2). In Part 2, questionnaire items were devised based on the codes identified during the thematic analysis of the qualitative data. The developed items focused on children’s activity and participation within the home, school and community, and the environmental and personal factors affecting these areas of functioning. Face validity of the questionnaire was determined by verifying that the items adequately described and measured the most relevant aspects of the underlying construct. Content validity was established through expert opinion of the questionnaire’s items. The questionnaire was piloted on seven parents of children with CP and was modified accordingly prior to its use in the population-based study. In Part 3, 84 children with CP born between August 2005 and August 2007 were recruited through the Victorian Cerebral Palsy Register. This represented 37% of the known living population of this age range. Participants were representative of non-participants across key variables (e.g., CP type and distribution, Gross Motor Function Classification level, cognitive status). Children underwent a comprehensive face-to-face assessment. The primary outcome measures were the Diagnostic Evaluation of Articulation and Phonology, the Preschool Language Scale-4, and the developed activity and participation questionnaire. The internal consistency of the questionnaire was examined during this population-based study and was found to be good-to-excellent for items relating to activity and participation. Findings from the population-based study revealed that 80% (66/83) of the sample had some form of speech and/or language difficulty (24%, 20/84, were non-verbal). Speech delay/disorder and language impairment were identified in 66% (40/61) and 62% (51/82) of children, respectively. The subgroups of language impairment identified were isolated receptive (7%, 6/82) or expressive (5%, 4/82), and mixed receptive-expressive (45%, 37/82). Speech delay/disorder consisted of the following subgroups: articulation delay (26%, 16/61), articulation disorder (28%, 17/61), phonological delay (17%, 10/60), consistent phonological disorder (7%, 4/58), and inconsistent phonological disorder (19%, 11/58). Children at most risk for speech delay/disorder and/or language impairment were those with mixed or non-spastic CP motor types, quadriplegia, severe fine motor limitations, epilepsy, hearing, cognitive or vision impairment, and those born with an extremely low birth weight. Non-verbal children and those with language impairment were at the greatest risk for restrictions in activity and participation compared to children with age appropriate speech and language and those with speech delay/disorder in the absence of language impairment. Activity and participation were best facilitated by positive child attributes and the support received from others, whereas barriers included communication partners (e.g., their negative attitudes or not understanding the child) and the difficulty parents experienced in supporting their child. Study findings highlight that the majority of children with CP experience some form of speech and/or language difficulty at 5 and 6 years of age, underscoring the need to routinely assess and monitor these areas. The subtypes of speech delay/disorder and language impairment identified provide suggestions for how to characterise speech and language in children with CP. Findings indicate that activity and participation-based therapy may not be warranted for children with speech delay/disorder who have age appropriate language skills. To confirm this, further research is needed comparing the activity and participation outcomes of this subgroup against typically developing children. The environmental and personal factors that were found to have the strongest impact on children’s activity and participation may provide suggestions for how to optimise activity and participation through intervention. For instance, the effectiveness of reinforcing positive child attributes and counselling parents on early communicative strategies that encourage children to indicate their needs may be trialled in future research to determine their clinical utility in enhancing activity and participation outcomes.
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    Cerebral palsy in Victoria: a population-based study
    Reid, Susan Margaret ( 2012)
    BACKGROUND AND AIMS: This thesis reports an exploration of the epidemiology of cerebral palsy in Victoria and a comparison of Victorian data with data from other populations. Each of six broad aims related to a particular area of investigation and thesis chapter. The aims were to describe 1) the frequency of cerebral palsy in Victoria, 2) overall temporal trends and by gestational age group, 3) clinical characteristics, including motor type, topographical pattern, and motor severity, 4) accompanying disorders and impairments, 5) neuroimaging patterns and their relationship to perinatal variables and clinical characteristics, and 6) survival. METHODS: The Victorian Cerebral Palsy Register was used to extract data on individuals born in Victoria with non-postneonatally acquired cerebral palsy based on specific criteria for each chapter. A systematic review of the literature was also undertaken to enable comparison of classification, definitions, and epidemiological data with the Victorian data. RESULTS: The decade-specific prevalence of cerebral palsy in Victoria was 1.6 per 1000 live births for the 1980s, 1.8 for the 1990s and 1.7 for 2000-2004. In comparison, the range of prevalence estimates from 33 surveillance systems was 1.8-4.0 per 1000 for 1990s birth cohorts and 1.2-3.0 per 1000 for 1980s cohorts; no methodological or population factors explained the wide variability. The rate of cerebral palsy in Victoria increased during the 1980s and early 1990s, particularly for extremely preterm infants, but also for infants born at term. The data suggest a reversal of this trend from the mid-1990s for both term and extremely preterm infants, a finding observed in other populations. In general, the proportion of cerebral palsy cases with each motor type, topographical pattern, motor severity level, and associated impairment varied widely between surveillance systems, and there was a lack of consensus on definitions and classifications. Victorian data showed a relative increase in mild motor impairment between 1970 and 2003 and an increase in the proportion of cerebral palsy cases with spastic hemiplegia and diplegia relative to spastic quadriplegia; however, no improvement in survival was seen, even for the most severely affected. The most common imaging finding was periventricular white matter injury (PWMI), a pattern associated with spastic diplegia and hemiplegia, and mild-moderate motor impairment. While PWMI was the primary pattern in children born preterm, it was also common in children born at term. Grey matter injuries and malformations were predominantly associated with term birth, and bilateral brain injuries were associated with poor gross motor function. The majority of focal vascular insults and unilateral malformations resulted in spastic hemiplegia. CONCLUSIONS: Through a series of systematic reviews, this thesis has contributed to a better understanding of issues relating to the definition and classification of cerebral palsy and, in particular, emphasised the potential for these to affect estimates of prevalence and distribution of clinical subtypes. In addition, it has resulted in a deeper understanding of clinical, temporal and neuropathological patterns in cerebral palsy and exemplifies the potential benefits of inclusion of MRI data in epidemiological research into a condition with diverse causal networks and clinical manifestations.