Evaluation of RuralkidsGPS; A Novel Integrated Paediatric Care Coordination Model of Care in Rural Australia - a Mixed-Methods Study Protocol.

Introduction: The Kids Guided Personalised Service (KidsGPS) is an integrated model of care coordination for children and young people (CYP) living with medical complexity. After successful implementation in an urban setting, the model of care will be rolled-out at scale to four rural regions in New South Wales, Australia to establish RuralKidsGPS. This paper describes the approach and methods for the outcome and implementation evaluation of RuralKidsGPS. Description: The evaluation aims to assess health, economic and implementation outcomes and processes whilst identifying barriers and enablers to inform future rollouts. Measures of health service utilisation (primary outcome), child health related quality of life and parent/carer experiences will be assessed. The implementation evaluation will occur alongside the outcomes evaluation and is underpinned by the Consolidated Framework for Implementation Research and informed by validated quantitative measures and qualitative interviews with patients, families, healthcare providers and service managers. An economic analysis will determine incremental cost effectiveness ratios for the new model of care using health service utilisation data. Conclusion: RuralKidsGPS, if effective, has the potential to improve equity of access to integrated care for CYP and their families and this protocol may inform other evaluations of similar models of care delivered at scale.

Global prevalence of cerebral palsy: A systematic analysis.

AIM: To determine trends and current estimates in regional and global prevalence of cerebral palsy (CP). METHOD: A systematic analysis of data from participating CP registers/surveillance systems and population-based prevalence studies (from birth year 1995) was performed. Quality and risk of bias were assessed for both data sources. Analyses were conducted for pre-/perinatal, postnatal, neonatal, and overall CP. For each region, trends were statistically classified as increasing, decreasing, heterogeneous, or no change, and most recent prevalence estimates with 95% confidence intervals (CI) were calculated. Meta-analyses were conducted to determine current birth prevalence estimates (from birth year 2010). RESULTS: Forty-one regions from 27 countries across five continents were represented. Pre-/perinatal birth prevalence declined significantly across Europe and Australia (11 out of 14 regions), with no change in postneonatal CP. From the limited but increasing data available from regions in low- and middle-income countries (LMICs), birth prevalence for pre-/perinatal CP was as high as 3.4 per 1000 (95% CI 3.0-3.9) live births. Following meta-analyses, birth prevalence for pre-/perinatal CP in regions from high-income countries (HICs) was 1.5 per 1000 (95% CI 1.4-1.6) live births, and 1.6 per 1000 (95% CI 1.5-1.7) live births when postneonatal CP was included. INTERPRETATION: The birth prevalence estimate of CP in HICs declined to 1.6 per 1000 live births. Data available from LMICs indicated markedly higher birth prevalence. WHAT THIS PAPER ADDS: • Birth prevalence of pre-/perinatal cerebral palsy (CP) in high-income countries (HICs) is decreasing. • Current overall CP birth prevalence for HICs is 1.6 per 1000 live births. • Trends in low- and middle-income countries (LMICs) cannot currently be measured. • Current birth prevalence in LMICs is markedly higher than in HICs. • Active surveillance of CP helps to assess the impact of medical advancements and social/economic development. • Population-based data on prevalence and trends of CP are critical to inform policy.

Predictors of Rehabilitation Service Utilisation among Children with Cerebral Palsy (CP) in Low- and Middle-Income Countries (LMIC): Findings from the Global LMIC CP Register.

BACKGROUND: We assessed the rehabilitation status and predictors of rehabilitation service utilisation among children with cerebral palsy (CP) in selected low- and middle-income countries (LMICs). METHODS: Data from the Global LMIC CP Register (GLM-CPR), a multi-country register of children with CP aged <18 years in selected countries, were used. Descriptive and inferential statistics (e.g., adjusted odds ratios) were reported. RESULTS: Between January 2015 and December 2019, 3441 children were registered from Bangladesh (n = 2852), Indonesia (n = 130), Nepal (n = 182), and Ghana (n = 277). The proportion of children who never received rehabilitation was 49.8% (n = 1411) in Bangladesh, 45.8% (n = 82) in Nepal, 66.2% (n = 86) in Indonesia, and 26.7% (n = 74) in Ghana. The mean (Standard Deviation) age of commencing rehabilitation services was relatively delayed in Nepal (3.9 (3.1) year). Lack of awareness was the most frequently reported reason for not receiving rehabilitation in all four countries. Common predictors of not receiving rehabilitation were older age at assessment (i.e., age of children at the time of the data collection), low parental education and family income, mild functional limitation, and associated impairments (i.e., hearing and/or intellectual impairments). Additionally, gender of the children significantly influenced rehabilitation service utilisation in Bangladesh. CONCLUSIONS: Child's age, functional limitation and associated impairments, and parental education and economic status influenced the rehabilitation utilisation among children with CP in LMICs. Policymakers and service providers could use these findings to increase access to rehabilitation and improve equity in rehabilitation service utilisation for better functional outcome of children with CP.

Use of health services by preschool-aged children who are developmentally vulnerable and socioeconomically disadvantaged: testing the inverse care law.

AIM: The inverse care law suggests that those with the greatest need for services are least likely to receive them. Our aim of this study was to test the inverse care law in relation to the use of health services by children aged 4-5 years in Australia who were developmentally vulnerable and socioeconomically disadvantaged. METHOD: Cross-sectional data were collected from the Longitudinal Study of Australian Children birth cohort when the children were aged 4-5 years. Children were grouped according to the combination of developmental vulnerability (yes, no) and socioeconomic disadvantage (lower, higher), resulting in four groups (reference group: developmentally vulnerable and disadvantaged). Multivariate regression was used to examine the impact of the combination of developmental vulnerability and disadvantage on health service use, adjusting for other sociodemographic characteristics. RESULTS: 3967 (90%) of children had data on developmental vulnerability at 4-5 years. A third of children (32.6%) were classified as developmentally vulnerable, and 10%-25% of these children had used health services. Non-disadvantaged children who were developmentally vulnerable (middle need) had 1.4-2.0 times greater odds of using primary healthcare, specialist and hospital services; and non-disadvantaged children who were not developmentally vulnerable (lowest need) had 1.6-1.8 times greater odds of using primary healthcare services, compared with children who were developmentally vulnerable and disadvantaged (highest need). CONCLUSION: We found some evidence of the inverse care law. Equity in service delivery remains a challenge that is critically important to tackle in ensuring a healthy start for children.

Congenital Cytomegalovirus Prevention, Awareness and Policy Recommendations - A Scoping Study.

BACKGROUND: Congenital cytomegalovirus (cCMV) is known to cause childhood deafness, neurodevelopmental disability and death. Simple hygiene precautions are effective in reducing maternal risk of CMV infection. OBJECTIVE: To review i) awareness of CMV infection and available primary prevention strategies both in the community and amongst health professionals ii) available cCMV information sources in the literature, grey literature and published professional guidelines. METHODS: Scoping study to i) identify literature pertaining to cCMV awareness amongst parents and health professionals using MedLine and CINAHL databases via EBSCO ii) review one high income country's guidelines and recommendations regarding cCMV infection and pregnancy (example country Australia) iii) grey literature for parental information. RESULTS: Worldwide awareness of cCMV and of available prevention strategies amongst women and health professionals are poor. Findings internationally suggest at least half of maternity care health professionals do not routinely provide advice to women regarding simple hygiene precautions that can reduce their risk of infection during pregnancy. Though information resources regarding cCMV are available, they are frequently not included within general healthy pregnancy advice and require individuals to search for 'congenital cytomegalovirus'. CONCLUSION: cCMV is a preventable cause of serious congenital disability and death. Prevention opportunities are being missed because most women are not aware of cCMV or how to reduce their risk of infection in pregnancy, in part due to poor health professional awareness. New strategies to disseminate cCMV information to the community and to support health professionals to embed cCMV advice within routine pregnancy counselling is required.

Hip dysplasia among children with spastic cerebral palsy in rural Bangladesh.

BACKGROUND: Hip dysplasia is common among children with cerebral palsy (CP), particularly in spastic CP. It can result in pain, reduced function and quality of life. However, the burden of hip dysplasia among children with CP in llow-and middle-income countries (LMICs) like Bangladesh is unknown. We aimed to define the burden of hip dysplasia among children with spastic CP in Bangladesh. METHODS: This study includes a subset of the Bangladesh CP Register (BCPR) study cohort who were registered between January and March 2015. The BCPR is a population-based surveillance of children with CP (aged < 18 years) operating in a northern sub-district (Shahjadpur; child population ~ 226,114) of Bangladesh. Community-based key informant's method (KIM) survey conducted to identify children with CP in the surveillance area. A diagnosis of CP was made based on clinical history and examination by the study physicians and physiotherapist. Study participants had an antero-posterior (AP) X-ray of their pelvis. The degree of subluxation was assessed by calculating the migration percentage (MP). RESULTS: During the study period, 196 children with CP were registered, 144 had spastic CP. 40 children with spastic CP (80 hips) had pelvic X-Rays (mean age 9.4 years, range 4.0-18.0 years) and 32.5% were female. Gross Motor Function Classification System (GMFCS) showed 37.5% (n = 15) with GMFCS level I-II and 62.5% (n = 25) with GMFCS level III-V. Twenty percent (n = 8) of the children had hip subluxation (MP: 33-80%). Osteopenic changes were found in 42.5% (n = 17) children. CONCLUSIONS: To the best of our knowledge this is one of the first studies exploring hip dysplasia among children with spastic CP in Bangladesh. Our findings reflect that hip dysplasia is common among children with spastic CP. Introduction of hip surveillance programmes is imperative for prevention of secondary complications, reduced function and poor quality of life among these children.

Impact of social disadvantage on cerebral palsy severity.

AIM: To investigate the impact of socio-economic disadvantage on indicators of cerebral palsy (CP) severity - motor impairment, intellectual disability, and the presence of severe comorbidities - in children with CP in Australia. METHOD: Data from the Australian Cerebral Palsy Register were analysed. Socio-economic disadvantage was assessed using maternal age, maternal country of birth, and a measure of neighbourhood socio-economic status (SES) at the time of the child's birth. Descriptive bivariate analysis, trend analysis, risk ratios, and mediation analysis were undertaken to examine the impact of disadvantage on the indicators of CP severity. RESULTS: A socio-economic gradient was demonstrated with an increasing proportion of children with non-ambulant status, at least moderate intellectual disability, and the presence of severe comorbidities (having epilepsy, functional blindness, bilateral deafness, and/or no verbal communication) with decreasing neighbourhood SES, adolescent motherhood, and maternal minority ethnicity. INTERPRETATION: In Australia, socio-economic disadvantage at birth impacts adversely on CP severity at age 5 years. By identifying that socio-economically disadvantaged children with CP are at greater risk of more severe functional outcomes, we can inform targeted interventions at the family and neighbourhood level to reduce these inequities for children with CP. WHAT THIS PAPER ADDS: Socio-economic disadvantage is associated with increased severity of cerebral palsy functional outcomes. This encompasses low neighbourhood socio-economic status, adolescent motherhood, and maternal minority ethnicity.

IMPACTO DE LA DESVENTAJA SOCIAL SOBRE LA SEVERIDAD DE LA PARÁLISIS CEREBRAL: OBJETIVO: Investigar el impacto de la desventaja socioeconómica en los indicadores de severidad de la parálisis cerebral (PC), definida como - deterioro motor, discapacidad intelectual y la presencia de severidad de las comorbilidades: en niños con PC en Australia. MÉTODO: Se analizaron los datos del Australian Cerebral Palsy Register. La desventaja socioeconómica se evaluó utilizando la edad materna, el país de nacimiento de la madre, y una medida de estado socioeconómico del vecindario (SES) en el momento del nacimiento del niño. Se realizaron análisis bivariados, análisis de tendencias, índices de riesgo y análisis de mediación para examinar el impacto de la desventaja en los indicadores de severidad PC. RESULTADOS: Se demostró un gradiente socioeconómico con una proporción creciente de niños con estado no ambulante, al menos discapacidad intelectual moderada, y la presencia de comorbilidades graves (con epilepsia, ceguera funcional, sordera bilateral y / o sin comunicación verbal) con SES vecinales, maternidad adolescente, y etnia de la minoría materna. INTERPRETACIÓN: En Australia, la desventaja socioeconómica al nacer tiene un impacto adverso en la severidad de PC a la edad de 5 años. Al identificar a los niños con desventajas socioeconómicas con PC tienen un mayor riesgo de resultados funcionales más severos. Con esta información podemos guiar intervenciones a nivel familiar y de vecindario para reducir estas inequidades en los niños con PC.

IMPACTO DA DESVANTAGEM SOCIAL NA SEVERIDADE DA PARALISIA CEREBRAL: OBJETIVO: Investigar o impacto da desvantagem sócio-econômica nos indicadores de severidade da paralisia (PC) - comprometimento motor, deficiência intelectual, e a presença de comorbidades graves - em crianças com PC na Austrália. MÉTODO: Dados do Registro Austrliano de Paralisia Cerebral foram analisados. Desvantagens sócio-econômicas foram avaliadas usando-se a idade materna, o país de Nascimento da mãe, e uma medida do estado sócio-econômico (ESE) da vizinhança no momento do nascimento da criança. Análise bivariada descritiva, análise de tendências, taxas de risco, e análise de mediação foram realizados para examinar o impacto da desvantagem nos indicadores da severidade da PC. RESULTADOS: Um gradiente sócio-econômico foi demonstrado com um aumento da proporção de crianças com situação não ambilante, deficiência intelectual pelo menos moderada, e presença de comorbidades severas (epilepsia, cegueira funcional, surdez bilateral, e/ou nenhuma comunicação verbal) quanto menor o ESE da vizinhança, a minoria étnica materna e maternidade na adolescência. INTERPRETAÇÃO: Na Austrália, a desvantagem sócio-econômica ao nascimento impacta adversamente a severidade da PC na idade de 5 anos. Ao identificar que crianças com PC em desvantagem sócio-econômica têm maior risco de resultados funcionais mais severos, podemos informar intervenções específicas voltadas para a família e para a vizinhança para reduzir o nível destas inequidades para crianças com PC.

Predicting equipment needs of children with cerebral palsy using the Gross Motor Function Classification System: a cross-sectional study.

BACKGROUND: Children with cerebral palsy (CP) routinely use assistive equipment to improve their independence. Specialist equipment is expensive and therefore not always available to the child when needed. AIM: The aim of this study was to determine whether the assistive equipment needs of children with CP and the associated costs could be predicted. METHOD: A cross-sectional study using a chart audit was completed. Two hundred forty-two children met eligibility criteria and were included in the study. Data abstracted from files pertained to the child's CP, associated impairments and assistive equipment prescribed. The findings were generated using linear regression modelling. RESULTS: Gross Motor Function Classification System (GMFCS) level [B = 3.01 (95% CI, 2.36-3.57), p = 0.000] and the presence of epilepsy [B = 2.35 (95% CI, 0.64-4.06), p = 0.008] predicted the prescription of assistive equipment. The more severely affected the gross motor function impairment, the more equipment that was required and the more the equipment cost. INTERPRETATION: The equipment needs of children with CP can be predicted for the duration of childhood. This information may be useful for families and for budget and service planning.