Prioritized strategies to improve diagnosis and early management of cerebral palsy for both Maori and non-Maori families.

AIM: To identify prioritized strategies to support improvements in early health service delivery around the diagnosis and management of cerebral palsy (CP) for both Maori and non-Maori individuals. METHOD: Using a participatory approach, health care professionals and the parents of children with CP attended co-design workshops on the topic of early diagnosis and management of CP. Health design researchers facilitated two 'discovery' (sharing experiences and ideas) and two 'prototyping' (solution-focused) workshops in Aotearoa, New Zealand. A Maori health service worker co-facilitated workshops for Maori families. RESULTS: Between 7 and 13 participants (14 health care professionals, 12 parents of children with CP across all functional levels) attended each workshop. The discovery workshops revealed powerful stories about early experiences and needs within clinician-family communication and service provision. The prototyping workshops revealed priorities around communication, and when, what, and how information is provided to families; recommendations were co-created around what should be prioritized within a resource to aid health care navigation. INTERPRETATION: There is a critical need for improved communication, support, and guidance, as well as education, for families navigating their child with CP through the health care system. Further input from families and health care professionals partnering together will continue to guide strategies to improve health care service delivery using experiences as a mechanism for change.

Incidence of Cerebral Palsy in Korea and the Effect of Socioeconomic Status: A Population-Based Nationwide Study.

PURPOSE: Cerebral palsy (CP) is a neurodevelopmental disorder that causes serious disability. Prematurity and low birth weight (LBW) are known to be the strongest risk factors of CP. While socioeconomic status (SES) has been found to influence the occurrence of CP, prematurity, and LBW, no studies have investigated this effect in Korea. The aim of this study was to evaluate the incidence of CP, prematurity, and LBW in Korea, as well as the effect of SES thereon. MATERIALS AND METHODS: Data were obtained from the National Health Information Database from 2007 to 2013; persons with a history of CP, prematurity, and LBW were investigated by year. SES was defined in accordance with income quintiles, birth regions, and coverage classification. RESULTS: The incidence of CP decreased over the last five years, despite increased rates of prematurity and LBW. CP incidence was significantly lower in affluent groups than in the most deprived group, although this difference disappeared after controlling for confounders. The incidence of CP was significantly higher in medical aid beneficiaries, even after controlling for confounders. CONCLUSION: CP incidence in Korea has decreased over the last five years, despite an increase in high-risk deliveries. Income level had no effect in CP incidence. These results may aid CP management and prevention policies.

Association of COL4A1 gene polymorphisms with cerebral palsy in a Chinese Han population.

The basement membrane (BM) is an extracellular matrix associated with overlying cells and is important for proper tissue development, stability, and physiology. COL4A1 is the most abundant component of type IV collagen in the BM, and COL4A1 variants can present with variable phenotypes that might be related to cerebral palsy (CP). We postulated, therefore, that variations in the COL4A1 gene might play an important role in the etiology of CP. In this study, six single nucleotide polymorphisms (SNPs) in the COL4A1 gene were genotyped among 351 CP patients and 220 healthy controls from the Chinese Han population. Significant association was found for an association between CP and rs1961495 (allele: p = 0.008, odds ratio (OR) = 1.387, 95% confidence interval (CI) = 1.088-1.767) and rs1411040 (allele: p = 0.009, OR = 1.746, 95% CI = 1.148-2.656) SNPs of the COL4A1 gene. Multifactor dimensionality reduction analysis suggested that these SNPs had interactive effects on the risk of CP. This study is the first attempt to investigate the contribution of polymorphisms in the COL4A1 gene to the susceptibility of CP in a Chinese Han population. This study shows an association of the COL4A1 gene with CP and suggests a potential role of COL4A1 in the pathogenesis of CP.

Measles imported to the United States by children adopted from China.

In July 2013, the National Immunization Program of China was notified by the US Centers for Disease Control and Prevention that measles was detected in 3 newly adopted, special needs children with cerebral palsy (CP) from China. We report an investigation of measles transmission in China that led to infection of these children. Interviews were conducted with welfare institute staff and panel physicians; health records of the potentially exposed population were reviewed; and immunization coverage was assessed among institute residents. Five residents with CP, all unvaccinated against measles, among who were the 3 adoptees, were linked epidemiologically into 3 generations of measles transmission antecedent to the US outbreak. In a random sample of residents, first dose of measles containing vaccine (MCV1) and MCV2 coverage was 16 of 17 (94%) and 7 of 11 (64%) among children with CP, and 100% (32 of 32) and 96% (21 of 22) among children without CP. Vaccinators reported reluctance to vaccinate children with CP because the China pharmacopeia lists encephalopathy as a contraindication to vaccination. Panel physicians reported to investigators no necessity of vaccination for adoptees to the United States if US parents sign an affidavit exempting the child from vaccination. We recommend that the China pharmacopeia vaccine contraindications be reviewed and updated, the United States should reconsider allowing vaccination exemptions for internationally adopted children unless there are true medical contraindications to vaccination, and US pediatricians should counsel adopting parents to ensure that their child is up-to-date on recommended vaccinations before coming to the United States.

The role of socio-economic status and perinatal factors in racial disparities in the risk of cerebral palsy.

AIM: To determine whether racial disparities in cerebral palsy (CP) risk among US children persist after controlling for socio-economic status (SES) (here indicated by maternal education) and perinatal risk factors. METHOD: A population-based birth cohort study was conducted using the Autism and Developmental Disabilities Monitoring Network surveillance and birth data for 8-year-old children residing in multi-county areas in Alabama, Georgia, Missouri, and Wisconsin between 2002 and 2008. The birth cohort comparison group included 458 027 children and the case group included 1570 children with CP, 1202 with available birth records. χ(2) tests were performed to evaluate associations and logistic regression was used to calculate relative risks (RR) and adjusted odds ratios (OR) with 95% confidence intervals (CI). RESULTS: The risk of spastic CP was more than 50% higher for black versus white children (RR 1.52, 95% CI 1.33-1.73), and this greater risk persisted after adjustment for SES (OR 1.35, 95% CI 1.18-1.55), but not after further adjustment for preterm birth and size for gestational age. The protective effect of maternal education remained after adjustment for race/ethnicity and perinatal factors. INTERPRETATION: Maternal education appears to independently affect CP risk but does not fully explain existing racial disparities in CP prevalence in the US.

Transcultural adaptation and validation of the Korean version of Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILD).

PURPOSE: The aim of this study was to translate and transculturally adapt the Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILD) questionnaire into Korean language, and to test the reliability and validity, including the internal consistency, known-group validity and factor analysis of the Korean version of the CPCHILD. METHODS: A Korean version of CPCHILD was produced according to internationally accepted guidelines. For validity testing, 194 consecutive parents or caregivers of children with cerebral palsy (CP) were recruited and completed the questionnaire. Internal consistency, test-retest reliability, and known-groups validity were evaluated and factor analysis was performed to validate the Korean version of the CPCHILD. RESULTS: In terms of internal consistency, a Cronbach's alpha was above 0.90 in all domains of the CPCHILD (range 0.921 to 0.966), except the 5th domain (0.628). In terms of known-groups validity, the total score of the CPCHILD was significantly different according to the Gross Motor Function Classification System (GMFCS) level (p < 0.001). Intra-class correlation coefficient spanned from 0.517 to 0.801. Factor analysis showed that the five-factor solution of the CPCHILD explained 76.7% of the variance with 59.0, 6.5, 5.1, 4.2 and 3.2% of variance by each components number. CONCLUSIONS: The Korean version of CPCHILD was found to be a reliable and valid questionnaire of caregivers' perspectives on the health-related quality of life in severely affected children with CP. However, the Korean version of CPCHILD contains some redundant items, and factor analysis suggested a five-domain questionnaire. Implication for Rehabilitation The Korean version of CPCHILD is a reliable, internally consistent, valid instrument for assessing the health-related quality of life in severely affected children with CP from the perspective of caregivers. After the transcultural adaptation and validation of the Korean CPCHILD, it can be reliably used in clinical and research settings to evaluate the health-related quality of life in Korean patients with CP.

Disparities in Canadian indigenous health research on neurodevelopmental disorders.

OBJECTIVE: To map the landscape of research on autism (ASD), cerebral palsy (CP), and fetal alcohol spectrum disorder (FASD) in Canadian Aboriginal children. METHOD: The authors used a detailed search strategy to identify and access publications on ASD, CP, and FASD involving Canadian Aboriginal children, families, and communities from online databases. They analyzed these materials for the type of research, stated objectives, methodologies, and the level of engagement of Aboriginal Peoples. RESULTS: The authors found a total of 52 reports published since 1981 relevant to Aboriginal children. Of these, 51 focused exclusively on FASD. They also found a near-complete failure to acknowledge community involvement in research decisions or dissemination of results in any of the publications. CONCLUSIONS: The focus on FASD in Aboriginal children and the absence of research on the other 2 major childhood disorders are at odds with rates of these disorders across Canadian children. The authors argue that this trend violates fundamental principles ensuring equitable representation of all children regardless of background in research and access to benefits of research in health care and perpetuates stigma in an already marginalized population.

The association between GAD1 gene polymorphisms and cerebral palsy in Chinese infants.

Studies suggest that GAD1 gene was a functional candidate susceptibility gene for cerebral palsy (CP). In order to investigate the contribution of GAD1 gene to the etiology of CP in Chinese infants, we carried out a case-control association study between GAD1 gene and CP. In this study, 374 health controls and 392 infants with CP were recruited. Genomic DNA was extracted from venous blood and all three single nucleotide polymorphisms in GAD1 (rs3791874, rs3791862 and rs16858977) were genotyped by Sequenom's MassARRAY system. There were no significant differences in allele or genotype frequencies between CP or mixed CP patients and controls at any of the three genetic polymorphisms. Through haplotype analysis we found that haplotype GG (rs3791862, rs16858977) frequency demonstrated significantly statistical difference between mixed CP patients and controls (p = 0.0371). Our positive findings of haplotype GG suggested that variation of GAD1 gene was an important risk factor for mixed CP.

Disparities in obstetrical outcomes in ART pregnancies compared with natural conceptions.

Health disparities are observed in all fields of medicine and reproductive health is not immune to this phenomenon. The incidence of women using infertility treatments to conceive is increasing. Women undergoing assisted reproduction appear to be at increased risk of adverse outcomes, and minority women tend to be at even greater risk. This article examines several adverse obstetrical outcomes including preterm birth, congenital malformations, and preeclampsia among women receiving infertility treatments compared with those who conceive spontaneously. It will further examine societal costs associated with these procedures.

The role of migration and choice of denominator on the prevalence of cerebral palsy.

AIM: Differential migration and choice of denominator have been hypothesized to contribute to differences between period prevalence and birth prevalence of cerebral palsy (CP). The purpose of this study was to evaluate the effects of migration and choice of denominator on the prevalence of CP. METHOD: Data from the Metropolitan Atlanta Developmental Disabilities Surveillance Program and census and birth certificate files were used to calculate various CP prevalence estimates for 2000. RESULTS: The overall CP period prevalence was 3.2 (95% confidence interval [CI] 2.7-3.8) per 1000 8-year-olds and was similar for those born in Atlanta who resided there at age 8 years (3.3; 95% CI 2.7-4.1) and those born outside Atlanta who moved into Atlanta by age 8 years (3.0; 95% CI 2.3-3.9). CP prevalence in these two migration strata was similar by sex and race/ethnicity. CP birth prevalence of 8-year-olds in Atlanta in 2000 was 2.0 (95% CI 1.6-2.5) per 1000 live births in 1992. INTERPRETATION: The authors found no evidence to support the hypothesis that differential in-migration explained higher period than birth prevalence of CP in Atlanta. Comparability of CP prevalence across geographic areas will be enhanced if future studies report both period and birth prevalence.

The PEDALS stationary cycling intervention and health-related quality of life in children with cerebral palsy: a randomized controlled trial.

AIM: The aim of this study was to assess health-related quality of life (HRQOL) following a stationary cycling intervention in children with cerebral palsy (CP). METHOD: This was a phase I multisite randomized controlled trial with single blinding. HRQOL was evaluated using the Pediatric Quality of Life Inventory SF15 (PedsQL; children) and Pediatric Outcomes Data Collection Instrument (PODCI; parent proxy) before and after a 3-month stationary cycling intervention. Sixty-two children (29 male, 33 female; mean age 11y; range 7-18y) with spastic diplegic CP, classified as levels I to III on the Gross Motor Function Classification System, were enrolled. Paired and independent t-tests were used to evaluate within- and between-group differences respectively. RESULTS: Between-group differences, favoring the cycling group, were found for PedsQL emotional functioning (p=0.046) and Parental PODCI treatment expectations scores (p=0.006). Between-group differences were not found for other scales. Within-group improvements were found in the cycling group: PedsQL total score (+5.8; p=0.006), psychosocial health summary (+6.9; p=0.008), and school functioning (+8.0; p=0.038). PODCI satisfaction with symptoms decreased significantly only in the control group (-12.0; p=0.046). INTERPRETATION: A beneficial influence of exercise on pediatric emotional well-being and parental treatment expectations was found. The evidence was not strong for other aspects of HRQOL. Results support the positive relationship between physical fitness and emotional well-being in the general population. A child's perception is important when examining change in his or her emotional well-being due to intervention.

Correlação entre a gravidade da paralisia cerebral e a qualidade de vida de crianças, adolescentes e seus cuidadores / Correlation between severity of cerebral palsy and quality of life of children, adolescents and their caregivers

Introdução: A paralisia cerebral (PC) é caracterizada por um distúrbio não progressivo que interfere na postura e no movimento, podendo repercutir negativamente na qualidade de vida (QV), tanto dos indivíduos acometidos quanto de seus cuidadores. Objetivos: O objetivo deste estudo foi verificar se existe correlação entre a gravidade da PC e a) a QV de crianças/adolescentes e b) a QV de seus cuidadores. Métodos: Trata-se de um estudo transversal, descritivo e exploratório. A QV de crianças e adolescentes, com diagnóstico comprovado de PC, foi avaliada por meio do CHQ-PF50 e a gravidade da PC, classificada de acordo com o GMFCS. A QV dos cuidadores foi avaliada por meio do SF-36. Os participantes do estudo foram recrutados em quatro instituições da Grande Belo Horizonte, MG. Resultados: Foram incluídos no estudo 88 crianças/adolescentes com PC, com idade média de 8,94 + 4,4 anos, bem como um de seus cuidadores primários (75 mães e 13 pais, com idade média de 30,6 + 6,7). O coeficiente de correlação de Spearman demonstrou correlação estatisticamente significativa, entretanto, de fraca a regular (r<0.4, p<0.05), entre a gravidade do acometimento e o domínio ?função física? do CHQ-PF50 e entre a gravidade do acometimento e o domínio ?dor? do SF-36. Conclusões: Os resultados deste estudo sugerem que a QV de crianças e adolescentes com PC e a QV de seus cuidadores parecem ser parcialmente influenciadas pela gravidade da patologia.

Cerebral palsy among Asian ethnic subgroups.

OBJECTIVE: Asians have a reduced risk for cerebral palsy (CP) compared with whites. We examined whether individual Asian subgroups have a reduced risk of CP and whether differences in sociodemographic factors explain disparities in CP prevalence. METHODS: In a retrospective cohort of 629 542 Asian and 2 109 550 white births in California from 1991 to 2001, we identified all children who qualified for services from the California Department of Health Services on the basis of CP. Asians were categorized as East Asian (Chinese, Japanese, Koreans), Filipino, Indian, Pacific Islander (Guamanians, Hawaiians, and Pacific Islanders), Samoan, or Southeast Asian (Cambodian, Laotian, Thai, Vietnamese). RESULTS: Overall, CP prevalence was lower in Asians than whites (1.09 vs 1.36 per 1000; relative risk = 0.80, 95% confidence interval [CI] = 0.74-0.87) and ranged from 0.61/1000 in Thai children to 2.08/1000 in Samoan children. Several Asian subgroups had low risk profiles with respect to maternal age, educational attainment, and birth weight. However, after we adjusted for maternal age and education, infant gender, and birth weight, the adjusted risk of CP remained lower in East Asians (odds ratio [OR] = 0.75, 95% CI = 0.65-0.87), Filipinos (OR = 0.87, 95% CI = 0.75-0.99), Indians (OR = 0.59, 95% CI = 0.44-0.80), Pacific Islanders (OR = 0.62, 95% CI = 0.40-0.97), and Southeast Asians (OR = 0.68, 95% CI = 0.57-0.82) compared with whites. CONCLUSIONS: Most Asian national origin subgroups have a lower rate of CP than whites, and this disparity is unexplained. Additional studies that focus on the cause of ethnic disparities in CP may provide new insights into pathogenesis and prevention.

Family and quality of life: key elements in intervention in children with cerebral palsy.

Modern thinking about children's health, as embodied in the framework of the World Health Organization's International Classification of Functioning, Disability and Health - Child and Youth Version, requires that we be attentive to the 'context' of children's lives, namely their families and the well-being of their families. Family-centred services provide both a guide to the 'processes' of service by service providers and measurable evidence-based outcomes that link better 'processes' with better parental 'outcomes'. This brief paper provides an overview of these topics, arguing that the themes we address in services, and the way we do that, can have important effects on families, and by extension, on their children.

Measuring care and comfort in children with cerebral palsy: the care and comfort caregiver questionnaire.

OBJECTIVE: To evaluate the psychometric properties of the Care and Comfort Caregiver Questionnaire (CareQ), which was developed to measure the perceived effort of caregivers in providing care for children with moderate to severe cerebral palsy (CP). DESIGN: Cross-sectional data collection from a representative sample of a large racially/ethnically diverse geographic region. SETTING: Outpatient CP clinics at a metropolitan rehabilitation institution. PARTICIPANTS: A total of 100 primary caregivers of children with CP whose Gross Motor Function Classification System (GMFCS) levels were III-V. METHODS: The CareQ was administered to primary caregivers of children with CP. Internal consistency of the CareQ and its 3 domains (Personal Care, Positioning/Transfers, and Comfort) were evaluated with the Cronbach α. Construct validity of the CareQ was evaluated by its correlation with the Pediatric Functional Independence Measure (WeeFIM). Associations between CareQ scores and child and caregiver characteristics were assessed. RESULTS: Mean CareQ scores for children with GMFCS levels III, IV, and V were 30.6, 42.8, and 45.1, respectively (P < .01). The Cronbach α was 0.90 for total CareQ and 0.93, 0.80, and 0.82 for its Personal Care, Positioning/Transfers, and Comfort domains, respectively. Total CareQ and WeeFIM scores were negatively correlated (r = -.22; P = .03). Total CareQ scores were positively correlated with the child's age (r = .38; P < .01) and with body weight (r =.37; P < .01); however, no caregiver characteristics were associated with CareQ scores. CONCLUSIONS: The CareQ is a concise and internally consistent measure of difficulty of care as perceived by caregivers of children whose GMFCS levels were III-V. Further investigation should include assessment of responsiveness of the CareQ to changes in the child's functional status over time and/or with rehabilitation interventions.

Racial disparities in health-related quality of life in a cohort of very low birthweight 2- and 3-year-olds with and without cerebral palsy.

We examine racial differences in health-related quality of life (HRQoL) among 2- and 3-year-olds born very low birthweight (VLBW, <1500 g). The sample included 611 children (290 males and 321 females) from the Newborn Lung Project, a cohort of VLBW infants hospitalized in Wisconsin's newborn intensive care units during 2003 to 2004. Of the 611 children, 14% (86/611) were black, non-Hispanic and 86% (525/611) were white, non-Hispanic and 4% (23/611) had cerebral palsy. HRQoL was measured using the Pediatric Quality of Life Inventory. Black children scored nearly four points lower (mean difference -3.6, 95% confidence interval [CI]: -6.9 to -0.3) on HRQoL than their white peers. Cerebral palsy is associated with lower HRQoL (mean difference -24.4, 95% CI: -29.3 to -19.5), especially among black children, but does not explain racial differences in HRQoL. Living in a neighborhood with lowest levels of female education is associated with lower HRQoL (mean difference -5.6, 95% CI: -9.2 to -2.1), but does not explain the racial difference in HRQoL.

Parental views from rural Cambodia on disability causation and change.

PURPOSE: This study explored the beliefs of Cambodian parents of children with cerebral palsy regarding disability causation and their perceptions of the effectiveness of interventions in bringing about change in their child. RESULTS: Beliefs around disability causation were mixed, with equal numbers of participants attributing their child's disability to biomedical causes as to traditional causes incorporating elements of Theravada Buddhism, animism and Brahmanism. While many participants had initially sought traditional interventions for their child, few found them to be effective and most had subsequently utilised medical and rehabilitation services. Parents whose children were moderately or severely impaired perceived both traditional interventions and rehabilitation to be less effective than parents of children with mild impairments. Participants generally judged the effectiveness of interventions based on functional changes in their child. CONCLUSIONS: The complexity of Khmer belief systems was reflected in the range of participants' perceptions of the cause of their child's disability, yet beliefs around disability causation did not appear to have determined their care-seeking behaviour or their perceptions of effectiveness of interventions.

Racial, ethnic, and socioeconomic disparities in the prevalence of cerebral palsy.

OBJECTIVE: Racial and ethnic disparities in cerebral palsy have been documented, but the underlying mechanism is poorly understood. We determined whether low birth weight accounts for ethnic disparities in the prevalence of cerebral palsy and whether socioeconomic factors impact cerebral palsy within racial and ethnic groups. METHODS: In a retrospective cohort of 6.2 million births in California between 1991 and 2001, we compared maternal and infant characteristics among 8397 infants with cerebral palsy who qualified for services from the California Department of Health Services and unaffected infants. RESULTS: Overall, black infants were 29% more likely to have cerebral palsy than white infants (relative risk: 1.29 [95% confidence interval: 1.19-1.39]). However, black infants who were very low or moderately low birth weight were 21% to 29% less likely to have cerebral palsy than white infants of comparable birth weight. After we adjusted for birth weight, there was no difference in the risk of cerebral palsy between black and white infants. In multivariate analyses, women of all ethnicities who did not receive any prenatal care were twice as likely to have infants with cerebral palsy relative to women with an early onset of prenatal care. Maternal education was associated with cerebral palsy in a dose-response fashion among white and Hispanic women. Hispanic adolescent mothers (aged <18 years) had increased risk of having a child with cerebral palsy. CONCLUSIONS: The increased risk of cerebral palsy among black infants is primarily related to their higher risk of low birth weight. Understanding how educational attainment and use of prenatal care impact the risk of cerebral palsy may inform new prevention strategies.