Unmet environmental needs and unmet healthcare needs in a population of young adults with cerebral palsy: what the SPARCLE study tells us.

Introduction: Optimizing care for young adults with cerebral palsy is crucial for their physical and psychological well-being. The inadequacy of proximal environment may play a role in the provision of health services. The aim of this study is to explore the association between unmet environmental needs in the physical, social and attitudinal domains and unmet healthcare needs in four interventions: physiotherapy, occupational therapy, speech therapy and psychological counselling. Methods: Young adults with cerebral palsy were recruited in the SPARCLE3 European multicenter cross-sectional study. Healthcare needs and coverages were assessed using the Youth Health Care, Satisfaction, Utilization and Needs questionnaire. The need and availability of environmental factors in physical, social and attitudinal domains were collected using the European Adult Environment Questionnaire. Logistic regressions were conducted separately for each intervention to measure associations between unmet environmental needs and unmet healthcare needs. Results: We studied 310 young adults with cerebral palsy, with a mean age of 24.3 years; 37.4% could not walk independently, 51.5% had an IQ below 70, 34.2% had severe communication difficulties. The most commonly expressed need was physiotherapy (81.6% of participants). Unmet healthcare needs were reported by 20.9%, 32.4%, 40.3% and 49.0% of participants requiring physiotherapy, occupational therapy, psychological counselling and speech therapy, respectively. The physical environment was never significantly associated with unmet healthcare needs. In contrast, the social environment was significantly associated with unmet healthcare needs across all interventions, with odds ratios over 2.5, depending on the number of unmet needs and the nature of intervention needed. With regard to the attitudinal environment, when at least one unmet attitudinal environmental need was reported, the odds of also reporting an unmet healthcare need were of 3.68 for speech therapy and 3.77 for physiotherapy. The latter association was significant only for individuals with severe motor impairment. Discussion: Our results highlight the importance of the social and attitudinal environment in meeting healthcare needs in young adults with cerebral palsy. The lack of correlation between unmet healthcare needs and the physical environment suggests that it can be partly compensated for by social support.

Assessing the Adequacy of the Physical, Social, and Attitudinal Environment to the Specific Needs of Young Adults With Cerebral Palsy: The European Adult Environment Questionnaire.

OBJECTIVES: To present the development of the European Adult Environment Questionnaire (EAEQ), to assess to what extent it covers the International Classification of Functioning, Disability and Health (ICF), and to describe the adequacy of the physical, social, and attitudinal environment to the specific needs of young adults with cerebral palsy (CP). DESIGN: Cross-sectional. SETTING: Administrative regions in France, Germany, Italy, Portugal, and Sweden. PARTICIPANTS: Young adults with CP (N=357), with varying severity profiles, aged 19-28 years at time of interview (2018-20). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURE(S): Physical, social, and attitudinal environment unmet needs. RESULTS: Relevant environmental factors (EFs) for young adults with CP were identified during focus groups in England and Portugal. EFs were mapped to the ICF environmental classification and the EAEQ analytical structure resulted from this linking procedure. It comprised 61 items, linked to 31 ICF environmental classification categories, and covered 4 of its 5 chapters. Content validity assessed with the bandwidth index (percentage coverage of ICF Core Sets for adults with CP) was satisfactory (79.3%). A descriptive analysis was carried out. Participants had a mean age of 24 years, 56% were men, 38% had severely limited mobility. Less than 16% reported unmet needs for EFs relating to home, college/work/day placement, and communication in the Products and technology chapter. Unmet needs were higher (>20%) for the other items in the Public use and Land development categories. Social support, attitudes, and understanding of relatives were often adequate to the participants' needs. The proportion of unmet needs varied by sex (women were more often concerned) and raised with increasing gross motor impairment. CONCLUSION: The EAEQ describes in detail the adequacy of the environment to the specific needs of young adults with CP. Its ICF-based structure opens up possibilities for use in a universal conceptual framework.

Quality of life and mental health in emerging adults with cerebral palsy compared to the general population.

BACKGROUND: While evidence concerning Quality of Life (QoL) in youth with cerebral palsy (CP) in comparison to the general population has been accumulating, there is a lack of studies exploring differences on a wider range of positive and negative mental health outcomes in emerging adults. METHODS: This binational case control study is part of the SPARCLE cohort study on QoL and participation of youth with CP. QoL (WHOQOL-BREF), depression (PHQ-9), anxiety (GAD-7) and self-efficacy (GSE) were assessed in 198 emerging adults with CP and 593 emerging adults from the general population, matched for country of residence, age and gender. ANCOVAs with impairment and pain as covariates were run. RESULTS: Similar levels of QoL were found in both samples, except for the environmental domain, with better QoL for emerging adults with CP compared to the general population. There were significant descriptive differences regarding depression with worse levels in the CP sample, however, also worse levels of self-efficacy. Pain as a covariate had a significant negative impact on all measures, leading to poorer self-efficacy while worsening depression and anxiety; impairment had a significant worsening impact on physical QoL and self-efficacy only. CONCLUSION: Similar expressions of mental health outcomes in emerging adults with CP and the general population indicate the high adaptive capability of emerging adults with CP.

Quality of Life in Young Adults With Cerebral Palsy: A Longitudinal Analysis of the SPARCLE Study.

Introduction: While most people with cerebral palsy (CP) will have a life expectancy similar to that of the general population, international research has primarily focused on childhood and adolescence; and knowledge about the quality of life (QoL) of young adults with CP, its trajectories, and associated factors remains scarce. Methods: This longitudinal study included young adults with CP living in five European regions and who had previously participated in the SPARCLE cohort as children and/or adolescents. Their QoL in the psychological well-being and social relationships domains was estimated using age-appropriate validated instruments (KIDSCREEN-52 in childhood/adolescence and WHOQOL-Bref in young adulthood). We used generalized linear mixed-effect models with random intercept to estimate long-term trajectories of QoL in both domains and to investigate whether severity of impairment, pain, and seizure influenced these trajectories. We sought to identify potentially different trajectories of QoL from childhood to adulthood using a shape-based clustering method. Results: In total, 164 young adults with CP aged 22-27 years participated in the study. Psychological well-being linearly decreased by 0.78 points (scale 0-100) per year (95% confidence interval (CI) -0.99 to -0.56) from childhood to young adulthood, whereas QoL in the social relationships domain increased (ß coefficient 1.24, 95% CI 0.92-1.55). Severity of impairment was associated with reduced QoL in all life periods of the study (childhood, adolescence, and young adulthood): motor impairment with social relationships, and more nuancedly intellectual disability with psychological well-being and social relationships. At all periods, frequent pain significantly reduced psychological well-being, and seizures were associated with lower QoL in the social relationships domain. In both domains, we identified a group of individuals with CP who presented a reverse trajectory compared with the general QoL trajectory. Conclusion: Identification of QoL trajectories and their associated factors yields improved knowledge about the experience of individuals with CP until young adulthood. Further studies are needed to better understand the determinants that have the greatest influence on the different shapes of long-term trajectories of QoL.

Family history of cancer and the risk of childhood brain tumors: a pooled analysis of the ESCALE and ESTELLE studies (SFCE).

PURPOSE: Although some specific genetic syndromes such as neurofibromatosis (NF) have been identified as risk factor of childhood brain tumors (CBT), the potential role of inherited susceptibility in CBT has yet to be elucidated. METHODS: To further investigate this, we conducted a pooled analysis of two nationwide case-control studies ESCALE and ESTELLE. The mothers of 509 CBT cases and 3,102 controls aged under 15 years who resided in France at diagnosis/interview, frequency-matched by age and gender, responded to a telephone interview conducted by trained interviewers. Pooled odds ratio (OR) and 95% confidence intervals (95% CI) were estimated using unconditional logistic regression. RESULTS: CBT was significantly associated with the family history of cancer in relatives (OR 1.2, 95% CI 1.0-1.5). The OR was slightly higher for maternal relatives than for paternal relatives, and when at least two relatives had a history of cancer. CBT was significantly associated with a family history of brain tumor (OR 2.1, 95% CI 1.3-3.7). This association seemed stronger for first-degree relatives (mother, father, and siblings), for whom, by contrast, no association was seen for cancers other than CBT. No specificity by CBT subtypes or by age of the children were found for any of these findings. CONCLUSION: Our findings support the hypothesis of a familial susceptibility of CBT, not due to being a known NF carrier.

Maternal residential pesticide use during pregnancy and risk of malignant childhood brain tumors: A pooled analysis of the ESCALE and ESTELLE studies (SFCE).

Some previous epidemiological studies have suggested that pesticide exposure during pregnancy may have a possible role in the development of childhood brain tumors (CBT). We pooled data from two French national population-based, case-control studies to investigate the association between maternal residential use of pesticides during pregnancy and the risk of CBT. The mothers of 437 CBT cases and 3,102 controls aged under 15 years who resided in France at diagnosis/interview, frequency-matched by age and gender, answered a structured telephone interview conducted by trained interviewers. Unconditional logistic regression was used to estimate pooled odds ratio (OR) and 95% confidence intervals (95% CI). CBT was significantly associated with the maternal home use of pesticides during pregnancy (OR 1.4, 95% CI 1.2-1.8) and, more specifically, with insecticide (OR 1.4, 1.2-1.8). We could not draw any conclusions about herbicides and/or fungicides because few women used them during pregnancy and most of these mothers also used insecticides. Although potential recall bias cannot be excluded, our findings of this pooled analysis support the hypothesis that residential maternal use of pesticides during pregnancy and particularly insecticides may increase the risk of CBT. Future investigations to verify these findings and to explore for CBT subtypes and dose-response are necessary to have a better understanding of the possible role of pesticides in etiology of CBT.