Impairments and comorbidities in adults with cerebral palsy and spina bifida: a meta-analysis.

Introduction: Aging with a childhood-onset disability, such as cerebral palsy (CP), spina bifida (SB), and muscular diseases (MD), comes along with significant impairments and comorbidities. Despite the increasing evidence an overall picture is lacking. This study aimed to review the literature about adults with CP/SB/MD and impairments and comorbidities to perform a meta-analysis. Materials and methods: Embase, PubMed, Cinahl, and Google Scholar were searched (2000-2020). Search terms included adults with one of the aforementioned disabilities combined with impairments and comorbidities. If specific impairments or comorbidities were reported by at least four studies, these were included in the study. Pooled prevalence (95% Confidence Interval) of impairments/comorbidities were calculated. Results: The search yielded 7,054 studies of which 95 were included in the meta-analysis (64 CP, 31 SB, 0 MD). In total estimates were calculated for 26 (CP) and 11 (SB) outcomes. In adults with CP, pain [56.4% (95%CI 48.8-63.8)], deformities [44.2% (95%CI 12.9-78.4)], intellectual disability [37.2% (95%CI 26.7-48.3)], and fatigue [36.9% (95%CI 24.6-50.1)] were most prevalent; renal disease [3.0% (95%CI 2.1-4.2)] and stroke/rheumatic diseases {4.8% (95%CI 3.4-6.5; 4.8% (95%CI 1.5-9.9)] respectively} were least prevalent. For adults with SB, bladder incontinence [60.0% (95%CI 50.5-69.2)], bowel incontinence [49.2% (95%CI 34.5-64.0)], pain [44.1% (95%CI 27.4-61.5)], and sleeping problems [30.3% (95%CI 4.7-65.8)] were most prevalent; diabetes [4.8% (95%CI 2.8-7.3)] and renal disease [8.7% (95%CI 2.0-19.9)] were least prevalent. The included studies showed large heterogeneity. Conclusions: More research is needed to study health issues in adults with MD. Adults with CP or SB deal with a variety of health issues. More attention for the mental health of these adults is needed. There also is a need for accessible and adequate screening, preventive measures and clinical follow-up.

Stress and strain: a qualitative study into the impact of having relatives with addiction problems on students' health and daily lives.

PURPOSE: To explore the impact of having relatives with addiction problems on students' health, substance use, social life, and cognitive functioning, and to establish possible contributions of the participants' gender, type of relationship, and type of addiction of the relative(s). METHODS: A qualitative, cross-sectional study of semi-structured interviews with thirty students from a University of Applied Sciences in the Netherlands who had relatives with addiction problems. RESULTS: Nine major themes were identified: (1) violence; (2) death, illness, and accidents of relatives; (3) informal care; (4) perception of addiction; (5) ill health, use of alcohol and illegal drugs; (6) financial problems; (7) pressured social life; (8) affected cognitive functioning, and(9) disclosure. CONCLUSIONS: Having relatives with addiction problems severely affected the life and health of participants. Women were more likely to be informal carers, to experience physical violence, and to choose a partner with addiction problems than men. Conversely, men more often struggled with their own substance use. Participants who did not share their experiences reported more severe health complaints. It was impossible to make comparisons based on the type of relationship or type of addiction because participants had more than one relative or addiction in the family.

Effect evaluation of a vocational rehabilitation program for young adults with chronic physical conditions at risk for unemployment: A controlled clinical trial.

BACKGROUND: The work participation of young adults with chronic physical conditions lag behind compared to healthy age-mates. 'At Work' is a vocational rehabilitation intervention provided by occupational therapists, that supports them for entering the competitive labour market after graduating post-secondary education. AIM: To evaluate the effects of 'At Work' on self-efficacy, work-ability and employment status as compared to usual care. MATERIALS AND METHODS: In total, 88 young adults were included in a multicentre controlled trial; 49 entered 'At Work', 39 received usual care. GEE-analyses were applied. RESULTS: Scores on all outcome measures substantially improved over time in the intervention group, but no significant effects were found as compared to the control group. The effect on general self-efficacy showed a positive trend in favour of the intervention group. CONCLUSIONS AND SIGNIFICANCE: Unlike previous study results pointing to positive outcomes of At Work', the current study did not support the effectiveness of this program on work-related self-efficacy, work-ability and paid employment, as compared to usual care. Yet, we did find an indication for positive intervention effect on general self-efficacy, which is known to be an important capacity to achieve social participation.

Impact of the COVID-19 pandemic on students with relatives with addiction problems: A longitudinal qualitative study.

Addiction problems impact not only the persons with these problems but also family members. This study aims to examine the impact of the COVID-19 pandemic on stress, strain on health, study experiences, coping strategies, and access to support of students with relatives with addiction problems. Thirty students, aged 18-30 years, from a University of Applied Sciences in the Netherlands participated in a three-year qualitative longitudinal interview study. One round of individual semi-structured interviews was conducted before the COVID-19 pandemic, and three during the COVID-19 pandemic. Directed Content Analysis was applied, using the Stress-Strain-Information-Coping-Support-model. Four major themes were identified: (1) Increase in stress and strain; (2) Decrease in stress and strain; (3) Coping strategies, and (4) Access to social, professional, and educational support. Before the pandemic, most participants had health problems, especially mental health problems, including problems with their own substance use. Some had study delay. Analysis revealed that during the pandemic, most participants experienced an increase in these problems. This appeared to be related to their living situation: An increase in violence and relapse of relatives increased stress, especially for those living with their relatives. The coping strategies 'standing up' or 'putting up', and a decrease in support-social, professional, and educational-also contributed to stress. A few participants experienced less health problems and study problems. This was related to diminishing addiction problems of relatives, less social pressure, available help, and the coping strategy 'withdrawing'. Withdrawing was much easier for participants who did not live with their relatives with addiction problems. It is recommended to keep schools and universities open during pandemics, offering a safe haven for students at risk in the home situation.

Towards functional improvement of motor disorders associated with cerebral palsy.

Cerebral palsy is a lifelong neurodevelopmental condition arising from non-progressive disorders occurring in the fetal or infant brain. Cerebral palsy has long been categorised into discrete motor types based on the predominance of spasticity, dyskinesia, or ataxia. However, these motor disorders, muscle weakness, hypotonia, and impaired selective movements should also be discriminated across the range of presentations and along the lifespan. Although cerebral palsy is permanent, function changes across the lifespan, indicating the importance of interventions to improve outcomes in motor disorders associated with the condition. Mounting evidence exists for the inclusion of several interventions, including active surveillance, adapted physical activity, and nutrition, to prevent secondary and tertiary complications. Avenues for future research include the development of evidence-based recommendations, low-cost and high-quality alternatives to existing therapies to ensure universal access, standardised cerebral palsy registers to harmonise epidemiological and clinical information, improved adult screening and check-up programmes to facilitate positive lived experiences, and phase 3 trials for new interventions.

Trends in perinatal outcomes of women with chronic medical conditions: A 10-year population-based study in the Netherlands.

OBJECTIVE: To gain insight into perinatal outcomes in women with chronic medical conditions, in order to contribute to the optimization of personalized perinatal care. We hypothesize that women with a chronic medical condition have poorer perinatal outcomes than women without a known chronic medical condition. DESIGN: Population-based study using data of the Netherlands Perinatal Registry between 2010-2019. SETTING: Nationwide study in the Netherlands. PARTICIPANTS: Pregnancies of women who were diagnosed with chronic medical conditions by a medical specialist before pregnancy (n=36,835), divided into seven sub-groups and a reference group of pregnancies of women without known chronic medical conditions (n=1,084,623). MEASUREMENTS AND FINDINGS: The primary outcome measure was mode of birth. Secondary outcomes measures were onset of labour, preterm birth, asphyxia, Neonatal Intensive Care Unit (NICU) admission, and perinatal mortality. Spontaneous birth ranged from 45.0% (orthopaedic conditions) to 71.3% (neurological conditions) to 82.6% in the reference group. Assisted vaginal birth, planned caesarean birth, and emergency caesarean birth occurred significantly more in all groups compared to the reference group (p<0.001). Preterm birth was significantly more likely in the studied groups as well as perinatal asphyxia and NICU admission (all p<0.001). Adjusting for mode of birth, parity, age and ethnicity did not change the outcomes for the group of women with chronic medical conditions. Perinatal mortality was seen in all groups but in none of the separate groups significantly more than in the reference group. Descriptive statistics, univariate and multivariable logistic regression analyses were applied. KEY CONCLUSIONS: Women with chronic medical conditions are more likely to experience preterm birth, caesarean births and NICU admission of the new-born. IMPLICATIONS FOR PRACTICE: Knowledge about perinatal outcomes of women with chronic medical conditions is a first step for obstetrics care providers in order to optimize personalized care.

Aging With Cerebral Palsy: A Photovoice Study Into Citizenship.

Background: Adults with cerebral palsy (CP) may experience an increasing impact of their disability on daily life and this may interfere with their citizenship. Citizenship is a layered construct. Next to formal and theoretical significations, and civil rights acts such as the UN Convention on the Rights for Persons with Disabilities (CRPD), the meaning of citizenship is formed by the person themselves. The present study aimed to gain insight into what citizenship means for adults with CP 40 years or older and what is needed to support and pursue their citizenship to improve person-centered rehabilitation which can facilitate this process. Methods: Adults with CP (>40 years) without intellectual disability were recruited from medical records of a large rehabilitation center to participate in a qualitative study using the photovoice method. Participants were asked to take photos of objects or life situations that constituted citizenship for them; these photos were then the prompts for the semi-structured interviews that were held face-to-face at their homes. Background and clinical characteristics were gathered using a short face-to-face questionnaire. Data were analyzed through inductive thematic analysis. Results: Nineteen adults participated [mean age (SD) 57.8 (9.4) years (range 44-79), six men]. From the analysis four themes emerged: (a) Meanings of citizenship; (b) Citizenship: Facilitator and barriers; (c) Paradoxes of support and participation; and (d) Future. Furthermore, next to the ability to participate in society without restrictions, sense of belonging was reported to be an important aspect of "meanings of citizenship." The physiotherapist was perceived as an important health professional to maintain physical activity and deal with the impact of aging with CP on daily activities. Complex healthcare and support services regulations and aging affected citizenship negatively. Conclusion: Middle-aged and older adults with CP view citizenship as the ability to participate and belong in society. To optimize their citizenship the challenges and individual needs must be seen and supported by person-centered rehabilitation and support services. Simplification of complex healthcare and services regulations can further improve citizenship.

Development of an ICF Core Set for adults with cerebral palsy: capturing their perspective on functioning.

AIM: To examine the most relevant aspects of functioning of adults with cerebral palsy (CP) from their perspective, in order to develop an International Classification of Functioning, Disability and Health (ICF) Core Set for adults with CP. METHOD: We conducted six focus group discussions with adults with CP without intellectual disability and seven interviews with adults with CP with intellectual disability and caregivers, addressing all ICF components. Meaningful concepts were identified from verbatim transcripts and linked to ICF categories by two independent researchers. RESULTS: In total, 31 adults with CP without intellectual disability (mean [SD] age 46y 1mo [14y 1mo]; 20 females, 11 males; Gross Motor Function Classification System [GMFCS] levels I-IV) and seven adults with CP and intellectual disability (mean [SD] age 25y 8mo [6y 8mo]; four females, three males; GMFCS levels III-V) participated. We identified 132 unique second-level categories: 47 body functions, seven body structures, 43 activities and participation, and 35 environmental factors. The most frequently mentioned categories were emotional function, pain, muscle tone function, support of family, products and technology, and health services. INTERPRETATION: Adults with CP experienced problems in a broad range of body functions and activities and indicated the importance of environmental factors for functioning. The identified categories will be added to the list of candidate items to reach consensus on an ICF Core Set for adults with CP. What this paper adds Including the lived experience is crucial for fully understanding functioning of adults with cerebral palsy (CP). Adults with CP perceive environmental factors as essential elements for everyday functioning. Adults with intellectual disability should be considered as a group with specific problems.

Pain in adults with cerebral palsy: A systematic review and meta-analysis of individual participant data.

BACKGROUND: There is little focus on adults with cerebral palsy (CP) in research and health care and insufficient knowledge on how to identify and manage pain in this population. OBJECTIVES: This systematic review and meta-analysis aimed to determine whether pain prevalence in adults with CP is high and to explore variations in pain prevalence of subgroups, pain locations, pain severity and pain interference. METHODS: Potential datasets were identified by experts in the field and literature searches in Embase, MEDLINE, and Cochrane, from January 2000 to October 2016. Included studies had a representative sample of ≥25 adults with CP and ≥1 pain outcomes. Methodological quality assessment, pain prevalence estimates and logistic regression models for subgroup effects on pain prevalence were conducted. RESULTS: In total, 17 eligible studies were identified from 4584 publications. A meta-analysis was performed with individual participant data from 15 studies totalling 1243 participants (mean [SD] age 34.3 [12.6] years). Overall mean pain prevalence was 70% (95% CI 62-78). Women were more likely to have pain than men (P<0.001). The odds of pain was increased in adults with gross motor function level II (odds ratio [OR] 1.92, 95% CI 1.22-3.12) and IV (OR 1.77, 95% CI 1.03-4.29). Participants with pain reported pain predominantly in the legs (76%, 95% CI 66-84), and mean pain severity was 3.7/10 (95% CI 2.7-4.7) and pain interference 3.5/10 (95% CI 2.5-4.5). CONCLUSIONS: This meta-analysis provides the first reliable pain prevalence estimate in a large international sample of adults with CP. The high prevalence of pain, 70%, suggests that adults with CP should be routinely screened for pain and treated accordingly. The range of measurement instruments used by the included studies emphasizes using common outcome measures specific to pain internationally.

Entering the labor market: increased employment rates of young adults with chronic physical conditions after a vocational rehabilitation program.

PURPOSE: Employment of young adults with chronic physical conditions entering the labor market after finishing post-secondary education remains behind compared to typically developing peers. The aim of this study is to evaluate changes in their paid employment levels after following a vocational rehabilitation intervention ('At Work'). MATERIALS AND METHODS: Participants aged between 16 and 27 years (n = 90) were recruited via rehabilitation physicians and a jobcoach agency and participated in a vocational rehabilitation program. Cochran's Q and McNemar tests served to test the development of intervention participants' paid employment over time. Chi-square tests were used to compare intervention participants' paid employment level with national reference data selected on age and having a self-reported chronic physical condition. RESULTS: Paid employment level of the intervention cohort significantly increased from 10.0% at baseline to 42.4% at 2-years follow-up (p < 0.001). At 2-years follow-up, their employment rates approached the employment rates of national reference data (42.4% versus 52.9%, p = 0.17). CONCLUSION: Starting from a disadvantaged position, the paid employment rate of the intervention cohort substantially increased over time, approaching the employment rate of reference data. 'At Work' seems to be appropriate for supporting this specific group who face obstacles to enter the labor market, to find competitive employment.IMPLICATIONS FOR REHABILITATION'At Work' is a vocational rehabilitation intervention for young adults with chronic physical conditions who experience problems with finding and maintaining competitive employment after finishing post-secondary education.The 'At Work' intervention entails a combination of group sessions and individual coaching sessions based on the supported employment methodology.Paid employment rates of the intervention cohort substantially increased on the short- and long term, and approached employment rates of reference data of persons with chronic physical conditions.The 'At Work' intervention seems appropriate to support young adults with chronic physical conditions who experience barriers for work participation, to enter the labor market and find competitive and sustainable employment.

Focus on autonomy: Using 'Skills for Growing Up' in pediatric rehabilitation care.

PURPOSE: Youth with disabilities face challenges regarding achieving autonomy. The 'Skills for Growing Up' tool was adapted for use in Dutch pediatric rehabilitation (SGU-D) to support development of autonomy. This study presents the experiences with the SGU-D tool. METHODS: The SGU-D was implemented in 18 settings, of which 4 participated in the evaluation. Rehabilitation professionals were trained in the use of the SGU-D. In a qualitative study, participants were interviewed for their opinions regarding the tool. RESULTS: Experience with the SGU-D was evaluated in 11 youth with disabilities, 11 parents and 8 rehabilitation professionals. They perceived the SGU-D as a helpful tool: i) to support development of autonomy, ii) to focus on future perspectives, and iii) to facilitate communication with family and rehabilitation professionals. Additional support from rehabilitation professionals on using the SGU-D was appreciated. CONCLUSION: Youth with disabilities, their parents and rehabilitation professionals value the SGU-D as a practical tool for working on autonomy, and to identify important areas of development.

Epidemiology of Cerebral Palsy in Adulthood: A Systematic Review and Meta-analysis of the Most Frequently Studied Outcomes.

OBJECTIVE: To describe the epidemiology of health status, impairments, activities and participation in adults with cerebral palsy (CP). DATA SOURCES: Embase, MEDLINE, Web of Science, PsycINFO, Cumulative Index to Nursing and Allied Health, Cochrane, and Google Scholar were searched for 3 themes ("cerebral palsy," "adult," and "outcome assessment") in literature published between January 2000 and December 2018. STUDY SELECTION: Full-article peer-reviewed English journal articles on descriptive, observational, or experimental studies describing the most studied outcomes in adults with CP (n≥25, age≥18y) were included. Studies were included in the analyses if frequently studied outcomes were described in at least 3 studies using similar methods of assessment. DATA EXTRACTION: Data were extracted independently by 2 authors from 65 articles (total N=28,429) using a standardized score sheet. DATA SYNTHESIS: Meta-analyses revealed that overall, on average 65.1% (95% confidence interval [CI], 55.1-74.5) of adults with CP experienced pain, 57.9% (95% CI, 51.1-64.6) were ambulant, 65.5% (95% CI, 61.2-69.7) had little or no limitation in manual ability, 18.2% (95% CI, 10.6-27.2) had tertiary education, 39.2% (95% CI, 31.5;47.1) were employed, and 29.3% (95% CI, 9.0-55.3) lived independently. In adults without intellectual disability, proportions of individuals who were ambulant (72.6% [95% CI, 58.8-84.5]) and lived independently (90.0% [95% CI, 83.8-94.9]) were higher (P=.014 and P<.01, respectively). The Fatigue Severity Scale score was 4.1 (95% CI, 3.8-4.4). Epilepsy (28.8% [95% CI, 20.1-38.4]) and asthma (28.3% [95% CI, 18.7-38.9]) were especially prevalent comorbidities. CONCLUSIONS: The present systematic review and meta-analysis on the epidemiology of adults with CP provided state-of-the-art knowledge on the most frequently studied outcomes. On average, adults with CP are fatigued, and a majority experience pain, are ambulant, and have little or no difficulty with manual ability. On average, 40% are employed and 30% live independently. More uniformity in assessment and reports is advised to improve knowledge on epidemiology and gain insight in more outcomes.

Autonomy in participation in cerebral palsy from childhood to adulthood.

AIM: To determine the long-term development of autonomy in participation of individuals with cerebral palsy (CP) without intellectual disability. METHOD: Individuals with CP (n=189, 117 males, 72 females; mean age [SD] 21y 11mo [4y 11mo], range 12-34y); were assessed cross-sectionally (46%) or up to four times (54%), between the ages of 12 and 34 years. Autonomy in participation was classified using phase 3 of the Rotterdam Transition Profile. A logistic generalized estimating equation regression model was used to analyse autonomy in six domains (independent variables: age, Gross Motor Function Classification System [GMFCS] level, and interaction between age and GMFCS level). Proportions of autonomy were compared to references using binomial tests (p<0.05). RESULTS: In most domains, over 90% of participants (n=189, 400 observations, 80% in GMFCS levels I and II) reached autonomy in participation in their late twenties, except for intimate/sexual relationships. Those in GMFCS levels III to V compared to those in GMFCS levels I and II had less favourable development of autonomy in the transportation, intimate relationships, employment, and housing domains, and more favourable development in the finances domain. Compared to references, fewer individuals with CP were autonomous in participation. INTERPRETATION: This knowledge of autonomy may guide the expectations of young people with CP and their caregivers. Furthermore, rehabilitation professionals should address autonomy development in intimate relationships, employment, and housing, especially in individuals with lower gross motor function. WHAT THIS PAPER ADDS: Individuals with cerebral palsy without intellectual disability achieved autonomy in most participation domains. Regarding intimate relationships, they continued to have less experience compared to age-matched references. Development of autonomy was less favourable for individuals in Gross Motor Function Classification System levels III to V.

AUTONOMÍA EN LA PARTICIPACIÓN EN LA PARÁLISIS CEREBRAL DESDE LA INFANCIA A LA EDAD ADULTA: OBJETIVO: Determinar el desarrollo a largo plazo de la autonomía en la participación de individuos con parálisis cerebral (PC) sin discapacidad intelectual. MÉTODO: Individuos con PC (n=189, 117 varones, 72 mujeres; edad media [DE] 21 años y 11 meses [4 años y 11meses], rango 12-34 años); fueron evaluados de forma transversal (46%) o hasta cuatro veces (54%), entre las edades comprendidas entre los 12 y los 34 años. La autonomía en la participación fue clasificada usando la fase 3 del perfil de transición de Rotterdam (Rotterdam Transition Profile). Se utilizó un modelo de regresión logístico generalizado estimando la ecuación para analizar la autonomía en 6 dominios (variables independientes: edad, nivel del Sistema de Clasificación de la Función Motora Gruesa, [GMFCS], y la interacción entre la edad y el nivel GMFCS). Las proporciones de la autonomía se compararon con las referencias mediante pruebas binomiales (p<0,05). RESULTADOS: En la mayoría de los dominios, más del 90% de los participantes (n=189.400 observaciones, 80% en los niveles I y II de la GMFCS) alcanzaron la autonomía en la participación bien entrados los 20 años, excepto para las relaciones íntimas/sexuales. Aquellos en los niveles III y V de la GMFCS en comparación con los niveles I y II de la GMFCS tuvieron un desarrollo de la autonomía menos favorable en el transporte, relaciones íntimas, empleo y en el dominio de la vivienda, y un desarrollo más favorable en el dominio de las finanzas. En comparación con las referencias, menos individuos con PC fueron autónomos en la participación. INTERPRETACIÓN: Este conocimiento de la autonomía puede guiar las expectativas de los jóvenes con PC y de sus cuidadores. Además, los profesionales de la rehabilitación deberían abordar el desarrollo de la autonomía en las relaciones íntimas, el empleo y la vivienda, especialmente en aquellos individuos con una función motora gruesa más baja.

AUTONOMIA NA PARTICIPAÇÃO EM PARALISIA CEREBRAL DA INFÂNCIA À VIDA ADULTA: OBJETIVO: Determinar o desenvolvimento em longo prazo da autonomia na participação de indivíduos com paralisia cerebral (PC) sem deficiência intelectual. MÉTODO: Indivíduos com PC (n=189, 117 do sexo masculino, 72 do sexo feminino; média de idade [DP] 21a 11m [4a 11m], variação 12-34a); foram avaliados transversalmente (46%) ou até quatro vezes (54%), entre as idades de 12 e 34 anos. A autonomia na participação foi classificada usando a fase 3 do Perfil de Transição de Rotterdam. Um modelo de regressão logística com generalizada com equações estimadas foi usado para analisar a autonomia em seis domínios (variáveis independentes: idade, Nível segundo o Sistema de Classificação da Função Motora Grossa [GMFCS], e interação entre idade e nível GMFCS). As proporções de autonomia foram comparadas com as referências usando testes binomiais (p<0,05). RESULTADOS: Na maior parte dos domínios, cerca de 90% dos participantes (n=189.400 observações, 80% nos níveis GMFCS I e II) atingiram autonomia na participação no final da terceira década de vida, exceto para relações íntimas/sexuais. Aqueles nos níveis GMFCS III a V comparados com os níveis I e II tiveram desenvolvimento menos favorável nos domínios da autonomia no transporte, relações íntimas, emprego, e domicílio, e mais desenvolvimento mais favorável no domínio das finanças. Em comparação com as referências, menos indivíduos com PC foram autônomos na participação. INTERPRETAÇÃO: Este conhecimento sobre a autonomia pode guiar as expectativas de jovens com PC e seus cuidadores. Além disso, profissionais da reabilitação devem abordar o desenvolvimento da autonomia nas relações íntimas, emprego e domicílio, especialmente nos indivíduos com menor função motora grossa.

Skills for Growing Up-Epilepsy: An exploratory mixed methods study into a communication tool to promote autonomy and empowerment of youth with epilepsy.

INTRODUCTION: The consequences of having epilepsy seriously hamper the development of autonomy for youth with epilepsy (YWE) and limit their social participation. This study was designed to provide insight into the impact of epilepsy on autonomy and empowerment and to evaluate the use of the Skills for Growing Up (SGU-Epilepsy) communication tool in pediatric epileptic care. METHODS: A mixed methods design was used to examine the impact of epilepsy on autonomy and empowerment and to evaluate the feasibility and use of the SGU-Epilepsy. Six focus groups with YWE and their parents were organized (N = 27), and the benefits of the SGU-Epilepsy were evaluated (N = 72). RESULTS: Youth with epilepsy struggled with social participation. Beliefs of YWE and their parents on managing daily life and taking medication were not always similar. Parents worried about the passive attitude of YWE, but autonomy of YWE seemed to be constrained by parents. The SGU-Epilepsy seemed to be feasible. It facilitated the communication on development and empowerment although it was sometimes confronting for parents. Not all YWE were motivated to use and discuss the tool. In the evaluation, no differences in perceived autonomy and empowerment between YWE who used SGU-Epilepsy and received usual care appeared. CONCLUSION: On the short-term, a beneficial effect of using the SGU-Epilepsy on autonomy and empowerment for YWE and their parents was not shown. The SGU-Epilepsy seemed to be a feasible instrument, however, to address nonmedical issues during pediatric consultations.

Neurological outcome in children and youth with acquired brain injury 2-year post-injury.

OBJECTIVE: To determine neurological outcome in children and youth with acquired brain injury (ABI) and explore associated factors. DESIGN: Cross-sectional study, two-years post-injury. PATIENTS: Hospital-based sample (n=112) aged 6-22 years. METHODS: Neurological outcome and participation were assessed with a multidimensional neurological examination and the Child and Adolescent Scale of Participation. Logistic regression analyses were used to explore the relationships. RESULTS: Both sensorimotor and cognitive deficits were found in 30-31%, language deficits and behavioural deficits in 10-17%. Non-traumatic injury had a negative impact on neurological outcome, specifically regarding sensorimotor and language deficits. Lower education level showed a significantly poorer neurological outcome. High levels of age-expected participation were reported, with a significant relation between deficits and participation restrictions, especially at school. CONCLUSION: One out of three have a poor neurological outcome, related to type of injury and lower level of education. The amount of deficits is associated with participation restrictions.

Long-Term Deterioration of Perceived Health and Functioning in Adults With Cerebral Palsy.

OBJECTIVE: To describe longitudinal change in perceived health, presence of health issues, and functional level in adults with cerebral palsy (CP). DESIGN: Prospective cohort study. SETTING: Participants' daily environment. PARTICIPANTS: Adults (N=49) with CP (age range, 35-45y; 27 [55%] men; 36 [75%] spastic) formerly known in pediatric rehabilitation care participated. INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Postal questionnaires were completed by the adults or their proxies (n=9). Health outcomes included perceived health (adapted from the 36-Item Short Form Health Survey) and presence of health issues such as pain, severe fatigue (dichotomized), and functional level (Barthel Index; walking performance). RESULTS: Over a 10-year period, the percentage of adults with CP worrying about their health increased (29%-54%; P=.008) and those indicating that health problems limit their activities increased (19%-45%; P=.002). In the same period, most adults continued to report good general health (93%-86%; P=.148). Presence of some health issues increased over time, such as pain; severe fatigue was a common health issue at follow-up (32%). Over a 14-year period, mobility and self-care deteriorated (Barthel Index, 17.1±4.8 to 16.3±5.6; P=.007). Walking performance, specifically indoors, declined (83%-71%; P=.010). CONCLUSIONS: Adults with CP experienced deterioration in health outcomes in the long term. Most notably, perceived health and functional level decreased. Pain and severe fatigue were the most common health issues in adult CP. More research is required to explore the mechanisms at work in the process of aging in persons with CP. Systematic follow-up of adults with CP appears necessary to timely detect and intervene in health problems and functional decline.

Course of employment in adults with cerebral palsy over a 14-year period.

AIM: To explore the course of employment in adults with cerebral palsy (CP) over 14 years, and to identify subgroups at risk for unemployment. METHOD: Sixty-five adults with CP (33 males, 32 females; baseline age 25y 8mo, standard deviation [SD] 3y 2mo; intellectual impairment 25%; bilateral CP 65%) participated in a prospective cohort study. Self-reports of employment and work hours per week in 1996, 2000, and 2010 were documented. The course of employment (including sheltered work) and work hours per week were analysed, using generalized estimating equations (GEE). RESULTS: Overall, employment rate was stable over time (38-45%, p=0.413), but lower than in the general population (75-86%, p<0.001). Employment rates were specifically low in adults with intellectual impairment, bilateral CP, and in adults with Gross Motor Function Classification System (GMFCS) levels IV and V. Work hours per week declined (35.0 [SD 7.9] to 31.2 [SD 10.3], p=0.033), especially among females (32.3 [SD 6.4] to 23.4 [SD 7.4], p<0.001). Similar to the general population, females often worked part-time. INTERPRETATION: Employment was low compared with the general population, but remained stable in the long term; however, work hours per week decreased. Adults with intellectual impairment, bilateral CP, and GMFCS levels IV and V are subgroups at risk for unemployment.

Health and participation problems in older adults with long-term disability.

BACKGROUND: More attention and understanding of the health and participation problems of adults with early and later onset disabilities in the Netherlands is needed. OBJECTIVE: To explore health/participation problems and unmet needs in adults aged ≥40 years with long-term disabilities and their relationship with the time of onset. METHODS: Participants were recruited in the Netherlands through newsletters and social media to participate in a web-based questionnaire. The questionnaire assessed background characteristics, (change in) health/participation problems, and unmet needs. Spearman's rho was used to examine the relationships with time of onset. RESULTS: Of the 163 survey respondents, 42% acquired their disability before age 25 years and reported fatigue (77%), walking problems (66%), and pain (59%). In 21% of the respondents with early-onset disability fatigue, pain and depressive feelings co-occurred. Early-onset disability correlated with joint deformities, pain and anxiety. Participation problems included loss of income and fewer social activities. Early-onset correlated with the need for more information about diagnosis and prognosis. CONCLUSIONS: People aged over 40 years with long-term disability have significant and increasing health and participation problems. Adults with early-onset disability are more likely to have health or participation problems than adults with late-onset disability. Early identification is needed for preventive care and access to specialized services that focus on improving and maintaining physical symptoms, energy management, and participation.

Evaluating outpatient transition clinics: a mixed-methods study protocol.

INTRODUCTION: To support young people in their transition to adulthood and transfer to adult care, a number of interventions have been developed. One particularly important intervention is the transition clinic (TC), where paediatric and adult providers collaborate. TCs are often advocated as best practices in transition care for young people with chronic conditions, but little is known about TC models and effects. The proposed study aims to gain insight into the added value of a TC compared with usual care (without a TC). METHODS AND ANALYSIS: We propose a mixed-methods study with a retrospective controlled design consisting of semistructured interviews among healthcare professionals, observations of consultations with young people, chart reviews of young people transferred 2-4 years prior to data collection and questionnaires among the young people included in the chart reviews. Qualitative data will be analysed through thematic analysis and results will provide insights into structures and daily routines of TCs, and experienced barriers and facilitators in transitional care. Quantitatively, within-group differences on clinical outcomes and healthcare use will be studied over the four measurement moments. Subsequently, comparisons will be made between intervention and control groups on all outcomes at all measurement moments. Primary outcomes are 'no-show after transfer' (process outcome) and 'experiences and satisfaction with the transfer' (patient-reported outcome). Secondary outcomes consider clinical outcomes, healthcare usage, self-management outcomes and perceived quality of care. ETHICS: The Medical Ethical Committee of the Erasmus Medical Centre approved the study protocol (MEC-2014-246). DISSEMINATION: Study results will be disseminated through peer-reviewed journals and conferences. The study started in September 2014 and will continue until December 2016. The same study design will be used in a national study in 20 diabetes settings (2016-2018).