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.

Real-time daily fatigue, sleep, physical activity, and health-related fitness in adults with cerebral palsy.

AIM: To explore whether subgroups of adults with cerebral palsy (CP) with different fatigue diurnal profiles can be discerned, and to explore whether sleep, physical activity, or health-related fitness are associated with these profiles. METHOD: Thirty-two adults (median age 29 years 8 months; range 20-54 years; 11 males, 21 females) with spastic CP (Gross Motor Function Classification System levels I-III) with physical activity-related fatigue complaints participated. Real-time fatigue and physical activity were assessed for 7 consecutive days by short message service text four times during the day and by wearing an accelerometer respectively. Sleep was assessed by the Pittsburgh Sleep Quality Index, and fitness by assessing body composition and aerobic capacity. Latent class growth modelling was used to classify subgroups according to their diurnal profiles of real-time fatigue. Univariable multinomial logistic regression analysis explored whether participant characteristics, sleep, physical activity, or health-related fitness were associated with diurnal profiles. RESULTS: Three distinct fatigue diurnal profiles were identified: stable low (n = 10), increasing (n = 14), and stable high (n = 8). Only aerobic capacity was associated with fatigue profiles (odds ratio 1.15, 95% confidence interval 1.00-1.34; p = 0.05). INTERPRETATION: Fatigue in adults with CP may be low or high stable or may increase during the day. These findings indicate the relevance of assessing fatigue variability. WHAT THIS PAPER ADDS: We found three patterns of daily fatigue in adults with cerebral palsy (CP). Only aerobic capacity was associated with fatigue profiles in adults with CP. Moment-to-moment variations in fatigue can help with personalized fatigue management.

An international clinical perspective on functioning and disability in adults with cerebral palsy.

BACKGROUND: This international, multi-center cross-sectional study is one of the preparatory studies in the development of the International Classification of Functioning, Disability and Health (ICF) Core Sets for adults with cerebral palsy (CP) to describe their functioning and health. OBJECTIVE: To identify the most common problems in functioning of adults with CP presenting in healthcare services, and facilitating and hindering environmental factors, using the ICF as a reference. METHODS: Participants were adults with CP who visited healthcare services in the Netherlands, Sweden, Thailand, and the United States. Structured interviews were performed using an adapted version of the generic ICF checklist 2.1a (106 categories) to rate the participant's functioning and the impact of environmental factors. Descriptive statistics were used for frequency analysis. RESULTS: In total, 101 participants were included, of whom 69 without intellectual disability (mean age ± SD of 38.4 ± 14.7 y; 85.5% with spastic type of CP; Gross Motor Function Classification System (GMFCS) levels I-V) and 32 with intellectual disability (mean age ± SD of 25.0 ± 6.4 y; 71.9% with spastic type of CP; GMFCS levels I-V). A total of 104 ICF categories in the ICF checklist were frequently present in adults with CP: 27 body functions, 4 body structures, 53 activities and participation, and 20 environmental factors. CONCLUSIONS: The most common problems of adults with CP presenting in healthcare services are diverse and highly prevalent. The study results add the clinical perspective on relevant categories of functioning to the basis for developing the ICF Core Sets for adults with CP.

A core outcome set for multimorbidity risk in individuals with cerebral palsy.

AIM: To: (1) investigate the importance of outcome measurement instruments (OMIs) within a core outcome set (COS) for multimorbidity (at least two chronic health conditions) risk in individuals with cerebral palsy (CP); (2) investigate the feasibility of OMIs within the COS in international clinical research settings in adolescents and adults with CP; and (3) describe the associations between the COS data and Gross Motor Function Classification System (GMFCS) levels. METHOD: Eighty-three individuals with CP completed a survey on health outcomes: physical behaviour, nutrition, sleep, endurance, body composition, blood pressure, blood lipids, and glucose. A cross-sectional study assessed the feasibility of the COS in 67 adolescents and adults with CP (mean age 30y, SD 15y 1mo, min-max: 14-68y, 52.2% male) at four centres. Prevalence of multimorbidity risk and associations with GMFCS levels are described. RESULTS: Most participants rated physical behaviour, nutrition, sleep, and endurance as very important. Body composition, blood pressure, nutrition, and sleep were highly feasible since data were collected in 88% or more participants who consented to having the assessments. Physical behaviour, cardiorespiratory endurance, and blood draws were collected in less than 60% of participants. Total time sedentary (ρ=0.53, p<0.01) and endurance (ρ=-0.46, p<0.01) were significantly associated with GMFCS level. INTERPRETATION: The COS identified that most participants had poor sleep quality and endurance, did not have healthy diets, and showed increased sedentary behaviour. Individuals with CP valued these outcomes as most important, suggesting a need to assess these modifiable behaviours in this population. Objective measures of physical behaviour and cardiorespiratory endurance in the COS required additional personnel, time, and participant burden. We recommend that healthcare providers should perform a simpler first screen using questionnaire-based assessments and then focus the use of the remainder of the COS if required for the patient.

UM DESFECHO CENTRAL DEFINIDO PARA RISCO DE MULTIMORBIDADE EM INDIVÍDUOS COM PARALISIA CEREBRAL: OBJETIVO: (1) Investigar a importância dos instrumentos de medição de desfechos (IMD) dentro de um conjunto de desfechos principais (CDS) para risco de multimorbidade (pelo menos duas condições crônicas de saúde) em indivíduos com paralisia cerebral (PC); (2) Investigar a viabilidade de IMD dentro do CDS em cenários internacionais de pesquisa clínica em adolescentes e adultos com PC; e (3) Descrever as associações entre os dados do CDS e os níveis do Sistema de Classificação da Função Motora Grossa (GMFCS). MÉTODO: Oitenta e três indivíduos com PC completaram uma pesquisa sobre desfechos em saúde: comportamento físico, nutrição, sono, resistência, composição corporal, pressão arterial, lipídios no sangue e glicose. Um estudo transversal avaliou a viabilidade do COS em 67 adolescentes e adultos com PC (idade média de 30 anos, desvio padrão de 15 anos e 1 mês, min-max: 14-68 anos, 52,2% do sexo masculino) em quatro centros. São descritas a prevalência do risco de multimorbidade e as associações com os níveis de GMFCS. RESULTADOS: A maioria dos participantes classificou o comportamento físico, nutrição, sono e resistência como muito importantes. Composição corporal, pressão arterial, nutrição e sono foram altamente viáveis, uma vez que os dados foram coletados em 88% ou mais dos participantes que consentiram em realizar as avaliações. Comportamento físico, resistência cardiorrespiratória e coleta de sangue foram coletados em menos de 60% dos participantes. O tempo total de sedentarismo (ρ = 0,53, p < 0,01) e resistência (ρ = −0,46, p < 0,01) foram significativamente associados ao nível de GMFCS. INTERPRETAÇÃO: O CDS identificou que a maioria dos participantes tinha má qualidade e resistência do sono, não tinha dietas saudáveis e apresentava um comportamento sedentário aumentado. Indivíduos com PC valorizaram esses desfechos como mais importantes, sugerindo a necessidade de avaliar esses comportamentos modificáveis nessa população. Medidas objetivas de comportamento físico e resistência cardiorrespiratória no CDS exigiram pessoal adicional, tempo e sobrecarga do participante. Recomendamos que os profissionais de saúde realizem uma primeira triagem mais simples usando avaliações baseadas em questionários e, em seguida, concentrem o uso do restante do CDS, se necessário para o paciente.

ICF Core Sets for the assessment of functioning of adults with cerebral palsy.

AIM: To report on the results of the online international consensus process to develop the comprehensive and brief International Classification of Functioning, Disability and Health (ICF) Core Sets for adults with cerebral palsy (CP). METHOD: An online iterative decision-making and consensus process involved 25 experts, including clinicians and researchers working with adults with CP, an adult with CP, and the parents of adults with CP from all six regions of the World Health Organization. The most relevant categories were selected from a list of 154 unique second-level candidate categories to develop the ICF Core Sets for adults with CP. This list resulted from evidence gathered during four preparatory studies, that is, a systematic literature review, a qualitative study, an expert survey, and an empirical study. RESULTS: The consensus process resulted in the comprehensive ICF Core Set containing 120 second-level ICF categories: 33 body functions; eight body structures; 50 activities and participation; and 29 environmental factors, from which the most essential categories, 33 in total, were selected for the brief ICF Core Set. For body functions, most of the categories were mental functions and neuromusculoskeletal and movement-related functions. Body structures were mostly related to movement. All the chapters of the activities and participation component were represented, with mobility and self-care as the most frequently covered chapters. For environmental factors, most of the categories addressed products and technology and services, systems, and policies. INTERPRETATION: The comprehensive and brief ICF Core Sets for adults with CP were created using a new online version of an established ICF Core Set consensus process. These Core Sets complement the age-specific ICF Core Sets for children and young people with CP and will promote standardized data collection worldwide.

Pain, fatigue, depressive symptoms and sleep disturbance in young adults with cerebral palsy.

PURPOSE: Investigate pain, fatigue, depressive symptoms and sleep disturbance in young adults with cerebral palsy compared to references. MATERIALS AND METHODS: Young adults with cerebral palsy (n = 97, aged 21-34 years) and age-matched references from the general population (n = 190) rated pain using a numeric rating scale and fatigue, depressive symptoms, sleep disturbance and global health using Patient-Reported Outcomes Measurement Information System® short forms. Scores were compared between cerebral palsy subgroups and the reference population. Correlation coefficients and linear regression analyses assessed interrelationships of health issues and associations with global health. RESULTS: Individuals with Gross Motor Function Classification System level I had less pain, fatigue and depressive symptoms, while individuals with levels II and III-V had more pain (53% and 56%, p < 0.001) and those with levels III-V more fatigue (39%, p = 0.035) than references (pain: 26%, fatigue: 14%). Pain and fatigue were more interrelated (correlation coefficients: 0.71 vs. 0.41) and stronger associated with global mental health in individuals with cerebral palsy. CONCLUSIONS: Young adults with Gross Motor Function Classification System levels II-V report more pain and those with levels III-V report more fatigue than references. Pain and fatigue are highly interrelated and specifically relate to mental health in individuals with cerebral palsy.Implications for rehabilitationExcept for those in the highest level of motor function, young adults with cerebral palsy report higher levels of pain and fatigue compared to the general population of the same age.Pain and fatigue are strongly interrelated and associated with mental health in young adults with cerebral palsy.The present study recommends to monitor pain and fatigue in young adults with cerebral palsy with low levels of gross motor function.We advise rehabilitation professionals to consider combined treatment for both pain and fatigue.

Blood pressure in adults with cerebral palsy: a systematic review and meta-analysis of individual participant data.

OBJECTIVES: This systematic review and meta-analysis was designed to determine the overall mean blood pressure and prevalence of hypertension among a representative sample of adults living with cerebral palsy by combining individual participant data. Additional objectives included estimating variations between subgroups and investigating potential risk factors for hypertension. METHODS: Potential datasets were identified by literature searches for studies published between January 2000 and November 2017 and by experts in the field. Samples of adults with cerebral palsy (n ≥ 10, age ≥ 18 years) were included if blood pressure data, cerebral palsy-related factors (e.g. cerebral palsy subtype), and sociodemographic variables (e.g. age, sex) were available. Hypertension was defined as at least 140/90 mmHg and/or use of antihypertensive medication. RESULTS: We included data from 11 international cohorts representing 444 adults with cerebral palsy [median (IQR) age of the sample was 29.0 (23.0-38.0); 51% men; 89% spastic type; Gross Motor Function Classification System levels I-V]. Overall mean SBP was 124.9 mmHg [95% confidence interval (CI) 121.7-128.1] and overall mean DBP was 79.9 mmHg (95% CI 77.2-82.5). Overall prevalence of hypertension was 28.7% (95% CI 18.8-39.8%). Subgroup analysis indicated higher blood pressure levels or higher prevalence of hypertension in adults with cerebral palsy above 40 years of age, men, those with spastic cerebral palsy or those who lived in Africa. BMI, resting heart rate and alcohol consumption were risk factors that were associated with blood pressure or hypertension. CONCLUSION: Our findings underscore the importance of clinical screening for blood pressure in individuals with cerebral palsy beginning in young adulthood.

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.

Effect of pain on mood affective disorders in adults with cerebral palsy.

AIM: To determine if pain is associated with 12-month incidence of mood affective disorders (MAD) in adults with cerebral palsy (CP). METHOD: Data from Optum Clinformatics® Data Mart (2013-2016) were used for this retrospective cohort study. Diagnostic codes were used to identify adults (≥18y) with CP, incident cases of MAD, and covariates (other neurodevelopmental conditions, sleep disorders, arthritis). Pain (any type, location) was identified between 1st October 2014 and 30th September 2015. The pain group was divided into new or consistent pain if they had a history of pain (i.e. consistent) in the 12 months before their first pain claim date. Crude incidence rates of MAD (expressed per 100 person-years) were estimated. Cox regression was used to estimate hazard ratio (95% confidence interval [CI]) of MAD after adjusting for covariates. RESULTS: Adults that had new pain (n=859; incidence rate=15.5) or consistent pain (n=1303; incidence rate=17.9) had greater crude incidence rate of MAD compared to adults without pain (n=3726; incidence rate=5.9). The elevated rate of MAD remained after adjusting for covariates, for new pain (hazard ratio=2.4; 95% CI=1.9-3.0) and consistent pain (hazard ratio=2.1; 95% CI=1.7-2.7). INTERPRETATION: Pain is associated with greater incidence of MAD in adults with CP. This association remained after accounting for potential confounding factors. WHAT THIS PAPER ADDS: What this paper adds Pain was associated with higher 12-month incidence of mood affective disorders (MAD). The 12-month MAD incidence was similar between new and consistent pain groups. The MAD incidence remained higher adjusting for neurodevelopmental comorbidities, sleep disorders, and arthritis.

Developing an ICF Core Set for adults with cerebral palsy: A Global expert survey of relevant functions and contextual factors.

OBJECTIVE: To identify areas of functioning in adults with cerebral palsy that are considered relevant by experts, in order to develop an International Classification of Functioning, Disability and Health (ICF) Core Set for adults with cerebral palsy. PARTICIPANTS: Experts from various professional backgrounds worldwide who had experience working with adults with cerebral palsy for ≥2 years and were able to complete the survey in the English language. METHODS: A cross-sectional study using an international internet-based survey. The experts were asked to address relevant areas of functioning in adults with cerebral palsy. These areas of functioning were then linked to the ICF and the frequencies analysed. RESULTS: A total of 126 experts from 32 countries completed the survey. From the responses, 217 unique second-level ICF categories were identified. The three most frequently mentioned categories were "design, construction and building products and technology of buildings for public use (e150, 77%) and private use" (e155, 67%), followed by "sensation of pain" (b280, 62%). CONCLUSION: The broad diversity of ICF categories reported by the experts emphasize the known heterogeneity of cerebral palsy and the variety of func-tioning in adulthood. They also reported on many environmental factors, illustrating the importance of person-environment interactions. These findings provide information about relevant issues for use in developing an ICF Core Set for adults with cerebral palsy.

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.

Detection of body postures and movements in ambulatory adults with cerebral palsy: a novel and valid measure of physical behaviour.

BACKGROUND: Accurate measurement of physical behaviour is paramount to better understand lifestyle, health, and functioning, particularly in adults with physical disability as they may be at higher risk of sedentary lifestyle and subsequent negative health consequences. This study aimed: 1) to evaluate the criterion validity of a novel and clinically applicable activity monitor (AM, Activ8), in the detection of body postures and movements in adults with spastic cerebral palsy (CP); and 2) to evaluate the extent that the AM's positioning affects validity. METHODS: In this cross-sectional study, 14 ambulatory adults with CP [9 men; mean (SD) age, 35.4 (13.1) years] performed standardized activities while wearing three Activ8 monitors - frontolateral thigh (primary position), frontal thigh, and pant pocket - and being video recorded (criterion measure). AM activity output was compared to synchronized video recordings. Absolute (seconds) and relative [(video time-AM time)/mean time, %] time differences between methods were calculated. Relative time differences of < 10% were indicative of good validity. Comparison of AM attachment positions was completed using Spearman Rho correlation coefficients and Meng's tests. RESULTS: Criterion validity of the AM (frontolateral thigh) was good (average relative time differences: 0.25% for sitting, 4.69% for standing, 2.46% for walking, 1.96% for upright activity, 3.19% for cycling), except for running (34.6%). Spearman Rho correlation coefficients were greater between video/frontolateral thigh position than video/frontal thigh position and video/pant pocket position for body posture and movement categories sitting, standing, walking, and upright activity (p < 0.01 for all). CONCLUSIONS: The AM, positioned on the frontolateral thigh, demonstrated good criterion validity in ambulatory adults with CP. Though the Activ8 offers potential as an objective measure of physical activity, appropriate positioning is paramount for valid measurement.

Focus on Risk Factors for Cardiometabolic Disease in Cerebral Palsy: Toward a Core Set of Outcome Measurement Instruments.

OBJECTIVE: To identify existing outcome measurement instruments (OMIs) assessing risk factors for cardiometabolic disease in adolescents and adults with cerebral palsy (CP) reported on in the literature or used in the field. DATA SOURCES: The COnsensus-based Standards for the selection of health Measurement Instruments database of systematic reviews and 4 electronic databases (Embase, MEDLINE/Ovid, MEDLINE/Pubmed, PsychINFO) were searched up to June 19, 2017, that yielded 2594 articles. Experts in the field were consulted to identify any additional OMIs. STUDY SELECTION: Two reviewers independently applied inclusion criteria to select eligible studies using or evaluating measurement properties of OMIs assessing 1 of 8 outcomes: cardiorespiratory endurance, body size, body composition, physical behavior, sleep, nutrition, blood pressure, and blood lipids and glucose. Studies with an experimental or observational design including ≥10 adolescents or adults with CP were included. DATA EXTRACTION: One reviewer extracted data that were summarized for study and sample characteristics, outcomes, OMIs used, and if applicable data on measurement properties. Two reviewers rated the methodological quality and the quality of the OMIs. Feasibility for clinical practice and research was rated by experts in the field. DATA SYNTHESIS: Ninety OMIs were identified from 56 included articles and by the experts. Seventy OMIs pertained to cardiorespiratory endurance, body size, body composition, and physical behavior, whereas only 5 were identified for sleep and nutrition. Overall synthesis revealed that there is moderate to poor evidence for good quality of OMIs in this population. Based on feasibility for clinical practice, experts agreed on a single OMI per outcome (and 2 for cardiorespiratory endurance) to be included in a core set. CONCLUSION: Despite the range of available OMIs to assess risk factors for cardiometabolic disease in adolescents and adults with CP, evidence of good quality is often lacking. Nonetheless, a preliminary core set of 9 OMIs was systematically developed.

Outcomes in adults with cerebral palsy: systematic review using the International Classification of Functioning, Disability and Health.

AIM: In the context of the development of an International Classification of Functioning, Disability and Health (ICF) Core Set for adults with cerebral palsy (CP), this systematic review sought to identify the outcome measures used in studies on adults with CP, to examine their content using the ICF as a reference, and to demonstrate the most studied areas in this population. METHOD: Embase, MEDLINE, Web of Science, PsycINFO, CINAHL, Cochrane, and Google Scholar were searched for studies on adults with CP published between 2000 and 2017. Meaningful concepts of commonly used outcome measures were linked to the ICF, and frequencies of resultant ICF categories were explored. RESULTS: In 274 included articles, 332 outcome measures were identified of which 155 were commonly used. In total, 4409 meaningful concepts were linked to the ICF. The component 'Activities and participation' included the most frequent categories, followed by 'Body functions'. The most frequent categories were b280 'Sensation of pain' (37.6%), d450 'Walking' (33.3%), and d850 'Remunerative employment' (27.5%). INTERPRETATION: The broad range of ICF categories identified in this systematic review emphasizes the heterogeneity of functioning and disability in adults with CP. The current results specifically reflect the researchers' perspective and will serve as candidate categories to consider in the development of an ICF Core Set for adults with CP. WHAT THIS PAPER ADDS: Outcomes studied in adults with cerebral palsy are captured in varying International Classification of Functioning, Disability and Health (ICF) categories. Activities and participation were studied more frequently than body functions and structures. Mobility, employment, and self-care were most frequent within ICF's 'Activities and participation' component. Pain and neuromusculoskeletal and movement-related functions were most frequent within 'Body functions'.

Multimorbidity risk assessment in adolescents and adults with cerebral palsy: a protocol for establishing a core outcome set for clinical research and practice.

BACKGROUND: Estimates of multimorbidity, defined as the presence of at least two chronic conditions, some of which attributable to modifiable behaviours, are high in adults with cerebral palsy (CP). An assessment protocol evaluating multimorbidity risk is needed in order to develop and evaluate effective interventions to optimize lifelong health in individuals with CP. The aim of this protocol paper is to describe the development of a core outcome set (COS) for assessing multimorbidity risk in adolescents and adults with CP, to be used in clinic and research. METHODS: The expert consortium will first define the target population and outcomes to be measured. Through a process of literature review and an international Delphi survey with expert clinicians and researchers, we will then determine which outcome measurement instruments (OMIs) can best measure those outcomes. The resulting OMIs will be used in a feasibility study with adolescents and adults with CP from an international clinical research network. Finally, a face-to-face stakeholder meeting with adolescents and adults with CP, their families/caregivers and researchers and clinicians who are experts in CP, will be organized to reach final agreement on the COS. DISCUSSION: This COS will guide clinicians and researchers in assessing multimorbidity risk in adolescents and adults with CP. The inclusion of experts and individuals with CP from international locations for establishing the COS lends strong support to its generalizability. Evidence of its feasibility and approval from all stakeholders will enable implementation in clinical practice, and guide future research using the COS in individuals with CP.

Low bone mineral density in ambulatory persons with cerebral palsy? A systematic review.

Purpose: Non-ambulatory persons with cerebral palsy are prone to low bone mineral density. In ambulatory persons with cerebral palsy, bone mineral density deficits are expected to be small or absent, but a consensus conclusion is lacking. In this systematic review bone mineral density in ambulatory persons with cerebral palsy (Gross Motor Function Classification Scales I-III) was studied. Materials and methods: Medline, Embase, and Web of Science were searched. According to international guidelines, low bone mineral density was defined as Z-score ≤ -2.0. In addition, we focused on Z-score ≤ -1.0 because this may indicate a tendency towards low bone mineral density. Results: We included 16 studies, comprising 465 patients aged 1-65 years. Moderate and conflicting evidence for low bone mineral density (Z-score ≤ -2.0) was found for several body parts (total proximal femur, total body, distal femur, lumbar spine) in children with Gross Motor Function Classification Scales II and III. We found no evidence for low bone mineral density in children with Gross Motor Function Classification Scale I or adults, although there was a tendency towards low bone mineral density (Z-score ≤ -1.0) for several body parts. Conclusions: Although more high-quality research is needed, results indicate that deficits in bone mineral density are not restricted to non-ambulatory people with cerebral palsy. Implications for Rehabilitation Although more high-quality research is needed, including adults and fracture risk assessment, the current study indicates that deficits in bone mineral density are not restricted to non-ambulatory people with CP. Health care professionals should be aware that optimal nutrition, supplements on indication, and an active lifestyle, preferably with weight-bearing activities, are important in ambulatory people with CP, also from a bone quality point-of-view. If indicated, medication and fall prevention training should be prescribed.

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.

Factors contributing to the longitudinal development of social participation in individuals with cerebral palsy.

AIMS: We aimed to determine factors associated with the longitudinal development of social participation in a Dutch population of individuals with Cerebral Palsy (CP) aged 1-24 years. METHODS AND PROCEDURES: For this multicentre prospective longitudinal study, 424 individuals with CP aged 1-24 years were recruited from various rehabilitation centers in The Netherlands. Social participation was measured with the Vineland Adaptive Behavior Scales. We assessed associations with age, intellectual impairment, level of gross motor function, gender, type of CP, manual ability, epilepsy, hearing-, visual-, speech impairment and pain, internalizing- and externalizing behavioral problems, type of education and parental level of education. Each individual was measured 3 or 4 times. The time between measurements was 1 or 2 years. OUTCOMES AND RESULTS: Epilepsy and speech impairment were each independently associated with the longitudinal development of social participation. The effects were rather small and did not change with age. Also, a trend was found that children attending special education develop less favorably in social participation. CONCLUSIONS AND IMPLICATIONS: Our results might provide parents and caregivers with starting points to further develop tailored support for individuals with epilepsy, with speech impairment and/or attending special education at risk for suboptimal social participation.