Spina Bifida Global Learning Collaborative: Educating the next generation of clinicians, researchers, and advocates.

PURPOSE: This project aimed to launch an international learning community to guide the development of a spina bifida (SB) curriculum for global health trainees. METHODS: Using a descriptive study design, a convenience sample of SB curricula were identified in 2022-23 by members of the Spina Bifida World Congress Outreach Committee and evaluated during a series of monthly Zoom calls to discuss SB education in a global health context. Participants included (1) leadership from the ReachAnother Foundation, (2) invited panelists from the Spina Bifida World Congress Global Health Symposium, and (3) global health students and preceptors. Education initiatives in Ethiopia, Sweden, Argentina, Ecuador, and the United States were evaluated vis-à-vis format and content. RESULTS: All of the education initiatives referenced the framework of the World Health Organization International Classification of Functioning, Disability and Health. Formats varied and included both virtual and interactive workshops, print materials, videos, and guides for small group discussion. Content addressed four domains: Folate Prevention, Neurosurgical Training, After Care, and Data Collection. A multidisciplinary approach, partnerships with families, and workforce pipeline training were identified as guiding themes for educating the next generation of SB researchers and clinicians in global health settings. CONCLUSION: The Spina Bifida Global Learning Collaborative is a transnational group of advocates, clinicians, and investigators whose mission is the advancement of SB-related global health education. Lessons learned from the collaborative are being leveraged to develop a global health curriculum for learners, which may improve services for individuals with SB around the globe.

Intervention with hand orthosis: experience from boys with Duchenne muscular dystrophy and their parents.

PURPOSE: To identify and describe possible ways of experiencing an intervention with hand orthoses in a study group including boys with Duchenne muscular dystrophy (DMD) and their parents. To capture the experiences of the persons directly involved as well as their family members. MATERIAL AND METHODS: Eight boys with DMD (aged 8-21; median age 17.7) and five of their parents were interviewed immediately post-intervention. Additionally, follow-up interviews were later performed with five boys and three parents. RESULTS: Ten categories of intervention experiences emerged and were allocated to three aspects: "Prerequisites in the treatment situation", "The intervention makes a difference" and "Instilling hope for the future". The requirements for intervention success include co-operation with parents and hand orthoses with a good fit. Maintained or increased joint mobility, reduced pain and improved occupational performance were experienced. CONCLUSION: The boys and parents perceived that the intervention with hand orthoses could counteract the deterioration of the boys' hands. This instilled hope of preserving occupational performance throughout life. They also considered that a good hand-orthosis fit, appropriate adjustments to daily routines and good co-operation with the people around them were important for the intervention to be and remain successful.

Preserving the ability to perform activities with the hands is of paramount importance for boys with Duchenne muscular dystrophy and their parents.Intervention with hand orthoses counteracts deterioration of the hands.Successful treatment with hand orthoses requires that specific prerequisites in the treatment situation are met, that the treatment makes a difference for activity performance and that it instils hope for the future when it comes to performing meaningful activities.

Intervention with the CO-OP Approach leads to a transfer effect over time to untrained goals for children with cerebral palsy or spina bifida.

PURPOSE: This study aims to investigate whether the treatment effects, in terms of goal attainment, transfer effects and impact on executive functions, of an intervention in children with cerebral palsy or spina bifida using the Cognitive Orientation to daily Occupational Performance (CO-OP) Approach are maintained over time, from immediately after the intervention to three months afterwards. METHOD: A three-month follow-up study, from an intervention using CO-OP. Thirty-four children (7-16 years) each identified four goals (one untrained to examine transfer) and participated in an eleven-session intervention. Assessments were performed at baseline, immediately after the intervention and at a three-month follow-up using the Canadian Occupational Performance Measure and the Performance Quality Rating Scale. Executive function and self-rated competence were assessed at the same timepoints. RESULTS: Statistically significant and clinically relevant improvements in goal achievement were demonstrated for both trained and untrained goals after the intervention and were maintained at follow-up. The clinically relevant improvement in untrained goals continued to increase until follow-up. Self-rated competence increased after the intervention and was maintained at follow-up. CONCLUSION: The CO-OP intervention was effective in achieving and maintaining the children's own goals over time. The transfer effect was confirmed by higher goal attainment for the untrained goals.

The children's self-defined goals were achieved after the Cognitive Orientation to daily Occupational Performance (CO-OP) intervention and remained so at the three-month follow-up.The CO-OP Approach is useful for children with cerebral palsy or spina bifida.A transfer effect was demonstrated for untrained goals by both subjective and objective assessments.Using children's self-defined goals is effective.

The Cognitive Orientation to daily Occupational Performance (CO-OP) Approach is superior to ordinary treatment for achievement of goals and transfer effects in children with cerebral palsy and spina bifida - a randomized controlled trial.

PURPOSE: Children with cerebral palsy (CP) or spina bifida (SB) often have executive dysfunction affecting activity performance. With the Cognitive Orientation to daily Occupational Performance (CO-OP) Approach, children find their own way to perform activities, using problem-solving strategies and meta-cognitive thinking. The present study aimed to investigate the effectiveness of the CO-OP Approach in children with CP or SB, compared with conventional rehabilitation, in achieving self-identified activity goals, and to explore any generalization and transfer effects. METHOD: Randomized controlled trial, CO-OP versus treatment as usual, 38 children (7-16 years) participated. Each child identified four goals (to study generalization and transfer, one remained untrained). Primary outcomes: Canadian Occupational Performance Measure (COPM) and Performance Quality Rating Scale (PQRS). Secondary outcomes assessed executive functions and self-rated everyday-life competence. RESULTS: Self-rated goal attainment (COPM) was significantly greater for both trained and untrained goals in the CO-OP group compared with the control group. The rating of observed performance (PQRS) was significantly higher for trained goals in the CO-OP group. The CO-OP group experienced fewer problems in everyday life after treatment. Executive functions did not differ significantly between groups. CONCLUSION: CO-OP is more effective than ordinary treatment in achieving both trained and untrained goals.IMPLICATIONS FOR REHABILITATIONCO-OP enables children with CP (MACS levels I-III) or SB without intellectual disabilities to reach self-identified goals.CO-OP shows transfer effects to new activities and situations, which may enhance children's self-efficacy.CO-OP is an important complement to conventional rehabilitation services for children with CP and SB.

A scoping review of cognition in spina bifida and its consequences for activity and participation throughout life.

AIM: The aim of this scoping review was to summarise findings concerning cognitive characteristics in people with spina bifida and explain how cognitive factors influence activities and participation in different areas and stages of life. METHODS: PubMed, Psych INFO, ERIC, Scopus, CINAHL and the Cochrane Library were searched for English language papers published in 2000-2018. A total of 92 papers were selected and quality was assessed according to the McMaster criteria. The results were presented related to body functions, activity and participation from the International Classification of Function and Health, ICF. RESULTS: People with spina bifida tended to have a lower IQ than those without. The majority also had cognitive difficulties manifested in problems with language, perception, memory, executive and attentional functions. Those difficulties affected activity and participation in all life domains in ICF. This may affect medical adherence and responsibility and by extension the prevention of secondary complications. CONCLUSION: It is important for caregivers, professionals and especially individuals with spina bifida themselves to understand and handle both physical and cognitive consequences in all life circumstances. Having insight into one's own assets and difficulties paves the way to managing life challenges, which could enhance health, self-management and participation in society.

Managing to learn bimanual activities - experiences from children and adolescents with cerebral palsy - a qualitative analysis.

PURPOSE: Children and adolescents with cerebral palsy often have impaired hand function. This makes it difficult for them to deal with everyday activities. The aim of the study was to explore the experiences of children and adolescents with unilateral spastic cerebral palsy when it comes to learning and dealing with activities requiring bimanual use. METHOD: Ten participants, attending mainstream schools, with unilateral spastic cerebral palsy (10-18 years, MACS-level I-III) took part in semi-structured interviews. Qualitative analysis with verbatim transcripts were analysed using a Grounded Theory approach. RESULTS: The learning of bimanual activities was described as a process taking place in interaction with the dynamics of everyday situations. Five categories describing the participants experiences emerged: "Reaching a point where you want to learn", "Awareness and acceptance of your own abilities", "Dealing with the boundaries of the disability", "Dealing with the impact of people around you" and "Strategies for learning". A multi-dimensional theory was derived, summarising how the participants learned bimanual activities in daily life. CONCLUSIONS: Children and adolescents with unilateral spastic cerebral palsy express that the process of learning bimanual activities can only take place when it fits in with life as it unfolds. Thus, they have to adapt to a changing context and their own developing skills.Implications for RehabilitationThis study support a person-centred perspective in the process of learning bimanual activities.Interventions need to be tailored to life situations and embedding in child's and adolescents everyday lives.The therapist must be compliant with the child's and adolescents desire for learning.

"With CO-OP I'm the boss" - experiences of the cognitive orientation to daily occupational performance approach as reported by young adults with cerebral palsy or spina bifida.

Purpose: Restrictions to activity and participation in persons with cerebral palsy or spina bifida are often due to both motor and executive dysfunction. Hence methods focusing solely on motor issues are not enough to enhance participation. The Cognitive Orientation to daily Occupational Performance ApproachTM is a performance-based approach offering clients opportunities to create their own strategies to learn skills. The aim of the present study was to explore and describe experiences of the Cognitive Orientation to daily Occupational Performance Approach as reported by young adults with cerebral palsy or spina bifida.Methods: Qualitative content analysis was used. Semi-structured individual interviews were conducted with the 10 participants aged 16-28, post-intervention and at 6-months follow-up.Results: The participants described how the Cognitive Orientation to daily Occupational Performance Approach enhanced their self-efficacy. Four categories describing the participants' experiences emerged: "CO-OP is a different way of learning", "CO-OP sometimes puts a strain on me", "CO-OP supports my way of thinking and doing" and "CO-OP boosts me".Conclusion: The young adults expressed that the Cognitive Orientation to daily Occupational Performance intervention, although sometimes challenging, was worth the effort because it provided them with an opportunity to master everyday-life problems by using meta-cognitive thinking, which enhanced their self-efficacy.Implications for rehabilitationThe Cognitive Orientation to daily Occupational Performance Approach™ - was perceived to provide opportunities to master everyday-life problems by using meta-cognitive thinking.The Cognitive Orientation to daily Occupational Performance Approach boosted the persons feeling of self-efficacy.The Cognitive Orientation to daily Occupational Performance Approach is person-centred and supports the person's own way of learning.

Long-term effects of repeated botulinum neurotoxin A, bimanual training, and splinting in young children with cerebral palsy.

AIM: To investigate long-term development of hand function after repeated botulinum neurotoxin A (BoNT-A) and occupational therapy at a young age. METHOD: Twenty children with unilateral spastic cerebral palsy (CP) (14 males, six females; median inclusion age 3y 1mo, range 1y 11mo-4y 3mo) participated in this longitudinal study. Ten children received occupational therapy after a randomized controlled trial and 10 repeated BoNT-A plus occupational therapy during 1-year. The Assisting Hand Assessment (AHA) and active supination, assessed the following 3 years. The assessments were compared with data from a reference group to investigate development over time. RESULTS: The improvement in AHA (7.5 AHA units) after BoNT-A plus occupational therapy was maintained at final follow-up. The occupational therapy group, unchanged after 1-year, improved by 5 AHA units (96% confidence interval [CI] 2-10), thus there was no difference between the groups. Median active supination increased in comparison with the reference group. In the BoNT-A/occupational therapy group, 9 out of 10 (97.85% CI 45 115) children improved in active supination. In the occupational therapy group, 7 out of 10 (97.85% CI -2 to 68) children improved in active supination. No correlation between active supination and AHA was found. INTERPRETATION: Bimanual performance achieved after BoNT-A plus occupational therapy was maintained, while it increased by follow-up in the occupational therapy group, suggesting that combined intervention gave earlier access to bimanual skills. Active supination was unrelated to AHA. WHAT THIS PAPER ADDS: Children whose bimanual performance improved after botulinum neurotoxin A and/or occupational therapy, maintained skills or progressed during follow-up. Bimanual performance increased with age, similar to a quality-register reference group. Increased active supination after intervention improved at follow-up but was not related to bimanual performance.

EFECTO A LARGO PLAZO DE LA ADMINISTRACIÓN REPETIDA DE NEUROTOXINA BOTULÍNICA A, ENTRENAMIENTO BIMANUAL Y FERULIZACIÓN EN NIÑOS CON PARÁLISIS CEREBRAL: OBJETIVO: Investigar el desarrollo de la funcionalidad de la mano a largo plazo, tras la administración repetida de neurotoxina botulínica A (NTBA) y la terapia ocupacional a edades tempranas. MÉTODO: En este estudio longitudinal participaron 20 niños con parálisis cerebral espástica unilateral (PC) (14 varones, 6 mujeres, edad media de inclusión 3 años 1 mes, con un rango de 1 año 11meses - 4años 3 meses). Diez niños recibieron terapia ocupacional después de un ensayo aleatorio controlado y 10 recibieron NTBA más terapia ocupacional durante un año. Se utilizó la escala de Evaluación de apoyo de la Mano (Assisting Hand Assesment AHA, siglas en inglés) y la supinación activa, se evaluó el seguimiento por 3 años. Las evaluaciones fueron comparadas con datos de un grupo de referencia para investigar el desarrollo con el tiempo. RESULTADOS: La mejoría en AHA (7,5 unidades AHA) después de NTBA en conjunto con terapia ocupacional se mantuvo hasta el final del seguimiento. El grupo de terapia ocupacional se mantuvo sin cambios después de 1 año, mejoró 5 unidades AHA (96% de Intervalo de confianza [IC] 2-10) por lo que no hubo diferencia entre los grupos. La mediana en la supinación activa incremento en comparación al grupo de referencia. En el grupo NTBA/terapia ocupacional, 9 de 10 (97,85% IC 45; 115) niños, mejoraron en la supinación activa. En el grupo de la terapia ocupacional 7 de cada 10 (97,85% CI -2: 68) niños mejoraron en la supinación activa. No hubo correlación entre la supinación activa y la AHA. INTERPRETACIÓN: El rendimiento bimanual alcanzado después de la NTBA en conjunto con la terapia ocupacional fue sostenida, mientras que aumentó tras el seguimiento en el grupo de terapia ocupacional, sugiriendo que la intervención combinada brinda un acceso temprano a las habilidades bimanuales. La supinación activa no estuvo relacionada con AHA.

EFEITOS DE LONGO PRAZO DA NEUROTOXINA BOTULÍNICA A REPETIDA, TREINO BIMANUAL, E USO DE ÓRTESES EM CRIANÇAS PEQUENAS COM PARALISIA CEREBRAL: OBJETIVO: Investigar o desenvolvimento em longo prazo da função manual após aplicação repetida de neurotoxina botulínica A (NTBo-A) e terapia ocupacional em idade precoce. MÉTODO: Vinte crianças com paralisia cerebral (PC) unilateral espástica (14 do sexo masculino, seis do sexo feminino; idade mediana na inclusão de 3a 1m, variação 1a 11m-4a 3m) participaram neste estudo longitudinal. Dez crianças receberam terapia ocupacional após um estudo clínico randomizado controlado e 10 receberal NTBo-A e terapia ocupacional durante 1 ano. A avaliação da mão auxiliar (AHA, na versão original) e a supinação ativa foram avaliados após 3 anos. As avaliações foram comparadas com dados de um grupo de referência para investigar o desenvolvimento ao longo do tempo. RESULTADOS: A melhora no AHA (7,5 unidades do AHA) após NTBo-A mais terapia ocupacional foi mantida ao final do acompanhamento. O grupo de terapia ocupacional, sem mudanças após 1 ano, melhorou 5 unidades do AHA (intervalo de confiança [IC] a 96% 2-10), portanto não houve diferença entre os grupos. A supinação ativa mediana aumentou em comparação com o grupo de referência. No grupo NTBo-A /terapia ocupacional, 9 em 10 (IC 97,85% 45; 115) crianças melhoraram a supinação ativa. No grupo terapia ocupacional, 7 em 10 (IC 97,85% 2; 68) crianças melhoraram a supinação ativa. Nenhuma correlação entre supinação ativa e AHA foi encontrada. INTERPRETAÇÃO: O desempenho bimanual obtido após NTBo-A mais terapia ocupacional foi mantido, e aumentou na avaliação de acompanhamento no grupo de terapia ocupacional, sugerindo que a intervenção combinada de acesso mais cedo a habilidades bimanuais. A supinação ativa não se relacionou com o AHA.

Evidence of Construct Validity for the Modified Mental Fatigue Scale When Used in Persons with Cerebral Palsy.

Introduction: Fatigue impacts negatively on everyday activities in individuals with cerebral palsy (CP). More knowledge is needed about how mental fatigue is manifested in this target group. The purpose of this study was to gather evidence about the validity of the modified Mental Fatigue Scale (m-MFS) in adults with CP.Methods: Mixed sequential exploratory design. The respondents were ten persons aged 22-56 with CP (MACS I-II).Results: The respondents perceived the m-MFS as easy to read and understand. Its structure was characterised as straightforward and the text of the rating options was deemed to assist identification with life situations. Very good agreement was seen between the respondents' and the instrument designers' intended meaning for the items in the m-MFS; the weighted kappa was 0.92.Conclusion: This study showed evidence of construct validity, based on response processes and content, for use of the modified MFS in adults with CP.

Potential benefits of the cognitive orientation to daily occupational performance approach in young adults with spina bifida or cerebral palsy: a feasibility study.

Purpose: People with cerebral palsy (CP) or spina bifida (SB) often struggle to perform everyday-life activities. Both groups frequently also have difficulties in creating and using strategies effectively when performing tasks. The cognitive orientation to daily occupational performance (CO-OP) Approach combines the learning of cognitive strategies with task-specific approaches through a client-centred procedure. The aim of this study was to investigate whether the CO-OP Approach is feasible for and potentially beneficial to adolescents and young adults with CP or SB in Sweden by analysing four areas of feasibility (acceptability, efficacy, adaptation, and expansion).Methods: Exploratory multiple-case study using mixed methods. Ten persons aged 16-28, five with each condition, participated in an intervention period. Assessments were performed on three occasions: baseline, post-intervention, and six-month follow-up.Results: The result demonstrates that the CO-OP Approach has the potential to enable adolescents and young adults with either condition to achieve personal goals and to enhance their planning skills and their ability to use strategies when performing activities. This approach is also compatible with the core values of habilitation in Sweden and was found by the participants to be highly meaningful and useful.Conclusions: The CO-OP Approach is feasible for adolescents and young adults with SB or CP in Sweden.Implications for rehabilitationThe Cognitive Orientation to daily Occupational Performance• is a feasible approach for adolescents and young adults with spina bifida and with cerebral palsy.• is a promising approach when it comes to enabling the achievement of personal goals.• might have potential to enhance executive functioning through strategy use.• is in line with the fundamental core values of disability rights of inclusion, empowerment, and participation.

Daily activity performance in congenital and childhood forms of myotonic dystrophy type 1: a population-based study.

AIMS: To identify and describe the profile characterizing motor and process skills during daily activity performance in individuals with congenital and childhood forms of myotonic dystrophy type 1 (DM1) and to investigate differences in performance between subgroups. METHOD: Sixty participants (34 males, 26 females, mean age=17y 8mo, SD=6y 0mo, range 5y 8mo-29y 0mo) were divided into severe congenital (n=9), mild congenital (n=20), and childhood (n=31) DM1 subgroups. Daily activity performance was evaluated using a standardized observational instrument: the Assessment of Motor and Process Skills. RESULTS: Deficits in performance were more pronounced in process than motor skills. Performance more than 2 SDs below age-specific norms was seen in 65% of participants for process skills and 33% of participants for motor skills. The cut-off scores indicated a potential need for assistance in daily activities for 79% of participants older than 18 years of age (n=28) due to deficient process skills. INTERPRETATION: Extensive deficits in daily activity performance were found in congenital and childhood forms of DM1, mainly owing to deficient process skills. Such skills impact on the ability to perform daily activities and could explain dependency in individuals with DM1. Process skills should be considered when evaluating daily activity performance. WHAT THIS PAPER ADDS: Young people with myotonic dystrophy type 1 show deficits in motor and process skills when performing daily activities, compared with normative data. Deficits in process skills were more pronounced than deficits in motor skills.

When I do, I become someone: experiences of occupational performance in young adults with cerebral palsy.

PURPOSE: Persons with cerebral palsy, even if they have relatively good motor functions, have a lower level of independence and participation in everyday activities than persons of the same age without disabilities. However, there are few descriptions of how persons with cerebral palsy themselves perceive their performance of activities in everyday life. The aim of this study was to describe the perceptions that young adults with cerebral palsy have of occupational performance in everyday life. METHODS: This qualitative interview study includes 10 participants with cerebral palsy classified with Manual Ability Classification System level I-II, aged 19-30 years. The data were analyzed using a phenomenographic approach. RESULTS: The interviews resulted in five categories: "Important to do"; "Demanding but can be facilitated"; "Excludes or includes"; "Diminishes me or makes me grow"; and "Comes at a price". CONCLUSIONS: The young adults with cerebral palsy consider that, despite life being so demanding, it is extremely important to perform activities themselves and to feel included, as this enables personal growth. Hence, it is necessary to advance intervention methods based on personally important activities to enable individuals with cerebral palsy to find their own way to perform activities. Further research is needed to increase opportunities for individuals with cerebral palsy to perform everyday activities without too much fatigue and struggle. Implications for Rehabilitation For young adults with cerebral palsy it is extremely important to perform everyday activities independently; by DOING activities they form their identity. Intervention models aimed to enable persons with cerebral palsy to be involved and find their own way to perform everyday activities should be emphasized. Attention must be paid to how mental fatigue is manifested in persons with cerebral palsy. To build self-awareness and self-efficacy, individuals with cerebral palsy need information, early in life, about cerebral palsy and the multifaceted difficulties the disability might lead to.

Gaze-controlled communication technology for children with severe multiple disabilities: Parents and professionals' perception of gains, obstacles, and prerequisites.

The aim of this study was to explore parents' and professionals' thoughts of how a gaze-controlled computer can be beneficial to children with severe multiple disabilities. All systems were provided primarily for symbol-based communication, but were also used for other purposes such as play, leisure and school activities. A further aim was to investigate factors affecting usability, specifically for communication. The study used a qualitative approach, involving content analysis of semistructured interviews with the children's key persons (N = 11). The analysis yielded three categories and twelve subcategories. There were gains for the children in terms of empowerment, social interaction, learning opportunities and efficient computer use. Inaccessibility, liability issues and technical failure were seen as obstacles, while the prerequisites included time, collaboration, stimulating content, know-how and opportunities. To sum up, this study suggests that gaze-controlled technology can provide children who have multiple disabilities involving severe motor dysfunction and communicative and cognitive problems with new opportunities to communicate, interact and perform activities independently, as long as conditions are right.

How children with cerebral palsy master bimanual activities from a parental perspective.

BACKGROUND: During childhood, children learn the daily life activities they want and need to do. Children with unilateral spastic cerebral palsy often have difficulties performing activities requiring two hands. AIM: To describe parental reasoning on how children with unilateral spastic cerebral palsy learn to master the performance of bimanual activities in everyday life. MATERIAL AND METHODS: Sixteen parents participated in focus groups, a qualitative research approach with its own methodological criteria and research methods. RESULTS: One overall theme emerged from the analysis: 'Finding harmony between pleasure and effort is the key to learning'. This overall theme arose as a synthesis of four themes: 'awakening of the inner drive', 'trying on one's own', 'enabling things to work' and 'it must be worth the effort´. The parents described when an activity woke their children´s inner drive to perform. Their children also strived to develop their own way to perform an activity, sometimes with the support of others, still, some activities were not possible to learn. CONCLUSIONS: Occupational therapists and others in the children's environment have an important mission to support the children to find their own harmony between pleasure and effort and their individual key to success in learning bimanual everyday activities.

Daily time management in children with spina bifida.

PURPOSE: Spina bifida (SB) often results in a complex disability and can also cause cognitive dysfunction. No previous study has investigated the ability to adapt to time in children with SB. This ability is crucial for an individual's possibility to develop autonomy in life. The purpose of this study was to investigate whether children aged 10-17 with SB have lower time-processing abilities than typically-developing children, and to describe the profile of time-processing in children with SB. METHOD: Participants comprised a consecutive sample of 21 children (drawn from a geographical cohort of 45) aged 10-17 years (mean: 14 years, SD: 2 years); 13 were boys. The instruments used were KaTid-Y, Time-S, and Time-P. RESULTS: The children with SB had lower time-processing abilities than typically-developing children (52.4% under -2SD), particularly difficulties to orient to and to estimate objective time, to understand time perspectives and with time planning. They also self-rated low use of strategies to adapt to time. The parents rated their children as having extensive difficulties in daily time management. CONCLUSIONS: The low time-processing ability found in children with SB is likely to be an important contributing factor to low autonomy and independence.

Validity and test-retest reliability of Children's Hand-use Experience Questionnaire in children with unilateral cerebral palsy.

AIM: To investigate the validity of the internet-based version of the Children's Hand-use Experience Questionnaire (CHEQ) by testing the new four-category rating scale, internal structure, and test-retest reliability. METHOD: Data were collected for 242 children with unilateral cerebral palsy (CP) (137 males and 105 females; mean age 9y 10mo, SD 3y 5mo, range 6-18y). Twenty children from the study sample (mean age 11y 8mo, SD 3y 10mo) participated in a retest within 7 to 14 days. Validity was tested by Rasch analysis based on a rating scale model and test-retest reliability by Kappa analysis and intraclass correlation coefficient (ICC). RESULTS: The four-category rating scale was within recommended criteria for rating scale structure. One item was removed because of misfit. CHEQ showed good scale structure according to the criteria. The effective operational range was >90% for two of the CHEQ scales. Test-retest reliability for the three CHEQ scales was: grasp efficacy, ICC=0.91; time taken, ICC=0.88; and feeling bothered, ICC=0.91. INTERPRETATION: The internet-based CHEQ with a four-category rating scale is valid and reliable for use in children with unilateral CP. Further studies are needed to investigate the validity of the internet-based version of CHEQ for children with upper limb reduction deficiency or obstetric brachial plexus palsy and the validity of the recommended improvements to the current version.

Botulinum toxin A injections and occupational therapy in children with unilateral spastic cerebral palsy: a randomized controlled trial.

AIM: To investigate the effects of repeated botulinum toxin A (BoNT-A) injections combined with occupational therapy, including a splint, compared with occupational therapy alone on hand function in children with unilateral spastic cerebral palsy (USCP), in all International Classification of Functioning, Disability and Health (ICF) domains. METHOD: This was a randomized controlled study, population-based and evaluator-blinded for primary outcome (October 2004 to September 2010). Twenty children (14 males; median age 3y 1mo) with USCP, recruited at a rehabilitation centre in Sweden, were assigned to one of two parallel groups using concealed allocation. In the course of one year, 10 children received occupational therapy, while 10 received repeated BoNT-A plus occupational therapy (BoNT-A/OT). Primary outcome (Assisting Hand Assessment [AHA]), and secondary outcome measures (range of movement [ROM], and Canadian Occupational Performance Measure), were measured at baseline, 3, 6, 9, and 12 months. RESULTS: AHA revealed a superior effect in the BoNT-A/OT group at 12 months: 6 out of 10 improved compared with 1 out of 10 in the occupational therapy group (p<0.03). A 95% Peskun exact confidence interval for the difference in proportions is given as 0.01 to 0.81. Secondary outcomes improved in both groups. INTERPRETATION: Repeated BoNT-A/OT appeared superior to occupational therapy alone for bimanual performance in young children with USCP. Active ROM and goal performance improved in both groups.

Is autonomy related to the quality of performance of everyday activities in children with spina bifida?

PURPOSE: To investigate the relationship between the level of autonomy and the quality of performance of everyday activities in a population-based cohort of children with spina bifida and to study the agreement between the children's and the parents' ratings of autonomy. METHODS: 50 dyads of children (aged 6-14) with spina bifida and their parents rated the children's level of autonomy with an adapted, Swedish version of the Autonomy Scale from the Arc's Self-Determination Scale. Each child's quality of performance of everyday activities was assessed with the Assessment of Motor and Process Skills (AMPS). RESULTS: The autonomy levels of the children with spina bifida were rated to be lowest in daily routines and highest in leisure activities. Binary logistic regression analyses revealed that age, motor skills and process skills were all significantly related to the autonomy level, but that process skills appeared to predominate in this respect. Concerning the perception of the autonomy level, little agreement was found between each child and his/her parent. CONCLUSIONS: It is important to understand and support the development of process skills as expressed in task performance in children with spina bifida and to pay attention to both the parent's and the child's opinion when setting goals and plans for interventions.

Are there cross-cultural differences of ADL ability in children measured with the Assessment of Motor and Process Skills (AMPS)?

INTRODUCTION: In many studies of self-care assessments for children, cultural differences in age-norm values have been shown. No study has evaluated whether there are cross-cultural differences in ADL motor and/or process skills in children when measured with the Assessment of Motor and Process Skills (AMPS). AIM: To investigate if there were systematic differences in ADL ability measured with the AMPS between children from the Nordic countries and North America and to evaluate the applicability of the existing international age-normative values for children from these two regions. METHODS: Values from a total of 4 613 children, 3-15 years old, without known disabilities, from these geographical regions were compared with ANOVA. The difference in logits between each region and the mean values for each age group were calculated. RESULTS: No differences of relevance in age-related ADL ability measures between children from the two geographical regions were found, and the age-norm values are applicable to both regions. IMPLICATIONS: The AMPS may be considered free from cultural bias and useful in both clinical practice and research concerned with children in both the Nordic countries and North America.