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 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.

"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.

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.

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.

Bimanual Fine Motor Function (BFMF) Classification in Children with Cerebral Palsy: Aspects of Construct and Content Validity.

UNLABELLED: The Bimanual Fine Motor Function (BFMF) is currently the principal classification of hand function recorded by the Surveillance of Cerebral Palsy in Europe (SCPE) register. The BFMF is used in a number of epidemiological studies, but has not yet been validated. AIMS: To examine aspects of construct and content validity of the BFMF. METHODS AND RESULTS: Construct validity of the BFMF was assessed by comparison with the Manual Ability Classification System (MACS) using register-based data from 539 children born 1999-2003 (304 boys; 4-12 years). The high correlation with the MACS (Spearman's rho = 0.89, CI: 0.86-0.91, p<.001) supports construct validity of the BFMF. The content of the BFMF was appraised through literature review, and by using the ICF-CY as a framework to compare the BFMF and MACS. The items hold, grasp and manipulate were found to be relevant to describe increasingly advanced fine motor abilities in children with CP, but the description of the BFMF does not state whether it is a classification of fine motor capacity or performance. CONCLUSION: Our results suggest that the BFMF may provide complementary information to the MACS regarding fine motor function and actual use of the hands, particularly if used as a classification of fine motor capacity.

The stability of the Manual Ability Classification System over time.

AIM: To evaluate the stability over time of the Manual Ability Classification System (MACS) levels. METHOD: The study group comprised 1267 children with cerebral palsy (746 males, 521 females) who were followed from 2005 to 2010 with two or more registered MACS classifications rated at least 1 year apart. Thirty-five percent of the children (n=445) had four MACS registrations. The children were between 4 and 17 years old at their first rating, The stability over time was also compared between children who were younger (4y of age) or older (≥10y) at the time of their first classification. RESULTS: An excellent stability was found between two ratings at 1-year intervals with an intraclass correlation coefficient (ICC) of 0.97 (95% CI 0.97-0.97) and 82% agreement (n=1267). The stability was also excellent for two ratings performed 3 to 5 years apart (ICC 0.96; 95% CI 0.95-0.97) with an agreement of 78% (n=445). Across four ratings, 70% of the children remained at the same level. The results were similar for younger and older children, indicating that stability was not influenced by age. INTERPRETATION: This study provides evidence that MACS levels are stable over time and that the classification has predictive value.

Exploration of the relationship between the Manual Ability Classification System and hand-function measures of capacity and performance.

PURPOSE: To further investigate the construct of Manual Ability Classification System (MACS) by evaluating the relationship between children's designated MACS levels and their outcomes on two different tests of hand function, measuring capacity and performance, respectively. Another aim was to use the International Classification of Functioning, Disability and Health-Child and Youth version (ICF-CY) as a framework to explore the uniqueness of the assessments. METHOD: Ninety-one children with cerebral palsy in MACS levels I-V, aged 5-17 years (mean 9.8, SD 3.0) participated. Data were collected using MACS, ABILHAND-Kids and Box and Block Test. RESULTS: A strong association between MACS and ABILHAND-Kids (rs = -0.88, p < 0.05) and MACS and Box and Block Test (rs = -0.81, p < 0.05) was demonstrated. Children's performance differed significantly between the different MACS levels (ABILHAND-Kids F (4:86) = 103.86, p < 0.001, Box and Block Test F (4:86) = 59.18, p < 0.001). The content comparison with ICF-CY, as a frame of reference, showed that these instruments capture fine hand use in the activity and participation component. The linking of the instruments to various ICF-CY categories demonstrated conceptual differences between the instruments. MACS had the broadest representation of ICF-CY domains. CONCLUSIONS: This study strengthens the construct, and thereby the validity, of MACS as a classification of children's hand function, expressed by the handling of objects in everyday activities in their daily environments. IMPLICATIONS FOR REHABILITATION: • This study has strengthened the evidence of Manual Ability Classification System (MACS) as being a valid and useful classification of children's hand function. • The various MACS levels describe different degrees of hand-function impairment. • MACS give a broad description of children's manual ability in a variety of daily life domains.

Self-care and mobility skills in children with cerebral palsy, related to their manual ability and gross motor function classifications.

AIM: The aim of this study was to investigate the acquisition of self-care and mobility skills in children with cerebral palsy (CP) in relation to their manual ability and gross motor function. METHOD: Data from the Pediatric Evaluation of Disability Inventory (PEDI) self-care and mobility functional skill scales, the Manual Ability Classification System (MACS), and the Gross Motor Function Classification System (GMFCS) were collected from 195 children with CP (73 females, 122 males; mean age 8 y 1 mo; SD 3 y 11 mo; range 3-15 y); 51% had spastic bilateral CP, 36% spastic unilateral CP, 8% dyskinetic CP, and 3% ataxic CP. The percentage of children classified as MACS levels I to V was 28%, 34%, 17%, 7%, and 14% respectively, and classified as GMFCS levels I to V was 46%, 16%, 15%, 11%, and 12% respectively. RESULTS: Children classified as MACS and GMFCS levels I or II scored higher than children in MACS and GMFCS levels III to V on both the self-care and mobility domains of the PEDI, with significant differences between all classification levels (p<0.001). The stepwise multiple regression analysis verified that MACS was the strongest predictor of self-care skills (66%) and that GMFCS was the strongest predictor of mobility skills (76%). A strong correlation between age and self-care ability was found among children classified as MACS level I or II and between age and mobility among children classified as GMFCS level I. Many of these children achieved independence, but at a later age than typically developing children. Children at other MACS and GMFCS levels demonstrated minimal progress with age. INTERPRETATION: Knowledge of a child's MACS and GMFCS level can be useful when discussing expectations of, and goals for, the development of functional skills.

Parents' and therapists' perceptions of the content of the Manual Ability Classification System, MACS.

The Manual Ability Classification System (MACS) is a newly developed five-level classification that describes how children with cerebral palsy (CP) use their hands when handling objects in daily life. Since the MACS level is to be determined by asking the parents or someone else who knows the child well, it is important that the classification is meaningful and easy to understand. The aim of this study was to investigate the content validity based on parents' and therapists' descriptions of the children's ability to use their hands in daily manual tasks. Twenty-five children were represented by a parent and a therapist. After a short presentation of MACS, the respondents rated the child's MACS level. Subsequently, in a short interview, they described their thoughts about the child's ability, the classification, and the scoring process. Parents and therapists found that MACS gives a good description of how children with CP use their hands in daily activities. They found the differences between the five levels meaningful and generally easy to determine. A unique description of the children's ability at each level confirmed the validity. This provided evidence which strengthened the content validity of MACS in a descriptive way. Additional information complementing the existing leaflet could facilitate the scoring process.

The Manual Ability Classification System (MACS) for children with cerebral palsy: scale development and evidence of validity and reliability.

The Manual Ability Classification System (MACS) has been developed to classify how children with cerebral palsy (CP) use their hands when handling objects in daily activities. The classification is designed to reflect the child's typical manual performance, not the child's maximal capacity. It classifies the collaborative use of both hands together. Validation was based on the experience within an expert group, a review of the literature, and thorough analysis of children across a spectrum of function. Discussions continued until consensus was reached, first about the constructs, then about the content of the five levels. Parents and therapists were interviewed about the content and the description of levels. Reliability was tested between pairs of therapists for 168 children (70 females, 98 males; with hemiplegia [n=52], diplegia [n=70], tetraplegia [n=19], ataxia [n=6], dyskinesia [n=19], and unspecified CP [n=2]) between 4 and 18 years and between 25 parents and their children's therapists. The results demonstrated that MACS has good validity and reliability. The intraclass correlation coefficient between therapists was 0.97 (95% confidence interval 0.96-0.98), and between parents and therapist was 0.96 (0.89-0.98), indicating excellent agreement.