Hand-Use-at-Home Questionnaire: validity and reliability in children with neonatal brachial plexus palsy or unilateral cerebral palsy.

OBJECTIVE: To investigate construct validity and test-retest reliability of the parent-rated Hand-Use-at-Home questionnaire (HUH) in children with neonatal brachial plexus palsy or unilateral cerebral palsy. DESIGN AND SUBJECTS: For this cross-sectional study, children with neonatal brachial plexus palsy or unilateral cerebral palsy, aged 3-10 years, were eligible. MAIN MEASURES: The HUH, Pediatric Outcome Data Collection Instrument Upper Extremity Scale (neonatal brachial plexus palsy only), and Children's Hand-Use Experience Questionnaire (unilateral cerebral palsy only) were completed. The HUH was completed twice in subgroups of both diagnoses. Lesion-extent (indication of involved nerve rootlets in neonatal brachial plexus palsy as confirmed during clinical observation and/or nerve surgery) and Manual Ability Classification System levels (unilateral cerebral palsy) were obtained from the medical records. Spearman correlation coefficients between the HUH and all clinical variables, agreement, standard error of measurement, smallest detectable change and intra-class correlation were calculated. RESULTS: A total of 260 patients participated (neonatal brachial plexus palsy: 181), of which 56 completed the second HUH (neonatal brachial plexus palsy: 16). Median age was 6.9 years for children with neonatal brachial plexus palsy, 116 had C5-C6 lesions. Median age for children with unilateral cerebral palsy was 6.4 years, 33 had Manual Ability Classification System Level II. The HUH correlated moderately with lesion-extent ( rs =-0.5), Pediatric Outcome Data Collection Instrument Upper Extremity Scale ( rs = 0.6) and Children's Hand-Use Experience Questionnaire ( rs = 0.5) but weakly with Manual Ability Classification System levels ( rs = -0.4). Test-retest reliability was excellent (intra-class correlation2,1 = 0.89, standard error of measurement = 0.599 and smallest detectable change = 1.66 logits) and agreement was good (mean difference HUH1 - HUH2 = 0.06 logits). CONCLUSION: The HUH showed good construct validity and test-retest reliability in children with neonatal brachial plexus palsy or unilateral cerebral palsy.

Responsiveness of Goal Attainment Scaling in comparison to two standardized measures in outcome evaluation of children with cerebral palsy.

OBJECTIVES: To assess the responsiveness of Goal Attainment Scaling compared with the Pediatric Evaluation of Disability Inventory (PEDI) and the 66-item Gross Motor Function Measure (GMFM-66) in multidisciplinary rehabilitation practice. DESIGN: Observational study. Pretest-posttest design. SUBJECTS/PATIENTS: Twenty-three children with cerebral palsy, aged 2-13 years. METHODS: Goal Attainment Scaling, PEDI and GMFM-66 assessments were performed before and after six months of treatment. Physical, occupational and speech therapists constructed and scored 6-point Goal Attainment Scaling scales meeting predetermined criteria, describing the main functional goal per discipline. The contents of the three measures were compared using International Classification of Functioning, Disability and Health child and youth version (ICF-CY) codes. Spearman's rho correlations between Goal Attainment Scaling change scores per discipline and change scores obtained with the PEDI functional skills scales and GMFM-66 were calculated. Complete goal attainment was compared with significant change in terms of the standardized measures. RESULTS: Twenty per cent of the Goal Attainment Scaling items were not covered by items of the PEDI or the GMFM-66. Inconclusive correlations were found between Goal Attainment Scaling and PEDI change scores (r 0.28-0.64). Even after exclusion of the non-corresponding items, correlations were moderate (r 0.57-0.73). Of 39/64 Goal Attainment Scaling scales scored as complete goal attainment, 16 individual PEDI scores did not show change on the corresponding scale. Low correlation was found between Goal Attainment Scaling change scores and GMFM-66 change scores. CONCLUSION: Goal Attainment Scaling, PEDI and GMFM-66 were complementary in their ability to measure individual change over time in children with cerebral palsy. Using only the standardized instruments could have caused many individual rehabilitation goals actually attained being missed in the outcome evaluation.

Interrater reliability of goal attainment scaling in rehabilitation of children with cerebral palsy.

OBJECTIVES: To determine the interrater reliability of Goal Attainment Scaling (GAS) in the routine practice of interdisciplinary rehabilitation of children with cerebral palsy, and to examine the difference in the interrater reliability of the scores between GAS scales constructed by the children's own therapists and the scales constructed by independent therapists. DESIGN: Individually tailored GAS scales, based on predetermined criteria, were constructed at the start of a 6-month rehabilitation period. The outcome was rated independently by 2 therapists at the end of the treatment period. Two different data sets were acquired, one consisting of scores on GAS scales constructed by the children's own therapists, the other of scores on GAS scales constructed by matched independent raters of the same profession. SETTING: A children's unit of a medium-sized rehabilitation center in The Netherlands. PARTICIPANTS: Physical therapists (n=8), occupational therapists (n=8), and speech therapists (n=4) participated in pairs. They constructed 2 sets of 64 GAS scales each, for 23 children with cerebral palsy. INTERVENTIONS: A 6-month interdisciplinary pediatric rehabilitation program. MAIN OUTCOME MEASURE: Interrater reliability was assessed using linear-weighted Cohen's kappa. RESULTS: The scales constructed by the children's therapists had an interrater reliability of .82 (95% confidence interval [CI], .73-.91). The interrater reliability for scales constructed by the independent raters was .64 (95% CI, .49-.79). The main reason for disagreement between raters was discrepancies in the professionals' interpretation of the children's capacities versus their actual performance during assessment. CONCLUSIONS: The interrater reliability of GAS used under optimal conditions was good, particularly for scales constructed by the children's own therapists.

Health-care use and information needs of children with neonatal brachial plexus palsy: A cross-sectional survey among 465 Dutch patients.

To investigate health-care use (HCU) and information needs of children aged 0-18 years with neonatal brachial plexus palsy (NBPP), a cross-sectional study was performed. Patients and/or parents seen in our NBPP clinic were invited to complete a survey comprising questions on HCU due to NBPP and current information needs. Outcomes were described for three age-groups (0-1/2-9/10-18 years), based on follow-up status (early/late/no-discharge). Four hundred sixty-five parents/patients participated (59 in the 0-1, 226 in the 2-9, and 180 in the 10-18-year group). Two hundred ninety-three patients had C5-C6 lesions, 193 were discharged from follow-up, 83 of whom categorized as 'early discharged' (<1 year of age). Over the past year, 198 patients had contact with the expert team (49 in the 0-1, 81 in the 2-9, and 68 in the 10-18-year group) and 288 with at least one other health-care professional (53 in the 0-1, 133 in the 2-9, and 102 in the 10-18-year group). Of the 83 patients discharged early, 34 reported health-care use. Two hundred twenty-eight participants reported current information needs of whom 23 were discharged early. HCU and information needs of Dutch children with NBPP remains considerable even in children who were discharged. Stricter follow-up and information provision for these patients is needed.

Participation restrictions among adolescents and adults with neonatal brachial plexus palsy: the patient perspective.

PURPOSE: To examine the impact of neonatal brachial plexus palsy (NBPP) on societal participation of adolescents and adults. METHODS: This cross-sectional study was conducted among patients with NBPP, aged ≥16 years, who had visited our NBPP clinic. Patients completed questions on the influence of NBPP on their choices regarding education/work and their work-performance, the Impact on Participation/Autonomy questionnaire and the Utrecht Scale for Evaluation of Rehabilitation-Participation (USER-P). In addition, health-related quality of life (HRQoL) was assessed. RESULTS: Seventy-five patients participated (median age 20, inter quartile range 17-27). Twenty were full-time students, 28 students with a job, 21 employed, two unemployed, and four work-disabled. Sixty-six patients had had a job at some stage. Patients' overall HRQoL was comparable to the general population. 27/75 patients reported that NBPP had affected their choices regarding education and 26/75 those regarding work. 33/66 reported impact on their work performance. On the Impact on Participation/Autonomy questionnaire, 80% (49/61) reported restrictions in the work-and-education domain, 74% in social-relations and 67% in autonomy-outdoors. 37/61 reported participation restrictions on the USER-P. CONCLUSIONS: Although their overall HRQoL was not impaired, a substantial proportion of adolescent/adult patients reported that NBPP had an impact on choices regarding education and profession, as well as on work-performance. Restrictions in participation, especially in work and education were also reported. Guiding patients in making choices on education and work at an early stage and providing tailored physical as well as psychosocial care may prevent or address restrictions, which may improve participation. Implications for Rehabilitation Adolescent and adult patients with neonatal brachial plexus palsy perceive restrictions in societal participation, especially regarding the work-and-education domain. All patients with neonatal brachial plexus palsy may perceive restrictions in societal participation regardless of lesion severity, treatment history and side of the lesion. Adolescents and adults with neonatal brachial plexus palsy report that their choices regarding education and work, as well as their work-performance are influenced by their neonatal brachial plexus palsy. Patients with neonatal brachial plexus palsy should be followed throughout their life in order to provide them with appropriate information and treatment when health- or participation-related issues arise. Rehabilitation treatment is the best option to address all of the aforementioned issues, as surgical options in adolescents and adults are limited.

Neonatal Brachial Plexus Palsy in Children Aged 0 to 2.5 Years; Parent-Perceived Family Impact, Quality of Life, and Upper Extremity Functioning.

BACKGROUND: To investigate whether parents perceive impact of neonatal brachial plexus palsy on family and quality of life and upper extremity functioning in children less than 2.5 years. METHODS: This cross-sectional study used the PedsQL Family Impact Module (36 items/one total/four scales/scores 0 to 100), TNO-AZL (Dutch Organisation of Applied Natural Science and Academic Hospital Leiden) Preschool Children Quality of Life (43 items/12 scales/scores 0 to 100) and 21 upper extremity functioning questions. Associations between neonatal brachial plexus palsy/patient characteristics and family impact, perceived quality of life, and upper extremity functioning were investigated using regression analysis. RESULTS: Parents of 59 children (median age, 18 months) participated, 49 with C5-C6/C5-C7 lesions. Median Family Impact Module and TNO-AZL Preschool Children Quality of Life scores were 81.3 to 100.0/100.0 and 78.6 to 100.0/100.0. TNO-AZL Preschool Children Quality of Life scores did not differ significantly to healthy references except for stomach, skin, communication, and motor functioning problems. Parents reported around three upper extremity functioning problems. Greater lesion extent, lower age, still being in follow-up, and right-sided lesions were associated with greater family impact (P < 0.01 to P < 0.1). No clinically relevant associations were found for perceived quality of life. Greater lesion extent and nerve surgery history were associated with more upper extremity functioning problems (P < 0.01). Problems were associated with parental worrying (P < 0.05). CONCLUSIONS: Parents perceive having a child with neonatal brachial plexus palsy as impacting on their family depending on the side and severity of the lesion, treatment history, still being in follow-up, and age. They perceive the child's quality of life as relatively normal and not significantly different to healthy peers. However, parents noticed upper extremity functioning problems which increased parental worrying. Health care specialists should take these findings into account to better inform or counsel parents in an early stage during treatment.

Goal attainment scaling in paediatric rehabilitation: a critical review of the literature.

The aim of the study was to review the psychometric properties and use of goal attainment scaling (GAS) in paediatric rehabilitation research. We performed a critical literature review searching: (1) all studies whose main focus was to assess the psychometric properties of GAS in paediatric rehabilitation; and (2) all effect studies in paediatric rehabilitation that used GAS as one of the outcome measures. Three articles in the first group and six in the second group met the inclusion criteria. None of the studies had investigated the content reliability of the scales. Interrater reliability had been investigated in one study and had been found to be good. Only one trial had assessed the content validity of the developed scales, which was found to be acceptable. Comparisons showed that GAS, because of its idiosyncratic nature, measures different constructs from those measured by some related instruments. Low concurrent validity was found. All included studies reported good sensitivity to change. We conclude that the literature supports promising qualities of GAS in paediatric rehabilitation. GAS is a responsive method for individual goal setting and for treatment evaluation. However, current knowledge about its reliability when used with children is insufficient. There is a need for further development of GAS and its application for children of different ages and disabilities, across therapists of different disciplines.

The effect of botulinum toxin type A treatment of the lower extremity on the level of functional abilities in children with cerebral palsy: evaluation with goal attainment scaling.

OBJECTIVE: To measure the effect of botulinum toxin type A (BTX-A) treatment in children with cerebral palsy with regard to individual goals concerning functional abilities, using goal attainment scaling. DESIGN: A single-blind randomized multiple baseline/treatment phase design across subjects. SETTING: The paediatric department of a rehabilitation centre. SUBJECTS: Eleven children with cerebral palsy participated. INTERVENTION: BTX-A treatment of the lower extremity. MAIN MEASURES: A six-point goal attainment scaling of three individual treatment goals at the level of functional abilities. Standardized video-tapes of each goal were recorded weekly for a period of 14 weeks. Rating on the predetermined goal attainment scaling was blinded. RESULTS: Nine of the 11 subjects showed significant improvement in 18 out of 33 goals. Seven subjects showed clinically relevant improvement (at least 2 points on the goal attainment scaling) in 11 goals. Testing the difference between all medians of baseline measurements (after correction for improvement during baseline) and the medians of the treatment phase measurements for all goal attainment scaling scores (n = 33) resulted in significant improvement (p < 0.001). Tested at subject level (medians of the three goal attainment scaling scores per assessment, n = 11), a significant improvement was also found (p = 0.005). The change in goal attainment scaling score was related to the moment of treatment with BTX-A. CONCLUSION: Clinically relevant improvement in individual rehabilitation goals at ability level, achieved with the treatment of BTX-A in children with cerebral palsy, were demonstrated using the goal attainment scaling method.