Pilot study of nasal expiratory positive airway pressure devices for the treatment of childhood obstructive sleep apnea syndrome.

STUDY OBJECTIVES: Alternative therapies for childhood obstructive sleep apnea syndrome (OSAS) are needed as OSAS may persist despite adenotonsillectomy, and continuous positive airway pressure (CPAP) adherence is low. Nasal expiratory positive airway pressure (NEPAP) devices have not been studied in children. We hypothesized that NEPAP would result in polysomnographic improvement. Further, we aimed to determine NEPAP adherence, effects on sleepiness, behavior, and quality of life. METHODS: A randomized, double-blind, placebo-controlled, crossover pilot study was performed. CPAP candidates, 8-16 years old, underwent NEPAP and placebo polysomnograms. Subjects with ≥ 50% reduction in the apnea hypopnea index (AHI) from placebo to NEPAP night or AHI < 5/h on NEPAP night wore NEPAP at home for 30 days. Adherence was assessed by daily phone calls/emails and collecting used devices. RESULTS: Fourteen subjects (age 13.4 ± 1.9 years, BMI z-scores 2.2 ± 1 [mean ± SD]) were studied. There was significant improvement in the obstructive apnea index with NEPAP vs. placebo: 0.6 (0-21.1)/h vs. 4.2 (0-41.9)/h (median [range], p = 0.010) and trends for improvement in other polysomnographic parameters. However, responses were variable, with 3 subjects not improving and 2 worsening. Older children and those with less hypercapnia had a better response. Eight subjects were sent home with devices; one was lost to follow-up, and adherence in the remainder was 83% of nights; these subjects had a significant improvement in sleepiness and quality of life. CONCLUSIONS: NEPAP devices are a potential alternative therapy for OSAS in a small subset of children. Due to variability in individual responses, efficacy of NEPAP should be evaluated with polysomnography. CLINICAL TRIAL REGISTRATION: www.clinicaltrials.gov, identifier: NCT01768065.

Investigating reasons for CPAP adherence in adolescents: a qualitative approach.

STUDY OBJECTIVES: Adolescents with obstructive sleep apnea syndrome (OSAS) represent an important but understudied subgroup of long-term continuous positive airway pressure (CPAP) users. The purpose of this qualitative study was to identify factors related to adherence from the perspective of adolescents and their caregivers. METHODS: Individual open-ended, semi-structured interviews were conducted with adolescents (n = 21) and caregivers (n = 20). Objective adherence data from the adolescents' CPAP machines during the previous month was obtained. Adolescents with different adherence levels and their caregivers were asked their views on CPAP. Using a modified grounded theory approach, we identified themes and developed theories that explained the adolescents' adherence patterns. RESULTS: Adolescent participants (n = 21) were aged 12-18 years, predominantly male (n = 15), African American (n = 16), users of CPAP for at least one month. Caregivers were mainly mothers (n = 17). Seven adolescents had high use (mean use 381 ± 80 min per night), 7 had low use (mean use 30 ± 24 min per night), and 7 had no use during the month prior to being interviewed. Degree of structure in the home, social reactions, mode of communication among family members, and perception of benefits were issues that played a role in CPAP adherence. CONCLUSIONS: Understanding the adolescent and family experience of using CPAP may be key to increasing adolescent CPAP adherence. As a result of our findings, we speculate that health education, peer support groups, and developmentally appropriate individualized support strategies may be important in promoting adherence. Future studies should examine these theories of CPAP adherence.

Determinants of intensity of participation in leisure and recreational activities by youth with cerebral palsy.

OBJECTIVE: To test a model of determinants of intensity of participation in leisure and recreational activities by youth with cerebral palsy (CP). DESIGN: Prospective cohort study. SETTING: Children's hospitals (N=7). PARTICIPANTS: Youth with CP (N=205; age, 13-21y) and their parents. The sample included 107 (57.2%) males and 26 (12.7%) to 57 (27.8%) youth in each of the 5 levels of the Gross Motor Function Classification System (GMFCS). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Youth completed the Children's Assessment of Participation and Enjoyment by means of an interview. Parents completed the Pediatric Outcomes Data Collection Instrument, Family Environment Scale, Coping Inventory, Measure of Processes of Care, a demographic questionnaire, and a services questionnaire. RESULTS: Structural equation modeling was used to test the model. Fit statistics indicate good model fit. The model explains 35% of the variance in intensity of participation. Path coefficients (P ≤ .05) indicate that higher physical ability, higher enjoyment, younger age, female sex, and higher family activity orientation are associated with higher intensity of participation. GMFCS level and caregiver education have indirect effects on intensity of participation. The path between services and intensity of participation was not significant. CONCLUSIONS: Participation by youth with CP is influenced by multiple factors. The influence of physical activity supports the importance of activity accommodations and assistive technology for youth who are not capable of improving physical ability. Knowledge of family activity orientation is important for identifying opportunities for participation. The unexplained variance suggests that the model should include other determinants, such as physical accessibility and availability of transportation and community leisure and recreational activities.

Comparing the priorities of parents and young people with cerebral palsy.

PURPOSE: Adolescence is a period of change and transition that may pose unique challenges for young people with cerebral palsy (CP). We compared statements of priorities, i.e. what adolescents (13-17) and youth (18-21) and their parents would like to be able to do to enable greater activity and participation. METHODS: Participants were 198 parents and 135 young people with CP (45% males) from seven Children's Hospitals in the United States. The interviews were structured using The Canadian Occupational Performance Measure. Priority statements were categorised as Adult Tasks, Biology, Self-Identity or Physical Activity. RESULTS: All parents identified more priorities for Biology and Adult Tasks (p  <  0.001). Adolescents identified the fewest priorities for Self-Identity (p  <  0.01). Youth identified, in descending order of frequency, priorities in Adult Tasks, Biology, Self-Identity and Physical Activity (p  <  0.05). In the parent-young people dyads when disagreements occurred, Self-Identity issues were identified more often by parents (p  <  0.05) and priorities for Physical Activity were identified more often by young people (p  <  0.01). CONCLUSIONS: The shared and differing priorities of parents and young people with CP may reflect different roles, perceptions and experiences. The findings have implications for healthcare professionals, providing a framework to compare and contrast the priorities of young people and their parents.

Determinants of intensity of participation in leisure and recreational activities by children with cerebral palsy.

AIM: To test a model of child, family, and service determinants of intensity of participation in leisure and recreational activities by children with cerebral palsy (CP). METHOD: Participants were 288 children with CP, age range 6 to 12 years (mean 9y 8mo, SD 2y), and their parents from seven children's hospitals. The sample comprised 166 (57.6%) males and 122 (42.4%) females, and between 40 (13.9%) and 74 (25.7%) children in each of the five levels of the Gross Motor Function Classification System. Children completed the Children's Assessment of Participation and Enjoyment by interview. Parents completed the Pediatric Outcomes Data Collection Instrument, Family Environment Scale, Coping Inventory, Measure of Processes of Care, and two questionnaires. Structural equation modeling was used to test the model. RESULTS: Fit statistics indicated a good model fit. The model explains 32% of the variance in intensity of participation. Path coefficients (p ≤ 0.05) indicate that higher gross motor function, higher enjoyment, more effective adaptive behavior, younger age, and higher family activity orientation are associated with higher intensity of participation. The path between services and participation was not significant. INTERPRETATION: Intensity of participation of children with CP is influenced by multiple child and family determinants. Children's gross motor function and behavior in life situations are important for participation; knowledge of activities the child and family enjoy has implications for opportunities for participation. Professionals are encouraged to address priorities for leisure and recreation identified by children with CP and their families.

Family priorities for activity and participation of children and youth with cerebral palsy.

BACKGROUND: Understanding family priorities for children and youth with cerebral palsy is essential for family-centered service. OBJECTIVE: The purposes of this study were: (1) to identify family priorities for activity and participation in children and youth with cerebral palsy and (2) to determine differences based on age and Gross Motor Functional Classification System (GMFCS) level. DESIGN: Five hundred eighty-five children and youth with cerebral palsy and their caregivers participated at regional children's hospitals. The children and youth were 2 to 21 years of age; 56% were male, and 44% were female. Their caregivers, predominantly mothers (80%), had a mean age of 40.3 years (SD=9.3). The Canadian Occupational Performance Measure was administered to caregivers to identify their priorities for their children. The priorities were coded into 3 categories (daily activities, productivity, and leisure) and 13 subcategories. The GMFCS levels were determined by assessors who met the criterion for reliability. Friedman and Kruskal-Wallis one-way analyses of variance were used to examine differences in priorities. RESULTS: Parents of children in all age groups and GMFCS levels II to V identified more priorities for daily activities. Parents of school-aged children and youth had more priorities for productivity than parents of younger children. For parents of children in all age groups and motor function levels, self-care was the most frequent priority subcategory. Sixty-one percent of parents identified at least one priority related to mobility. LIMITATIONS: The study did not include qualitative analysis of priorities of parents. CONCLUSIONS: Parents' priorities for their children and youth with cerebral palsy differed depending on age and gross motor function level; however, the most frequent priority for all age groups was daily activities. Interviews with families are recommended for identifying outcomes for activity and participation and developing an intervention plan.

Participation in home, extracurricular, and community activities among children and young people with cerebral palsy.

AIM: Participation in home, extracurricular, and community activities is a desired outcome of rehabilitation services for children and young people with cerebral palsy (CP). The purpose of this study was to investigate the effect of age and gross motor function on participation among children and young people with CP. METHOD: Five hundred participants (277 males, 223 females) were grouped by age and Gross Motor Function Classification System (GMFCS) level. There were 291 children aged 6 to 12 years and 209 young people aged 13 to 21 years. There were 128 participants in GMFCS level I, 220 in levels II/III, and 152 in levels IV/V. Participants completed the Children's Assessment of Participation and Enjoyment to measure number of activities (diversity) and how often they were performed (intensity) in the past 4 months. RESULTS: Children had higher overall participation diversity and intensity than young people (p<0.001). Children and young people in GMFCS level I had the highest overall participation, followed by children and young people in levels II/III and IV/V. Children had higher participation in recreational (p<0.001) but not formal (such as team sports or clubs) or physical activities. Children (p<0.01) and young people (p<0.001) in level I had the highest participation in physical activities; diversity and intensity were generally low. INTERPRETATION: The findings provide evidence of the effect of age and gross motor function on participation of children and young people with CP. Low participation in physical activities may have implications for fitness and health, especially for children and young people in GMFCS levels IV and V.

Social and community participation of children and youth with cerebral palsy is associated with age and gross motor function classification.

BACKGROUND: Through social and community participation, children and youth with cerebral palsy (CP) form friendships, gain knowledge, learn skills, express creativity, and determine meaning and purpose in life. OBJECTIVE: The purposes of this study were: (1) to determine whether social and community participation of children and youth with CP differ based on age, sex, and gross motor function, and (2) to identify the types of activities in which social and community participation are highest. DESIGN AND METHODS: A prospective cross-sectional analytic design was used. The participants were a sample of convenience of 291 children (6-12 years of age) and 209 youth (13-21 years of age) with CP (55.4% males, 44.6% females) receiving services from 7 children's hospitals. Participants completed the Children's Assessment of Participation and Enjoyment (CAPE) by structured interview. Gross Motor Function Classification System (GMFCS) level was determined by the researchers. RESULTS: Youth did a higher percentage of activities with friends and others and outside the home than children. Children and youth in level I did a higher percentage of activities with friends and others compared with children and youth in levels II and III and in levels IV and V. Children and youth in level I and in levels IV and V did a higher percentage of activities outside the home than children and youth in levels II and III. Differences were not found between females and males. The percentage of activities done with friends and others and outside the home was highest for physical and skill-based activities. LIMITATIONS: Findings cannot be attributed only to GMFCS level. CONCLUSIONS: The ability to walk without restrictions is desirable for social and community participation. For children and youth with CP who have limitations in mobility, physical therapists have roles as consultants for accessibility, activity accommodations, and assistive technology and as advocates for inclusive environments.