Associations of Coexisting Conditions with Healthcare Spending for Children with Cerebral Palsy.

OBJECTIVE: To determine which coexisting conditions have the strongest associations with healthcare use and spending among children with cerebral palsy (CP). STUDY DESIGN: Retrospective analysis of 16 695 children ages 0-18 years with CP - identified with International Classification of Diseases, Ninth Revision, Clinical Modification codes - using Medicaid from January 1, 2013 to December 31, 2013 from 10 states in the Truven MarketScan Medicaid Database. Using generalized linear models, we assessed which coexisting conditions (including medical technology) identified with Agency for Healthcare Research and Quality's Chronic Condition Indicators had the strongest associations with total healthcare spending across the healthcare continuum. RESULTS: Median per-patient annual Medicaid spending for children with CP was $12 299 (IQR $4826-$35 582). Most spending went to specialty (33.1%) and hospital (26.7%) care. The children had a median 6 (IQR 4-10) coexisting conditions; epilepsy was the most common (38.1%). Children with epilepsy accounted for 59.6% ($364 million) of all CP spending. In multivariable analysis, the coexisting conditions most strongly associated with increased spending were tracheostomy (median additional cost per patient = $56 567 [95%CI $51 386-61 748]) and enterostomy (median additional cost per patient = $25 707 [95%CI $23 753-27 660]). CONCLUSIONS: Highly prevalent in children with CP using Medicaid, coexisting conditions correlate strongly with healthcare spending. Tracheostomy and enterostomy, which indicate significant functional impairments in breathing and digestion, are associated with the highest spending. Families, providers, payers, and legislators may leverage these findings when designing policies positioned to enable the best health and care for children with cerebral palsy.

Population pharmacokinetics of oral baclofen in pediatric patients with cerebral palsy.

OBJECTIVE: To characterize the population pharmacokinetics (PK) of oral baclofen and assess impact of patient-specific covariates in children with cerebral palsy (CP) in order to support its clinical use. SUBJECTS DESIGN: Children (2-17 years of age) with CP received a dose of titrated oral baclofen from 2.5 mg 3 times a day to a maximum tolerated dose of up to 20 mg 4 times a day. PK sampling followed titration of 10-12 weeks. Serial R- and S-baclofen plasma concentrations were measured for up to 16 hours in 49 subjects. Population PK modeling was performed using NONMEM 7.1 (ICON PLC; Ellicott City, Maryland). RESULTS: R- and S-baclofen showed identical concentration-time profiles. Both baclofen enantiomers exhibited linear and dose/kg-proportional PK, and no sex differences were observed. Average baclofen terminal half-life was 4.5 hours. A 2-compartment PK model with linear elimination and transit absorption steps adequately described concentration-time profiles of both baclofen enantiomers. The mean population estimate of apparent clearance/F was 0.273 L/h/kg with 33.4% inter-individual variability (IIV), and the apparent volume of distribution (Vss/F) was 1.16 L/kg with 43.9% IIV. Delayed absorption was expressed by a mean transit time of 0.389 hours with 83.7% IIV. Body weight, a possible genetic factor, and age were determinants of apparent clearance in these children. CONCLUSION: The PK of oral baclofen exhibited dose-proportionality and were adequately described by a 2-compartment model. Our population PK findings suggest that baclofen dosage can be based on body weight (2 mg/kg per day) and the current baclofen dose escalation strategy is appropriate in the treatment of children with CP older than 2 years of age.

Growth and nutritional status in residential center versus home-living children and adolescents with quadriplegic cerebral palsy.

OBJECTIVE: To describe growth and nutrition in nonambulatory youth (<19 years of age) with cerebral palsy (CP) living in residential centers compared with similar youth living at home. STUDY DESIGN: A multicenter, cross-sectional, single observational assessment of 75 subjects living in a residential care facility compared with 205 subjects living at home. Primary outcome measures included anthropometric measures of height, weight, triceps, and subscapular skinfolds, and mid-upper-arm muscle area. Z scores were calculated from reference values for healthy children. Age, use of a feeding tube, and Gross Motor Functional Classification System (GMFCS) level were included as important confounders. RESULTS: Use of a feeding tube was associated with higher skinfold Z scores, and a significantly higher percentage of the residential subjects had a feeding tube. Height, weight, and arm-muscle area Z scores all diverged (negatively) from reference values with age, and the residential subjects were on average older than the home-living subjects. After controlling for age, GMFCS level and use of a feeding tube, residential living was associated with significantly greater weight, height, skinfold thicknesses, and mid-arm muscle area Z scores. CONCLUSION: Poor growth and nutrition in children with CP is a prevalent, important, and complex problem. Although factors intrinsic to the condition of CP likely play a significant role, it is also clear that environmental factors, including the living situation of the child, can have an impact.

Longitudinal changes in bone density in children and adolescents with moderate to severe cerebral palsy.

OBJECTIVE: To assess the natural history of "growth" in bone mineral density (BMD) in children and adolescents with moderate to severe cerebral palsy (CP). STUDY DESIGN: A prospective, longitudinal, observational study of BMD in 69 subjects with moderate to severe spastic CP ages 2.0 to 17.7 years. Fifty-five subjects were observed for more than 2 years and 40 subjects for more than 3 years. Each evaluation also included assessments of growth, nutritional status, Tanner stage, general health, and various clinical features of CP. RESULTS: Lower BMD z-scores at the initial evaluation were associated with greater severity of CP as judged by gross motor function and feeding difficulty, and with poorer growth and nutrition as judged by weight z-scores. BMD increased an average of 2% to 5%/y in the distal femur and lumbar spine, but ranged widely from +42%/y to -31%. In spite of increases in BMD, distal femur BMD z-scores decrease with age in this population. CONCLUSIONS: Children with severe CP develop over the course of their lives clinically significant osteopenia. Unlike elderly adults, this is not primarily from true losses in bone mineral, but from a rate of growth in bone mineral that is diminished relative to healthy children. The efficacy of interventions to increase BMD can truly be assessed only with a clear understanding of the expected changes in BMD without intervention.

Relationship of nutritional status to health and societal participation in children with cerebral palsy.

OBJECTIVES: To describe nutritional status in a population-based sample of children with moderate or severe cerebral palsy (CP) and to explore the relationships between nutritional status and health and functional outcomes. STUDY DESIGN: A population-based strategy was used to enroll children with CP at 6 geographic sites. Research assistants performed anthropometric assessment, determined severity of motor impairment, and interviewed caregivers with the Child Health Questionnaire and a questionnaire designed specifically for this study. Anthropometric measures were converted to Z scores and the relationship between health and nutritional status was assessed using regression models. RESULTS: Among the 235 participants, indicators of malnutrition were common. Poor nutritional status correlated with increased health care utilization (hospitalizations, doctor visits) and decreased participation in usual activities by the child and parent. CONCLUSIONS: Malnutrition is common in children with moderate or severe CP and associated with poorer health status and limitations in societal participation. Further studies are needed to determine the nature of these associations and how to manage nutrition in children with CP to optimize growth and health outcomes.