5-year fracture risk among children with cerebral palsy.

BACKGROUND: Epidemiologic evidence documenting fracture risk as children with cerebral palsy (CP) age throughout growth is lacking to inform on when to implement fracture prevention strategies. The objective was to characterize the 5-year risk of fractures by each year of age among <1-13 year olds with CP and effects by patient-level factors. METHODS: This retrospective cohort study used commercial administrative claims from 01/01/2001 to 12/31/2018 from children <1-13 years old with ≥5 years of insurance enrollment. Fractures were examined during the 5-year follow-up. For the CP cohort, the association between 5-year fracture rate and patient-level factors was assessed using Cox regression. RESULTS: Children with (n = 5559) vs. without (n = 2.3 million) CP had a higher 5-year fracture risk at the vertebral column, hip, and lower extremities at almost each year of age, but lower 5-year fracture risk at the upper extremities after 6 years old (all P < 0.05). Among children with CP, the 5-year fracture rate was elevated for co-occurring neurological conditions and non-ambulatory status at the vertebral column, hip, and lower extremities (hazard ratio [HR] range, 1.44-2.39), and higher for males at the upper extremities (HR = 1.29) (all P < 0.05). CONCLUSIONS: This study provides novel epidemiologic evidence of 5-year fracture risk for each year of age for children with CP. IMPACT: This study provides novel epidemiologic evidence of 5-year fracture risk for each year of age across important developmental stages for children with vs. without cerebral palsy (CP). Children with vs. without CP were more likely to fracture at the vertebral column, hip, lower extremities, and humerus and less likely to fracture at the forearm and hands. The age-related 5-year fracture risk was associated with clinically relevant patient-level factors, but in different ways by fracture region. Study findings may be used to enhance clinical detection of at-risk children and strategize when to implement fracture prevention efforts for children with CP.

Post-fracture rehabilitation pathways and association with mortality among adults with cerebral palsy.

OBJECTIVE: Rehabilitation may mitigate the high mortality rates and health declines post-fracture for adults with cerebral palsy, but this is understudied. The objectives were to characterize the post-fracture rehabilitation pathways and identify their association with 1-year survival among adults with cerebral palsy. METHODS: A retrospective cohort study of adults with cerebral palsy with a fragility fracture with continuous health plan enrollment ≥1-year prior to and ≥1 day after their fracture date was performed using a random 20% Medicare fee-for-service dataset. Participants were categorized as a home discharge or inpatient rehabilitation admission post-fracture. For the home discharge cohort, weekly exposure to outpatient physical/occupational therapy (PT/OT) was examined up to 6-month post-fracture. Cox regression examined the association between time-varying PT/OTuse within 6-month post-fracture and mortality from 30 days to 1-year post-fracture before and after adjusting for confounders (e.g. medical complexity). RESULTS: Of 3598 adults with cerebral palsy with an incident fragility fracture, 74% were discharged home without inpatient rehabilitation; they were younger, but more medically complex compared to the 26% admitted to inpatient rehabilitation. Among the home discharge cohort (n = 2662), 43.1% initiated PT/OTwithin 6-month post-fracture, and cumulative PT/OTexposure post-fracture was associated with improved survival; for example, per 3 weeks of PT/OTexposure, the adjusted mortality rate was 40% lower (95% confidence interval (CI) = 0.41-0.89). CONCLUSIONS: Most adults with cerebral palsy with a fragility fracture were discharged home rather than to inpatient rehabilitation, and only 43.1% of that group initiated outpatient PT/OTwithin 6 months post-fracture. Receiving outpatient PT/OTwas associated with improved 1-year survival.

Clinical bone health among adults with cerebral palsy: moving beyond assessing bone mineral density alone.

AIM: To understand associations among bone mineral density (BMD), bone mineral content (BMC), and bone area, and their association with fractures in adults with cerebral palsy (CP). METHOD: This retrospective cohort study included 78 adults with CP with a hip dual energy X-ray absorptiometry (DXA) from 1st December 2012 to 3rd May 2021 performed at the University of Michigan. Data-driven logistic regression techniques identified which, if any, DXA-derived bone traits (e.g. age/sex/ethnicity-based z-scores) were associated with fracture risk by sex and severity of CP. BMC-area associations were examined to study the structural mechanisms of fragility. RESULTS: Femoral neck area was associated with lower age-adjusted odds ratios (ORs) of fracture history (OR 0.72; 95% confidence interval [CI] 0.49-1.06; p=0.098), while higher BMD was associated with higher odds of incident fracture (OR 3.08; 95% CI 1.14-8.33; p=0.027). Females with fracture had lower area than females without fracture but similar BMC, whereas males with fracture had larger area and higher BMC than males without fracture. The paradoxical BMD-fracture association may be due to artificially elevated BMD from BMC-area associations that differed between females and males (sex interaction, p˂0.05): males had higher BMC at lower area values and lower BMC at higher area values compared to females. INTERPRETATION: BMD alone may not be adequate to evaluate bone strength for adults with CP. Further research into associations (or integration) between BMC and area is needed.

Chemotherapy-induced peripheral neuropathy onset is associated with early risk of depression and anxiety in breast cancer survivors.

OBJECTIVE: The objective was to assess for an association between chemotherapy-induced peripheral neuropathy (CIPN) onset and development of depression and anxiety in breast cancer (BrCa) survivors. METHODS: A retrospective observational cohort was used and identified from Optum's De-identified Clinformatics® Data Mart Database years 2012-2015. Three groups of women were derived based on BrCa and CIPN status: BrCa+/CIPN+ (n = 244), BrCa+/CIPN- (n = 8870), and BrCa-/CIPN- (n = 1,125,711). The ratio of the prevalence ratios (RPR) determined if the change in risk of depression and anxiety from the 12-month preindex period to postindex period I (0-6 months) and II (7-12 months) was different for BrCa+/CIPN+ compared to BrCa+/CIPN- and BrCa-/CIPN-. RESULTS: The adjusted RPR for depression was significantly elevated for BrCa+/CIPN+ compared to BrCa+/CIPN- and BrCa-/CIPN- for postindex periods I (RPR = 1.35 [1.10,1.65] and 1.33 [1.08,1.63], respectively) and II (RPR = 1.53 [1.21,1.94] and 1.50 [1.17,1.93], respectively). The RPR for anxiety was significantly elevated for BrCa+/CIPN+ compared to BrCa+/CIPN- and BrCa-/CIPN- for postindex periods I (RPR = 1.37 [1.12,1.67] and 1.31 [1.06,1.61], respectively) and II (RPR = 1.41 [1.13,1.76] and 1.28 [1.02,1.62], respectively). CONCLUSIONS: Among BrCa survivors, CIPN onset is associated with a subsequent increased 12-month risk of depression and anxiety. Depression and anxiety screening should be considered in BrCa+/CIPN+ survivors, particularly given their known impact on fall risk. The observed association between CIPN and an increased risk of depression and anxiety should be further studied in prospective studies.

Effect of levetiracetam and oxcarbazepine on 4-year fragility fracture risk among prepubertal and pubertal children with epilepsy.

OBJECTIVE: The objective of this study was to determine whether two commonly prescribed antiseizure medications (ASMs), levetiracetam (LEV) and oxcarbazepine (OXC), were associated with an increased risk of fragility fracture in children with epilepsy when initiating therapy during a crucial period of bone development, namely, pre- and midpuberty. METHODS: Claims data from January 1, 2009 to December 31, 2018 were extracted from the Optum Clinformatics Data Mart. Children aged 4-13 years at baseline with at least 5 years of continuous health plan enrollment were included to allow for a 1-year baseline (e.g., pre-ASM exposure) and 4 years of follow-up. Children with epilepsy who were ASM naïve were grouped based on whether ASM treatment initiation included LEV or OXC. The comparison group included children without epilepsy and without ASM exposure. Crude incidence rate (IR; n per 1000 person-years) and IR ratio (IRR; with 95% confidence interval [CI]) were estimated for nontrauma fracture (NTFx), a claims-based proxy for fragility fracture, for up to 4 years of follow-up. Cox proportional hazards regression estimated the hazard ratio (HR; with 95% CI) after adjusting for demographic variables, motor impairment, and baseline fracture. RESULTS: The crude IR (95% CI) of NTFx was 21.5 (21.2-21.8) for non-ASM-users without epilepsy (n = 271 346), 19.8 (12.3-27.2) for LEV (n = 358), and 34.4 (21.1-47.7) for OXC (n = 203). Compared to non-ASM-users, the crude IRR of NTFx was similar for LEV (IRR = .92, 95% CI = .63-1.34) and elevated for OXC (IRR = 1.60, 95% CI = 1.09-2.35); the crude IRR of NTFx was elevated for OXC compared to LEV (IRR = 1.74, 95% CI = 1.02-2.99). The findings were consistent after adjusting for covariates, except when comparing OXC to LEV (HR = 1.71, 95% CI = .99-2.93), which was marginally statistically insignificant (p = .053). SIGNIFICANCE: Initiating OXC, but not LEV, therapy among 4-13-year-olds with epilepsy is associated with an elevated risk of fragility fracture. Studies are needed to determine whether these children could benefit from adjunct bone fragility therapies.

Whitney Comorbidity Index to monitor health status for adults with cerebral palsy: validation and thresholds to assist clinical decision making.

AIM: To validate the Whitney Comorbidity Index (WCI), which was recently developed to monitor disease status for adults with cerebral palsy (CP), and to identify WCI scores associated with an increased mortality risk using a representative sample of adults with CP. METHOD: Data from 2016 to 2018 were used from a random 20% sample from the fee-for-service Medicare database for this retrospective cohort study. The WCI was examined as unweighted (WCIunw ) and weighted (WCIw ) among adults at least 18 years old with CP. Cox regression models were developed with mortality as the outcome after adjusting for demographics. A concordance statistic (C-statistic) of at least 0.70 was considered as showing sufficient validity. The hazard ratio of mortality for each WCI score was estimated. Secondary analyses were performed for subgroups with co-occurring epilepsy and/or intellectual disabilities. RESULTS: For the entire group (n=16 728) and subgroups, the WCI showed sufficient validity (C-statistic 0.73-0.81). For the entire group, the mortality rate was elevated for a score of 1 compared with 0 from the WCIunw (hazard ratio 3.06; 95% confidence interval [CI] 1.52-6.17) and WCIw (hazard ratio 4.08; 95% CI 1.69-9.85), and became larger with each WCI score. Results were similar for the subgroups. INTERPRETATION: The WCI is a valid marker for health/disease status for adults with CP. Several WCI score thresholds were identified to assist in clinical decision making for preventive medicine and intervention implementation. What this paper adds The Whitney Comorbidity Index (WCI) is valid among 16 728 adults with CP. The WCI is valid for those with co-occurring epilepsy and/or intellectual disabilities. Thresholds of the WCI score were identified to assist clinical decision making.

Development of a new comorbidity index for adults with cerebral palsy and comparative assessment with common comorbidity indices.

AIM: To develop a new comorbidity index for adults with cerebral palsy (CP), the Whitney Comorbidity Index (WCI), which includes relevant comorbidities for this population and better predicts mortality than the Charlson Comorbidity Index (CCI) and Elixhauser Comorbidity Index (ECI). METHOD: Data from the Optum Clinformatics Data Mart was used for this retrospective cohort study. Diagnosis codes were used to identify adults aged 18 years or older with CP (n=1511 females, n=1511 males; mean [SD; range] age=48y [19y 2mo; 18-89y]) and all comorbidities in the year 2014. The WCI was developed based on the comorbidities of the CCI and ECI and other relevant comorbidities associated with 2-year mortality using Cox regression and competing risk analysis. The WCI was examined as unweighted (WCIunw ) and weighted (WCIw ). The model fit and discrimination (C-statistic) of each index was assessed using Cox regression. RESULTS: Twenty-seven comorbidities were included in the WCI; seven new comorbidities that were not part of the CCI or ECI were added. The WCIunw and WCIw showed a better model fit and discrimination for 1- and 2-year mortality compared to the CCI and ECI. The WCIunw and WCIw were strong predictors for 1- and 2-year mortality (C-statistic [95% confidence interval] ranging from 0.81 [0.76-0.85] to 0.88 [0.82-0.94]). INTERPRETATION: The new WCI, designed to include clinically relevant comorbidities, provides a better model fit and discrimination of mortality for adults with CP. WHAT THIS PAPER ADDS: Common comorbidity indices exclude relevant comorbidities for adults with cerebral palsy (CP). A new comorbidity index for adults with CP was created by harmonizing clinical theory and data-driven methods. The Whitney Comorbidity Index better predicted 1- and 2-year mortality than other commonly used comorbidity indices.

Effectiveness of osteoporosis medication on site-specific fracture-risk attenuation among adults with epilepsy.

OBJECTIVE: The objective of this propensity score-matched, observational cohort study was to determine the effectiveness of osteoporosis medication on reducing the risk of non-trauma fracture (NTFx) among adults with epilepsy. METHODS: Data from 01/01/2012 to 09/30/2015 was extracted from Optum Clinformatics Data Mart. NTFx risk attenuation from 12 months prior to 12 months after the individual's index date was examined for each group of adults ≥50 years of age as risk ratios (RRs with 95% confidence intervals [CIs]). Groups were stratified based on: (1) epilepsy status, as with vs without epilepsy (EP); and (2) if and when osteoporosis medication was first prescribed, as new users (treatment naive), consistent users (osteoporosis medication prescribed in pre-index period), and no users. Comparison groups were matched 1:1 to EP new users (n = 828/group) for demographics, glucocorticoid and antiseizure medication, and the Elixhauser comorbidity index. Difference-in-difference analysis compared the change in pre- to post-index NTFx risk among groups as the ratio of the RR (RRR). RESULTS: The pre- to post-index NTFx risk at any site was reduced for EP new users (RR = 0.49; 95% CI = 0.40-0.61) and EP consistent users (RR = 0.70; 95% CI = 0.38-0.98), but nonsignificantly elevated for EP no users (RR = 1.39; 95% CI = 0.93-2.07)-findings were consistent for most sites (eg, vertebral column). EP new users had a larger NTFx risk attenuation at any site compared to EP no users (RRR = 0.35; 95% CI = 0.23-0.54) and EP consistent users (RRR = 0.70; 95% CI = 0.51-0.97). EP consistent users had a larger NTFx risk attenuation at any site compared to EP no users (RRR = 0.50; 95% CI = 0.32-0.79). The extent of NTFx risk attenuation at any site was similar for new users with vs without epilepsy (RRR = 0.99; 95% CI = 0.73-1.34) and consistent users with vs without epilepsy (RRR = 0.81; 95% CI = 0.55-1.17). There was evidence of site-specific effects (eg, hip). CONCLUSION: Osteoporosis medication is associated with a clinically meaningful 12-month NTFx risk attenuation for adults with epilepsy, especially for those just starting osteoporosis medication.

The mortality burden attributable to nontrauma fracture for privately insured adults with epilepsy.

OBJECTIVE: Individuals with epilepsy have poor bone development and preservation throughout the lifespan and are vulnerable to nontrauma fracture (NTFx) and post-NTFx complications. However, no studies have examined the contribution of NTFx to mortality among adults with epilepsy. The objective was to determine whether NTFx is a risk factor for mortality among adults with epilepsy. METHODS: Data from 2011 to 2016 were obtained from Optum Clinformatics Data Mart, a nationwide claims database from a single private payer in the United States. Diagnosis codes were used to identify adults (≥18 years old) with epilepsy, NTFx, and covariates (demographics and pre-NTFx cardiovascular disease, respiratory disease, diabetes, chronic kidney disease, cancer). Crude mortality rate per 100 person-years was estimated. Cox regression estimated hazard ratios (HRs) and 95% confidence intervals (CIs) were determined for mortality, comparing epilepsy and NTFx (EP + NTFx; n = 11 471), epilepsy without NTFx (EP without NTFx; n = 50 384), without epilepsy and with NTFx (without EP + NTFx; n = 423 041), and without epilepsy and without NTFx (without EP without NTFx; n = 6.8 million) after adjusting for covariates. RESULTS: The 3-, 6-, and 12-month crude mortality rates were highest among EP + NTFx (12-month mortality rate = 8.79), followed by without EP + NTFx (12-month mortality rate = 4.80), EP without NTFx (12-month mortality rate = 3.06), and without EP without NTFx (12-month mortality rate = 0.47). After adjustments, the mortality rate was elevated for EP + NTFx for all time points compared to EP without NTFx (eg, 12-month HR = 1.70, 95% CI = 1.58-1.85), without EP + NTFx (eg, 12-month HR = 1.41, 95% CI = 1.32-1.51), and without EP without NTFx (eg, 12-month HR = 5.23, 95% CI = 4.88-5.60). Stratified analyses showed higher adjusted HRs of 12-month mortality for EP + NTFx for all NTFx sites (ie, vertebral column, hip, extremities), all age categories (young, middle-aged, older), and for both women and men. SIGNIFICANCE: Among adults with epilepsy and compared to adults without epilepsy, NTFx is associated with a higher 12-month mortality rate. Findings suggest that NTFx may be a robust risk factor for mortality among adults with epilepsy.

Effect of pain on mood affective disorders in adults with cerebral palsy.

AIM: To determine if pain is associated with 12-month incidence of mood affective disorders (MAD) in adults with cerebral palsy (CP). METHOD: Data from Optum Clinformatics® Data Mart (2013-2016) were used for this retrospective cohort study. Diagnostic codes were used to identify adults (≥18y) with CP, incident cases of MAD, and covariates (other neurodevelopmental conditions, sleep disorders, arthritis). Pain (any type, location) was identified between 1st October 2014 and 30th September 2015. The pain group was divided into new or consistent pain if they had a history of pain (i.e. consistent) in the 12 months before their first pain claim date. Crude incidence rates of MAD (expressed per 100 person-years) were estimated. Cox regression was used to estimate hazard ratio (95% confidence interval [CI]) of MAD after adjusting for covariates. RESULTS: Adults that had new pain (n=859; incidence rate=15.5) or consistent pain (n=1303; incidence rate=17.9) had greater crude incidence rate of MAD compared to adults without pain (n=3726; incidence rate=5.9). The elevated rate of MAD remained after adjusting for covariates, for new pain (hazard ratio=2.4; 95% CI=1.9-3.0) and consistent pain (hazard ratio=2.1; 95% CI=1.7-2.7). INTERPRETATION: Pain is associated with greater incidence of MAD in adults with CP. This association remained after accounting for potential confounding factors. WHAT THIS PAPER ADDS: What this paper adds Pain was associated with higher 12-month incidence of mood affective disorders (MAD). The 12-month MAD incidence was similar between new and consistent pain groups. The MAD incidence remained higher adjusting for neurodevelopmental comorbidities, sleep disorders, and arthritis.

Greater Visceral Fat but No Difference in Measures of Total Body Fat in Ambulatory Children With Spastic Cerebral Palsy Compared to Typically Developing Children.

BACKGROUND: Individuals with cerebral palsy (CP) are at increased risk for obesity and obesity-related complications. Studies of total body fat in those with CP are inconsistent and studies of abdominal fat are lacking in children with CP. The objective of this study was to determine if ambulatory children with spastic CP have greater central adiposity compared to typically developing children. METHODOLOGY: Eighteen ambulatory children with spastic CP (n = 5 girls; 8.6 ± 2.9 yr) and 18 age-, sex-, and race-matched typically developing children (controls; 8.9 ± 2.1 yr) participated in this cross-sectional study. Children with CP were classified as I or II using the Gross Motor Function Classification System. Dual-energy X-ray absorptiometry assessed body composition, including total body, trunk and abdominal fat mass, fat-free mass, fat mass index (FMI), and fat-free mass index (FFMI). RESULTS: There were no group differences in fat mass, fat-free mass, FMI, and FFMI in the total body, fat mass, fat-free mass, and FFMI in the trunk, or fat mass, visceral fat mass, and subcutaneous fat mass in the abdomen (p > 0.05). Compared to controls, children with CP had higher trunk FMI, abdominal FMI, and visceral FMI (p < 0.05). Although marginally insignificant (p = 0.088), children with CP had higher subcutaneous FMI. CONCLUSIONS: Ambulatory children with spastic CP have elevated central adiposity, especially in the visceral region, despite no differences in measures of total body fat. How this relates to cardiometabolic disease progression in those with CP requires further investigation.

Prevalence of Mental Health Disorders Among Adults With Cerebral Palsy: A Cross-sectional Analysis.

Background: Persons with cerebral palsy (CP) have an increased risk for secondary chronic conditions during childhood, including mental health disorders. However, little is known about how these disorders affect adults with CP. Objective: To determine the prevalence of mental health disorders among adults with CP compared with those without CP. Design: Cross-sectional. Setting: 2016 Optum Clinformatics Data Mart. Patients: 8.7 million adults (including 7348 adults with CP). Measurements: Other neurodevelopmental comorbid conditions (intellectual disabilities, autism spectrum disorders, epilepsy) and 37 mental health disorders (as 6 categories) were identified on the basis of diagnosis codes. Direct age-standardized prevalence of the mental health disorder categories was estimated by sex for adults with CP alone, adults with CP and neurodevelopmental disorders, and adults without CP. Results: Men with CP alone had higher age-standardized prevalence than men without CP for schizophrenic disorders (2.8% [95% CI, 2.2% to 3.4%] vs. 0.7%), mood affective disorders (19.5% [CI, 18.0% to 21.0%] vs. 8.1%), anxiety disorders (19.5% [CI, 18.0% to 21.0%] vs. 11.1%), disorders of adult personality and behavior (1.2% [CI, 0.8% to 1.6%] vs. 0.3%), and alcohol- and opioid-related disorders (4.7% [CI, 3.9% to 5.5%] vs. 3.0%). The same pattern was observed for women. Compared with adults with CP alone, those with CP and neurodevelopmental disorders had similar or higher age-standardized prevalence of the 6 mental health disorder categories, except for the lower prevalence of alcohol- and opioid-related disorders in men. Limitations: Single claims code was used to define the cohort of interest. Information on the severity of CP was not available. Conclusion: Compared with adults without CP, those with CP have an elevated prevalence of mental health disorders, some of which may be more pronounced in patients with comorbid neurodevelopmental disorders. Primary Funding Source: National Institute on Disability, Independent Living, and Rehabilitation Research.

BMI does not capture the high fat mass index and low fat-free mass index in children with cerebral palsy and proposed statistical models that improve this accuracy.

BACKGROUND/OBJECTIVES: Children with cerebral palsy (CP) are at risk for having a misclassified overweight/obesity status based on BMI thresholds due to their lower fat-free mass and similar fat mass compared with typically developing children. The primary objective was to determine if BMI could predict fat mass index (FMI) and fat-free mass index (FFMI) in children with CP. SUBJECTS/METHODS: Forty-two children with CP and 42 typically developing children matched to children with CP for age and sex participated in the study. Dual-energy X-ray absorptiometry was used to assess body composition. Children with CP who could ambulate without assistance were considered ambulatory (ACP) and the rest were considered nonambulatory (NACP). RESULTS: Children with CP had higher percent body fat (%Fat) and FMI and lower fat-free mass and FFMI than controls (p < 0.05) but no difference in fat mass (p = 0.10). When BMI was statistically controlled, NACP had higher %Fat, fat mass and FMI and lower FFMI than ACP and controls (p < 0.05). NACP also had lower fat-free mass than controls (p < 0.05). ACP had higher %Fat and FMI and lower fat-free mass and FFMI than controls (p < 0.05). BMI was a strong predictor of FMI (r2 = 0.83) and a moderately strong predictor of FFMI (r2 = 0.49) in children with CP (both p < 0.01). Prediction of FMI (R2 = 0.86) and FFMI (R2 = 0.66) from BMI increased (p < 0.05) when age, sex and ambulatory status were included. CONCLUSION: Compared with typically developing children, children with CP have a higher FMI and lower FFMI for a given BMI, which is more pronounced in NACP than ACP. The finding suggests that the prevalence of overweight/obesity status may be underestimated in children with CP.

Mental health disorders, participation, and bullying in children with cerebral palsy.

AIM: To examine how social factors might mitigate the elevated risk of mental health disorders in children with cerebral palsy (CP). METHOD: This cross-sectional study included 6- to 17-year-olds with (n=111; 40.4% 6-11y, 59.6% 12-17y) and without (n=29 909; 50.2% 6-11y, 49.8% 12-17y) CP from the 2016 National Survey of Children's Health. Mental health disorders included depression, anxiety, behavior/conduct problems, and attention-deficit/hyperactivity disorder. Social factors included participation in activities, bully victimization, and difficulty with friendships. RESULTS: After adjusting for sociodemographic factors and the presence of chronic pain, children with CP had higher odds of anxiety (odds ratio [OR] 4.4; 95% confidence interval [CI] 1.9-8.5), behavior/conduct problems (OR 3.9; 95% CI 1.4-11.3), and multimorbidity (OR 2.8; 95% CI 1.1-7.0), but not depression (OR 1.4; 95% CI 0.6-3.8) or attention-deficit/hyperactivity disorder (OR 1.7; 95% CI 0.6-4.6), compared to controls. With adjustment for participation in activities, the odds of anxiety, behavior/conduct problems, and multimorbidity remained increased in children with CP. With adjustment for difficulty with friendships, the odds of anxiety, behavior/conduct problems, and multimorbidity were no longer increased in children with CP. With adjustment for bully victimization, the odds of behavior/conduct problems and multimorbidity were attenuated in children with CP; however, the odds of anxiety remained increased. INTERPRETATION: The elevated prevalence of certain mental health disorders in children with CP is partly associated with modifiable social factors. WHAT THIS PAPER ADDS: Difficulty with friendships predicts an elevated prevalence of psychiatric conditions in children with cerebral palsy (CP). Bully victimization predicts an elevated prevalence of behavior/conduct problems in children with CP. Low participation does not predict mental health disorders in this population.

Mental health disorders and physical risk factors in children with cerebral palsy: a cross-sectional study.

AIM: To examine the prevalence of mental health disorders among children with and without cerebral palsy (CP), and to examine how physical risk factors in children with CP might mitigate any elevated risk of mental health disorders in this population. METHOD: Children from 6 years to 17 years of age with (n=111) and without (n=29 909) CP from the 2016 National Survey of Children's Health were included in this cross-sectional study. Mental health disorders included depression, anxiety, behavior/conduct problems, and attention deficit disorder/attention-deficit/hyperactivity disorder (ADHD). Physical risk factors included physical activity (number of active days ≥60min), sleep duration, and pain. RESULTS: Adjusting for sociodemographics, children with CP had higher odds of mental health disorders (odds ratio [OR]=2.7-7.1, p<0.05) except for attention deficit disorder/ADHD (OR=2.5; 95% confidence interval [CI]=0.9-7.1). Further adjusting for physical factors, the odds of depression were no longer increased (i.e. attenuated) in children with CP (OR=1.0; 95% CI=0.3-3.3); however, the odds of anxiety (OR=3.8; 95% CI=1.9-7.8) and behavior/conduct problems (OR=3.8; 95% CI=1.3-11.1) remained elevated. Assessed individually, low physical activity and pain attenuated the odds of depression in children with CP (OR=1.9; 95% CI=0.7-5.3; OR=1.4; 95% CI=0.6-3.8 respectively). INTERPRETATION: Children with CP have an elevated prevalence of mental health disorders even after accounting for physical risk factors. Low physical activity and pain partially accounts for the association between CP and depression. WHAT THIS PAPER ADDS: Children with cerebral palsy (CP) have an elevated risk of developing mental health disorders. Physical factors do not fully account for higher mental health disorder prevalence. Physical activity partially accounts for the relationship between CP and depression. Pain partially accounts for the relationship between CP and depression.

TRASTORNOS DE SALUD MENTAL Y FACTORES DE RIESGO FÍSICO EN NIÑOS CON PARÁLISIS CEREBRAL: ESTUDIO TRANSVERSAL: OBJETIVO: Examinar la prevalencia de trastornos de salud mental en niños con y sin parálisis cerebral (PC), y examinar cómo los factores de riesgo físicos en niños con PC pueden mitigar cualquier riesgo elevado de trastornos de salud mental en esta población. MÉTODO: Los niños de 6 a 17 años de edad con (n=111) y sin (n=29.909) PC de la Encuesta nacional de salud infantil 2016 se incluyeron en este estudio transversal. Los trastornos de salud mental incluían depresión, ansiedad, problemas de conducta / comportamiento, y trastorno por déficit de atención / hiperactividad (TDAH). Los factores de riesgo físico incluían actividad física (número de días activos ≥60min), duración del sueño y dolor. RESULTADOS: Al ajustarse a los datos sociodemográficos, los niños con PC tenían mayores probabilidades de trastornos de salud mental (razón de probabilidades [OR] = 2,7−7,1, p<0,05) excepto por el trastorno por déficit de atención / TDAH (OR=2,5; intervalo de confianza del 95% [IC]=0,9−7,1). Al ajustar aún más los factores físicos, las probabilidades de depresión ya no aumentaron (es decir, se atenuaron) en niños con PC (OR=1,0; 95% CI=0,3−3,3); sin embargo, las probabilidades de ansiedad (OR=3,8; IC 95%=1,9−7,8) y problemas de conducta/conducta (OR=3,8; IC 95%=1,3−11,1) se mantuvieron elevadas. Evaluados individualmente, la actividad física baja y el dolor atenuaron las probabilidades de depresión en niños con PC (OR=1,9; IC del 95%=0,7−5,3; OR=1,4; IC del 95%=0,6, −3,8 respectivamente). INTERPRETACIÓN: Los niños con PC tienen una prevalencia elevada de trastornos de salud mental incluso después de tener en cuenta los factores de riesgo físicos. La baja actividad física y el dolor explican parcialmente la asociación entre PC y depresión.

DESORDENS DA SAÚDE MENTAL E FATORES DE RISCO FÍSICOS PARA CRIANÇAS COM PARALISIA CEREBRAL: ESTUDO TRANSVERSAL: OBJETIVO: Examinar a prevalência de desordens da saúde mental entre crianças com e sem paralisia cerebral (PC), e examiner como fatores de risco físico em crianças com PC podem mitigar qualquer risco elevando de desordens da saúde mental nesta população. MÉTODO: Crianças de 6 a 17 anos de idade com (n=111) e sem (n=29 909) PC no Levantamento Nacional de Saúde da Criança de 2016 foram incluídas neste estudo transversal. Desordens da saúde mental incluíram depressão, ansiedade, problemas de comportamento/conduta, e transtorno do deficit de atenção/atenção e hiperatividade (TDAH). Fatores de risco físico incluíram atividade física (número de dias ativos ≥60min), duração do sono e dor. RESULTADOS: Ajustando para variáveis sócio-demográficas, crianças com PC tiveram maior risco de desordens da saúde mental (odds ratio [OR]=2,7−7,1, p<0,05) exceto para transtorno de deficit de atenção/TDAH (OR=2,5; intervalo de confiança 95% [IC]=0,9−7,1). Ajustando para fatores físicos, os riscos de depressão não eram mais aumentados (ou seja, atenuados) em crianças com PC (OR=1,0; IC 95%=0,3−3,3); no entanto, os riscos de ansiedade (OR=3,8; IC 95%=1,9−7,8) e problemas de comportamento/conduta (OR=3,8; IC 95% =1,3−11,1) permaneceram elevados. Avaliando individualmente, baixos níveis de atividade física e dor atenuaram os riscos de depressão em crianças com PC (OR=1,9; IC 95% =0,7−5,3; OR=1,4; IC 95%=0,6, −3,8 respectivamente). INTERPRETAÇÃO: Crianças com PC tem prevalência aumentada de desordens da saúde mental, mesmo considerando fatores de risco físicos. Ativiade física baixa e dor respondem parcialmente pela associação entre PC e depressão.

Statistical Models to Assess Leg Muscle Mass in Ambulatory Children With Spastic Cerebral Palsy Using Dual-Energy X-Ray Absorptiometry.

Cerebral palsy (CP) is a movement disorder associated with small and weak muscles. Methods that accurately assess muscle mass in children with CP are scarce. The purpose of this study was to determine whether dual-energy X-ray absorptiometry (DXA) accurately estimates midleg muscle mass in ambulatory children with spastic CP. Ambulatory children with spastic CP and typically developing children 5-11 y were studied (n = 15/group). Fat-free soft tissue mass (FFST) and fat mass at the middle third of the tibia (i.e., midleg) were estimated using DXA. Muscle mass (muscleMRI) and muscle mass corrected for intramuscular fat (muscleMRIfc) in the midleg were estimated using magnetic resonance imaging (MRI). Statistical models were created to predict muscleMRI and muscleMRIfc using DXA. Children with CP compared to typically developing children had lower FFST (38%), muscleMRI (40%) and muscleMRIfc (47%) (all p < 0.05) and a lower ratio of muscleMRIfc to FFST (17%, p < 0.05). DXA-based models developed using data from typically developing children overestimated muscleMRI (13%) and muscleMRIfc (22%) (both p < 0.05) in children with CP. DXA-based models developed using data from children with CP explained 91% of the variance in muscleMRI and 90% of the variance in muscleMRIfc in children with CP (both p < 0.05). Moreover, the estimates were not different from muscleMRI and muscleMRIfc (both p > 0.99). We conclude that DXA-based statistical models accurately estimate midleg muscle mass in children with CP when the models are composed using data from children with CP rather than typically developing children.

Site-Specific Transmission of a Floor-Based, High-Frequency, Low-Magnitude Vibration Stimulus in Children With Spastic Cerebral Palsy.

OBJECTIVE: To determine the degree to which a high-frequency, low-magnitude vibration signal emitted by a floor-based platform transmits to the distal tibia and distal femur of children with spastic cerebral palsy (CP) during standing. DESIGN: Cross-sectional study. SETTING: University research laboratory. PARTICIPANTS: Children with spastic CP who could stand independently (n=18) and typically developing children (n=10) (age range, 4-12y) participated in the study (N=28). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: The vibration signal at the high-frequency, low-magnitude vibration platform (approximately 33Hz and 0.3g), distal tibia, and distal femur was measured using accelerometers. The degree of plantar flexor spasticity was assessed using the Modified Ashworth Scale. RESULTS: The high-frequency, low-magnitude vibration signal was greater (P<.001) at the distal tibia than at the platform in children with CP (.36±.06g vs .29±.05g) and controls (.40±.09g vs .24±.07g). Although the vibration signal was also higher at the distal femur (.35±.09g, P<.001) than at the platform in controls, it was lower in children with CP (.20±.07g, P<.001). The degree of spasticity was negatively related to the vibration signal transmitted to the distal tibia (Spearman ρ=-.547) and distal femur (Spearman ρ=-.566) in children with CP (both P<.05). CONCLUSIONS: A high-frequency, low-magnitude vibration signal from a floor-based platform was amplified at the distal tibia, attenuated at the distal femur, and inversely related to the degree of muscle spasticity in children with spastic CP. Whether this transmission pattern affects the adaptation of the bones of children with CP to high-frequency, low-magnitude vibration requires further investigation.

Age, sex, and multi-morbidity stratified mortality risk estimates for adults with cerebral palsy to inform clinical decision making.

BACKGROUND: While research has provided key insights into mortality rates and risks for individuals with cerebral palsy (CP), clinically useable mortality risk estimates remain unreported for adults with CP, especially by key patient-level factors. OBJECTIVE: The objective of this study was to generate clinically useable mortality risk estimates among adults with CP to inform clinical decision making. METHODS: This retrospective cohort study, using a fee-for-service Medicare database, identified adults ≥18-years-old with CP from 01/01/2008-12/31/2010 and followed through 12/31/2019 for death. Mortality risk at 1-, 3-, 5-, and 9-year intervals were selected based on common clinical length of time to reasonably benefit from preventive care. Sex-stratified analyses assessed risk estimates by narrow age group (18-25/26-34/35-44/45-54/55-64/65-74/≥75 years old) and multi-morbidity group (Whitney Comorbidity Index score 0-2/3/4-6/≥7). RESULTS: Of 24,767 adults with CP, n = 12,962 were men (mean [SD] age = 48.3 [15.0] years) and n = 11,805 were women (age = 49.7 [15.8] years). Loss to follow-up was rare. 1-year risk was similar between men and women (3.4 % vs. 3.3 %), but increased slightly more for men than women through 9-years (30.1 % vs. 28.0 %). As expected, the mortality risk increased with older age and higher WCI scores. The probability of death (and survival) is presented per age and multi-morbidity group for men and women with CP. CONCLUSIONS: Mortality risk estimates were reported at clinically relevant intervals by age, sex, and multi-morbidity status. This information can be used to weigh harm-to-benefit ratios of screening and treatment strategies based on mortality expectancy estimates.

Comorbidity Clusters Among Adults With Cerebral Palsy: A Latent Class Analysis.

INTRODUCTION: Adults with cerebral palsy are at risk for early multimorbidity onset, but little is known about the composition of multimorbidity profiles or how these profiles present across adulthood. The objective of this study was to identify multimorbidity profiles and association with mortality among adults with cerebral palsy. METHODS: This retrospective cohort study used a random 20% fee-for-service Medicare database from January 1, 2008 to December 31, 2019 from adults aged ≥18 years with cerebral palsy. Latent class analyses using 4-class models were conducted within each age cohort (young adults aged 18-39 years, middle adulthood aged 40-64 years, and older adults aged ≥65 years) to determine patterns of 30 comorbidities defined using the International Classification of Diseases, Ninth Revision codes, identified from January 1, 2008 to December 31, 2010, and their association with mortality through December 31, 2019 (up to 11 years of follow-up); statistical analysis was performed in 2023. RESULTS: Three classes were relatively consistent in the composition of comorbidities across young (n=7,020), middle (n=13,554), and older (n=4,193) cohorts: (1) low morbidity (low proportion of all comorbidities) representing 50.1% (young), 41.4% (middle), and 30.9% (older) of the cohorts; (2) neurologic multimorbidity (e.g., epilepsy, intellectual disabilities) representing 26.0% (young), 26.6% (middle), and 14.7% (older) of the cohorts; and complex multimorbidity (e.g., cardiorespiratory, nutritional, musculoskeletal, neurologic) representing 26.0% (young), 26.6% (middle), and 14.7% (older) of the cohorts. The fourth class varied by young (mental health disorders), middle (hypertension), and older (hypertension and osteoarthritis) age cohorts. Compared with the low morbidity class, other classes had an increased mortality rate for each age cohort (hazard ratio range=1.34-5.58, all p<0.001). CONCLUSIONS: Findings provide insight into varied multimorbidity profiles and associations with mortality across the life course for adults with cerebral palsy.

Opioid prescription patterns among commercially insured children with and without cerebral palsy.

PURPOSE: This study aimed to describe opioid prescription patterns for children with vs. without cerebral palsy (CP). METHODS: This cohort study used commercial claims from 01/01/2015-12/31/2016 and included children aged 2-18 years old with and without CP. Opioid prescription patterns (proportion exposed, number of days supplied) were described. A zero-inflated generalized linear model compared the proportion exposed to opioids in the follow-up year (2016) and, among those exposed, the number of days supplied opioids between cohorts before and after adjusting for age, gender, race, U.S. region of residence, and the number of co-occurring neurological/neurodevelopmental disabilities (NDDs). RESULTS: A higher proportion of children with (n = 1,966) vs. without (n = 1,219,399) CP were exposed to opioids (12.1% vs. 5.3%), even among the youngest age group (2-4 years: 9.6% vs. 1.8%), and had a greater number of days supplied (median [interquartile range], 8 [5-13] vs. 6 [4-9] days; P < 0.05). Comparing children with opioid exposure with vs. without CP, a greater number of days supplied was identified for older age, Asian race/ethnicity, and without co-occurring NDDs, and a lower number of days supplied was observed for Black race/ethnicity and with ≥1 co-occurring NDDs. CONCLUSION: Children with CP are more likely to be exposed to opioids and have a higher number of days supplied.