Abstract
Background:
Individuals living with cerebral palsy (CP) or spina bifida (SB) are at heightened risk for a number of chronic health conditions such as secondary comorbidities, that may develop or be influenced by the disability, the presence of impairment, and/or the process of aging. However, very little is known about the prevalence and/or risk of developing secondary-comorbidities among individuals living with CP or SB throughout adulthood. The objective of this study was to compare the prevalence of psychological, cardiometabolic, musculoskeletal morbidity, and multimorbidity among adults with and without CP or SB.
Methods:
Privately-insured beneficiaries were included if they had an ICD-9-CM diagnostic code for CP or SB (n= 29,841). Adults without CP or SB were also included (n= 5,384,849). Prevalence estimates of common psychological, cardiometabolic, and musculoskeletal morbidity and multimorbidity (≥2 conditions) were compared.
Results:
Adults living with CP or SB had a higher prevalence of all psychological disorders and psychological multimorbidity (14.6% vs 5.4%), all cardiometabolic disorders and cardiometabolic multimorbidity (22.4% vs. 15.0%), and all musculoskeletal disorders and musculoskeletal multimorbidity (12.2% vs. 5.4%), as compared to adults without CP or SB, and differences were to a clinically meaningful extent.
Conclusions:
Adults with CP or SB have a significantly higher prevalence of common psychological, cardiometabolic, and musculoskeletal morbidity and multimorbidity, as compared to adults without CP or SB. Efforts are needed to facilitate the development of improved clinical screening algorithms and early interventions to reduce risk of disease onset/progression in these higher risk populations.
Keywords: cerebral palsy, spina bifida, chronic disease, multimorbidity
Introduction
Cerebral Palsy (CP) is the most common pediatric-onset physical disability with an estimated prevalence ranging from 2.6–3.1 cases per 1,000 live births in the U.S.1 CP is caused by an insult to, or malformation of the developing brain which affects motor control centers, and causes alterations in growth, development, and overall health throughout the lifespan.2 The population of adults with CP is expanding because of the steady or marginally increased prevalence and increases in the childhood survival rate in recent decades.3–5 Spina bifida (SB) is another congenital birth defect that occurs in fewer (3 of 10,000) live births in the U.S.,6 and encompasses a spectrum of birth defects (meningomyelocele, myelomeningocele, myelocele, meningocele, and rachischisis), which are the result of an incomplete closure of the spinal column and lead to exposure of or herniation to the spinal cord/meninges.7 Although SB has a lower case fatality rate than other neural tube defects, it often results in severe life-long disability and morbidity.8,9
The framework that encompasses clinical care for patients with CP and SB has been largely confined to issues that arise during childhood and adolescence. Despite the shortage of research to track lifelong health and chronic disease trajectories in both of these populations, there is ample evidence demonstrating that individuals with CP and SB have significant and progressive motor impairment, excessive sedentary behavior profiles, inadequate muscle and bone development, increased obesity, and risk for secondary chronic disease.10–18 However, there have been very few U.S. population-representative studies of CP and SB to examine risk of common, age-related chronic disease and multimorbidity across multiple organ systems.19,20 Having CP or SB increases the risk for secondary health conditions that are causally linked to the impairments (e.g., muscle spasticity, chronic pain), or occurs as an indirect consequence of the disability itself (e.g., lack of physical activity and related conditions such as hypertension). Consequently, there is a need for approaching health care delivery for patients with CP or SB within the context of a “life course health development model”.21 In spite of a lack of surveillance to track health outcomes in this population, there is growing evidence that shows progressive motor impairment, diminished musculoskeletal density, chronic overlapping pain, respiratory complications, cardiometabolic disease risk factors, and excessive sedentary behavior profiles in adults with CP,22–26 all of which progress with age. These factors increase the risk for adults with CP and SB to experience secondary chronic conditions and mental health disorders that further worsen quality of life, and can lead to decreased independence. Therefore, the purpose of this study was therefore to examine the prevalence of common psychological, cardiometabolic, and musculoskeletal morbidity and multimorbidity in a large sample of adults with CP or SB, as compared to adults without CP or SB.
Methods
Data Source
The Clinformatics™ Data Mart Database (OptumInsight, Eden Prairie, MN), is a de-identified nationwide claims database of all beneficiaries from a single private payer. This is a de-identified administrative claims database of over 80 million adults and children with commercial insurance representing those on a single, large U.S. private payer who had both medical and pharmacy coverage throughout the enrollment. Data are organized by a patient identification number, which ensures longitudinal follow-up even if there were changes in plan details or gaps in insurance coverage. Since the are data de-identified and from administrative claims, the University of Michigan Institutional Review Board approved this as a non-regulated study, and thus subjects were not required to provide written informed consent.
Sample Selection
All individuals 18 years of age and older at the time of their enrollment which could start from 2007 to 2015 were potentially eligible for this analysis. We excluded individuals with less than 12 months of continuous enrollment to require sufficient claim history. All medical claims excluding laboratory and outpatient pharmacy was considered to identify prevalence or treatment for these conditions during the enrollment period.
Identification of Patients with CP and SB
The diagnosis of CP or SB was identified based on a single encounter that included one of the pertinent International Classification of Diseases, Ninth Revision, Clinical Modification (ICD-9) codes for CP or SB (see Appendix 1 for list of codes).
Chronic Disease Outcomes and Multimorbidity
All chronic disease outcomes were chosen based on established categories through the AHRQ indicators of clinical classification software (CCS).27 CCS is a software tool that aggregates ICD-9-CM diagnoses codes into higher levels of clinical classifications. The decision to follow the AHRQ clinical definitions was made a priori to provide uniformity
Psychological Morbidities
Physician-diagnosed psychological health disorders were identified based on a single encounter that included at least one of pertinent ICD-9 codes (in any position) (see Appendix 1 for list). The psychological health disorders were grouped into eleven categories: (1) Insomnia; (2) Adjustment disorders; (3) Anxiety disorders; (4) Post-traumatic stress disorders; (5) Dementia/Amnestic/Other Cognitive Disorders; (6) Impulse control disorders; (7) Mood affective disorders (e.g., depression); (8) Personality disorders; (9) Alcohol-related disorders; (10) Substance-related disorders; and (11) Central pain disorders. We defined psychological multimorbidity as the onset of at least two of the aforementioned psychological morbidities.
Cardiometabolic Morbidities
Physician-diagnosed cardiometabolic health disorders were identified based on a single encounter that included at least one of pertinent ICD-9 codes (in any position) (see Appendix 1 for list). The cardiometabolic health disorders were grouped into eight categories: (1) Cardiac dysrhythmias; (2) Heart failure; (3) Peripheral and visceral atherosclerosis; (4) Non-alcoholic fatty liver disease; (5) Chronic kidney disease; (6) Type 2 diabetes; (7) Hypercholesterolemia; and (8) Hypertension. We defined cardiometabolic multimorbidity as the onset of at least two of the aforementioned cardiometabolic morbidities.
Musculoskeletal Morbidities
Physician-diagnosed musculoskeletal health disorders were identified based on a single encounter that included at least one of pertinent ICD-9 codes (in any position) (see Appendix 1 for list). The musculoskeletal health disorders were grouped into eight categories, including: (1) osteoarthritis; (2) osteoporosis; (3) pathologic fracture; (4) disorders of muscle, joint, ligament, tendons, and connective tissue (e.g., upper extremity tendonitis; synovitis and tenosynovitis; other disorders of synovium and tendon [e.g., synovial hypertrophy]; bursitis; enthesopathies-lower limb [e.g., prepatellar bursitis, etc.]; other enthesopathies [e.g., lateral epicondylitis]; other and unspecified soft tissue disorders, not elsewhere classified [e.g., panniculitis]; calcification and ossification of muscle); (5) sarcopenia and weakness; (6) myalgia; and (7) rheumatoid arthritis, myositis, and musculoskeletal infections. We defined musculoskeletal multimorbidity as the onset of at least two of the aforementioned musculoskeletal morbidities.
Statistical Analysis
Patient characteristics were summarized using means and standard deviations (SDs) for continuous variables and frequencies and percentages for categorical variables. The primary analysis was carried out to compare the prevalence estimates of each of the primary psychological, cardiometabolic, and musculoskeletal morbidities, as well as multimorbidity, between adults with CP or SB, as compared to adults without CP or SB. Standardized mean differences via effect size (ES) calculations using Cohen’s h were used in conjunction with formal p-value determination of significance to better understand a clinically meaningful effect size, with SMD≥0.1 determined to be a clinically meaningful difference, as previously described.28 All analyses were performed using SAS version 9.4 (SAS Institute, Cary, NC).
Results
The mean time in the plan for eligible enrollees was 4.3±2.9 and 3.3±2.6 years for patients with CP or SB and controls respectively, and ranged from one year (inclusion criterion) to 11 years (Table 1). Adults with CP or SB had a higher prevalence of any psychological, cardiometabolic, and musculoskeletal diseases, as compared to adults without CP or SB (Figure 1).
Table 1.
Descriptive characteristics among adults with and without CP or SB.
| CP/SB (N= 29,841) | Controls (N=5,384,849) | ||
|---|---|---|---|
| Years Post Eligibility Start Date | |||
| N/A | N/A | ||
| 5171 (17.3%) | 1680768 (31.2%) | ||
| 4844 (16.2%) | 1105196 (20.5%) | ||
| 4524 (15.2%) | 803906 (14.9%) | ||
| 3596 (12.1%) | 505015 (9.4%) | ||
| 2778 (9.3%) | 348366 (6.5%) | ||
| 8928 (29.9%) | 941598 (17.5%) | ||
| Age Group | |||
| 15633 (52.4%) | 2728716 (50.7%) | ||
| 9256 (31.0%) | 1666194 (30.9%) | ||
| 4952 (16.6%) | 989939 (18.4%) | ||
| Gender | |||
| 16951 (56.8%) | 2764545 (51.3%) | ||
| 12890 (43.2%) | 2620304 (48.7%) | ||
| Race | |||
| 18282 (61.3%) | 3208220 (59.6%) | ||
| 2924 (9.8%) | 453498 (8.4%) | ||
| 2479 (8.3%) | 551264 (10.2%) | ||
| 618 (2.1%) | 235385 (4.4%) | ||
| 5538 (18.6%) | 936482 (17.4%) | ||
| Education | |||
| 166 (0.6%) | 40275 (0.7%) | ||
| 9028 (30.3%) | 1418489 (26.3%) | ||
| 15549 (52.1%) | 2744683 (51.0%) | ||
| 4290 (14.4%) | 979408 (18.2%) | ||
| 808 (2.7%) | 201994 (3.8%) | ||
| Net Worth | |||
| 6840 (22.9%) | 1097073 (20.4%) | ||
| 6347 (21.3%) | 911024 (16.9%) | ||
| 5326 (17.8%) | 989019 (18.4%) | ||
| 2771 (9.3%) | 553192 (10.3%) | ||
| 4087 (13.7%) | 846968 (15.7%) | ||
| 4470 (15.0%) | 987573 (18.3%) | ||
Figure 1.
Proportion of any psychological, cardiometabolic, and musculoskeletal morbidities among adults with and without CP or SB.
Psychological Morbidities
Adults with CP or SB had significantly higher prevalence of most of the psychological outcomes, including insomnia (5.0% vs. 2.7%), anxiety disorders (13.6% vs 6.6%), dementia disorders (4.0% vs 1.2%), impulse control disorders (0.5% vs 0.0%), mood affective disorders (18.3% vs 7.7%), substance abuse disorders (2.5% vs 1.0%), central pain disorders (7.7% vs 2.1%), and psychological multimorbidity (14.6% vs 5.4%), as compared to adults without CP or SB (all P<.01 and SMD≥0.1) (Table 2).
Table 2.
Prevalence of psychological, cardiometabolic, and musculoskeletal morbidities and multimorbidities among adults with and without CP or SB.
| CP/SB (N= 29,841) | Controls (N=5,384,849) | |
|---|---|---|
| Psychological | ||
| 9963 (33.4%)* | 887952 (16.5%) | |
| 1484 (5.0%)* | 142766 (2.7%) | |
| 919 (3.1%) | 92192 (1.7%) | |
| 4069 (13.6%)* | 357772 (6.6%) | |
| 245 (0.8%) | 14848 (0.3%) | |
| 1182 (4.0%)* | 62211 (1.2%) | |
| 396 (1.3%) | 24239 (0.5%) | |
| 152 (0.5%)* | 2615 (0.0%) | |
| 5461 (18.3%)* | 416251 (7.7%) | |
| 190 (0.6%) | 8171 (0.2%) | |
| 419 (1.4%) | 48140 (0.9%) | |
| 753 (2.5%)* | 54217 (1.0%) | |
| 2287 (7.7%)* | 112587 (2.1%) | |
| 4349 (14.6%)* | 292338 (5.4%) | |
| Cardiometabolic | ||
| 12797 (42.9%)* | 1669545 (31.0%) | |
| 3457 (11.6%)* | 336317 (6.2%) | |
| 1218 (4.1%)* | 95708 (1.8%) | |
| 1811 (6.1%)* | 124491 (2.3%) | |
| 463 (1.6%) | 43295 (0.8%) | |
| 1048 (3.5%)* | 100535 (1.9%) | |
| 3691 (12.4%)* | 489396 (9.1%) | |
| 3257 (10.9%) | 492780 (9.2%) | |
| 9428 (31.6%)* | 1273233 (23.6%) | |
| 6673 (22.4%)* | 806146 (15.0%) | |
| Musculoskeletal | ||
| 13254 (44.4%)* | 1237175 (23.0%) | |
| 619 (2.1%) | 55491 (1.0%) | |
| 3620 (12.1%)* | 368370 (6.8%) | |
| 1546 (5.2%)* | 134165 (2.5%) | |
| 218 (0.7%) | 15738 (0.3%) | |
| 11661 (39.1%)* | 1007360 (18.7%) | |
| 2064 (6.9%)* | 65404 (1.2%) | |
| 2116 (7.1%)* | 171957 (3.2%) | |
| 3626 (12.2%)* | 292755 (5.4%) |
P<.01 and standard mean difference (SMD) ≥0.1
Cardiometabolic Morbidities
Adults with CP or SB also had significantly higher prevalence of most of the cardiometabolic outcomes, including cardiac dysrhythmias (11.6% vs. 6.2%), heart failure (4.16% vs 1.8%), peripheral and visceral atherosclerosis (6.1% vs. 2.3%), type 2 diabetes (12.4% vs. 9.0%), hypertension (31.6% vs. 23.6%) and cardiometabolic multimorbidity (22.4% vs. 15.0%) (all P<.01 and SMD≥0.1) (Table 2).
Musculoskeletal Morbidities
Adults with CP or SB had significantly higher prevalence of most of the musculoskeletal outcomes, including osteoarthritis (12.1% vs. 6.8%), osteoporosis (5.2% vs. 2.5%), other connective tissue disease (39.1% vs. 18.7%), sarcopenia (6.9% vs. 1.1%), myalgia (7.1% vs. 3.2%), and musculoskeletal multimorbidity (12.2% vs. 5.4%) (all P<.01 and SMD≥0.1) (Table 2).
Discussion
The principal finding of this study was that adults living with CP or SB had a higher prevalence of most psychological, cardiometabolic, and musculoskeletal morbidities and multimorbidities than adults without CP or SB. This is the largest study to date to examine prevalence estimates of chronic diseases across multiple organ systems in adults living with CP or SB. Future research and clinical efforts are needed to not only better understand the healthcare burden associated with these conditions in the CP and SB populations, as well as across other subpopulations with neurodevelopmental and acquired physical disabilities, but also to understand the health disparities in access and disease progression experienced between privately- and federally-insured beneficiaries living with disabilities. Moreover, these findings support the need for improfved clinical screening algorithms and design of early behavioral interventions to reduce risk of disease onset/progression in these higher risk population.
These factors may further worsen functional status and quality of life, and can lead to decreased independence and early mortality.29,30 Although both CP and SB are considered “non-progressive” conditions, there is ample evidence that both represent a premature aging phenotype.23 Previous reports have demonstrated that chronic pain is the most commonly reported physical symptomology of CP and SB throughout the lifespan, and yet pain is perhaps the least understood comorbidity in these populations. Recent evidence has linked chronic pain and mood affective disorders among adults living with CP and other neurodevelopmental disorder;31 and yet, very little is known about the natural history of pain subtypes in these populations. Most studies on pain in CP or SB have focused on general pain prevalence, with some studies documenting pain intensity, location and presence of co-occurring mood disorders and fatigue.32–34 Chronic centralized pain and neuropathic pain have been found to be higher among adults with CP and SB,35 and thus future studies are needed to better understand how/if chronic pain may mediate the association between having CP or SB, and development of age-related noncommunicable diseases.
Noncommunicable diseases and multimorbidity are increasingly burdensome with the growing population of older adults in the U.S. Care coordination and healthcare access are woefully insufficient in meeting the complex healthcare needs of individuals with pediatric-onset disabilities (including CP and SB) across the lifespan.36–39 The number of people who live with these complex conditions is increasing, and they endure significant health inequalities as they age.5 Consequently, they experience lower quality of life and are at risk for early mortality-often from undiagnosed, yet preventable diseases.30 Despite the increasing number of adults living with CP and SB, there is a complete lack of coordinated services and adequately trained specialists to treat them, as these patients are still viewed collectively as having a “pediatric” medical condition. This is unquestionably troubling for youth with disabilities in the U.S., as they transition from parents’ insurance and are left to navigate our confusing and disjointed options for continued healthcare and services. Indeed, adults living with pediatric-onset disabilities in the U.S. consistently report feeling deserted by their health professionals when they are discharged from pediatric care, and struggle to access appropriate care when they begin experiencing declines in health and function in early adulthood. As an example, a recent report. demonstrated a consistent and significant rise in the population of adults with diseases originating in childhood, who remain discharged to or hospitalized at children’s hospitals.40 Policy and thought leadership are essential to increase research and promote best practices in preventive screening, diagnosis, and disease management. Simultaneously, we need to convene hospitals, insurers, and consumer groups to develop systematic, coordinated care options for these medically underserved, high-risk populations. These models could be integrated into primary care facilities as well as specialty or therapy clinics, community living centers, and even assisted living institutions.
Strengths and Weaknesses
A major strength of this study is the large sample of adults with CP or SB. It can be challenging to gather data on clinical sub-populations, and very little is known about health outcomes among individuals with CP or SB as they transition into and throughout adulthood. Another strength of this study is the number of chronic diseases across multiple organ systems that were investigated. Our comprehensive assessment of medically-diagnosed health disorder profiles among individuals with CP or SB throughout the adult lifespan may prompt the development of improved screening strategies and identification of individuals for early risk.
Our study also has several limitations that should be acknowledged. First, the sample with CP or SB may not be entirely representative of the U.S. population of adults with CP or SB, as further evidenced by a lower than expected prevalence. We were unable to determine the severity of CP or SB through claims-based data. However, we suspect that our sample may be more reflective of a healthier, higher functioning segment of the CP or SB population,41 because they had to be enrolled in private insurance, either by purchasing their own insurance, or by being covered through employment or marriage to someone who had private insurance. Individuals with more severe forms of CP or SB may be more likely to be on federally-subsidized health insurance, Medicare, or Medicaid state-sponsored programs. Therefore, results and comparisons to adults without CP or SB are likely conservative estimates, and the true extent of chronic disease may be underestimated in this study. Importantly, administrative claims data may be prone to inaccurate coding of medical diagnoses, such as CP or SB, as well as chronic diseases, which may have an effect on our prevalence estimates. While validation studies have shown that using >1 claim for a medical condition improves the ability to identify beneficiaries with that medical condition,42,43 single claim-based algorithms have been reported to have moderate-to-high positive predictive value (~80%) or specificity (up to 96%).42,44,45 However, the accuracy of identifying medical conditions using claims data depends on the number of years for the study period44 and the medical condition examined.42,44–46 Finally, we were unable to determine or account for the pathological etiologies of the CP or SB diagnoses (e.g., extent of white matter damage). It is possible that the extent, severity, and compensatory mechanisms for recovery from the initial brain damage leading to CP, or extent of meningocele leading to SB, may interfere with development in early life, and accelerate decline with age. Future studies are needed to disentangle the pathological features of CP and SB with the development of secondary disorders in this population.
Conclusion
In conclusion, adults with CP or SB have an elevated of a variety of chronic diseases and multimorbidity compared to the general adult population of privately insured beneficiaries without CP or SB. Individuals with CP and SB frequently utilize healthcare services as part of their routine clinical care. Therefore, increasing clinical awareness of the secondary chronic diseases and risks among adults with CP and SB, improving clinical screening strategies, and developing efficient referral resources for mental health care services may help reduce the burden of physical and mental health disorders in these population. Future research is also needed to better understand the burden of and healthcare access disparities associated with these conditions in the CP and SB populations, and other subpopulations with neurodevelopmental and acquired physical disabilities across the lifespan.
Supplementary Material
What is Known:
Individuals living with cerebral palsy or spina bifida are at heightened risk for a number of chronic health conditions such as secondary comorbidities, that may develop or be influenced by the disability, the presence of impairment, and/or the process of aging.
What is New:
Adults living with cerebral palsy or spina bifida have a significantly higher prevalence of common psychological (e.g., anxiety and depression, etc.), cardiometabolic (e.g., type 2 diabetes and hypertension, etc.), and musculoskeletal (e.g., osteoporosis and sarcopenia, etc.) morbidities and multimorbidity (≥2 chronic diseases), as compared to adults without cerebral palsy or spina bifida.
Acknowledgments
Funding/Support:
This research was developed in part under a grant from the National Institute on Disability, Independent Living, and Rehabilitation Research (NIDILRR #90RTHF0001-01-00).
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