- Email: [email protected]
Early-Onset Noncommunicable Disease and Multimorbidity Among Adults With Pediatric-Onset Disabilities
ORIGINAL ARTICLE
Early-Onset Noncommunicable Disease and Multimorbidity Among Adults With Pediatric-Onset Disabilities Daniel G. Whitney, PhD; Rachael T. Whitney, PhD; Neil S. Kamdar, MA; Edward A. Hurvitz, MD; and Mark D. Peterson, PhD, MS Abstract Objective: To determine the prevalence of major noncommunicable diseases among young adults with pediatric-onset disabilities (PoDs) compared with young adults without PoDs. Patients and Methods: Data were obtained from the Optum Clinformatics Data Mart, a de-identified nationwide claims database of beneficiaries from a single private payer in the United States. Beneficiaries were included if they were 18 to 40 years old and had an International Classification of Diseases, Ninth Revision, Clinical Modification diagnostic code for a PoD known to originate in childhood. Diagnostic codes were used to identify high-burden noncommunicable diseases: ischemic heart disease, cerebrovascular disease, hypertensive and other cardiovascular disease, type 2 diabetes, malignant cancer, osteoporosis, mood affective disorders, chronic obstructive pulmonary disease, chronic kidney disease, and liver disease. The prevalence of noncommunicable diseases and multimorbidity (2 diseases) was compared between adults with (N¼47,077) and without (N¼2,180,250) PoDs, before and after adjusting for sociodemographic characteristics. This study was conducted between July 1, 2018, and February 1, 2019. Results: Adults with PoDs had higher prevalences and adjusted odds of all noncommunicable diseases (odds ratio, 2.1-9.0; all P<.05) and multimorbidity (odds ratio, 3.8; 95% CI, 3.7-3.9) compared with adults without PoDs. After stratifying by the type of PoD (eg, musculoskeletal, circulatory), all PoD categories had higher prevalence of all noncommunicable diseases and multimorbidity compared with young adults without PoDs, except for ischemic heart disease and cerebrovascular disease among adults with PoDs of the genital organs. Conclusion: Young adults with PoDs have an early onset of several noncommunicable diseases that represent major contributors to the global and national burden of disease and mortality. ª 2019 Mayo Foundation for Medical Education and Research
T
here has been a considerable shift in the global burden disease, with the prevalence of noncommunicable diseases now outranking injuries and communicable, maternal, neonatal, and nutritional disorders combined.1 In the United States, chronic noncommunicable diseases represent the primary causes of death2 and economic burden, with related health care costs slated to reach $7 trillion by 2030.3 Importantly, modifiable risk factors (eg, diet, physical activity, substance use) are associated with the development of many noncommunicable diseases.3 Moreover, there are segments of the population that
n
Mayo Clin Proc. 2019;nn(n):1-9
experience an elevated burden of disease throughout the life span, as well as those with disparities in access and services (eg, individuals with disabilities).4-7 Therefore, identifying at-risk populations is necessary to develop targeted strategies to prevent, treat, and manage the burden of noncommunicable diseases. In particular, pediatric-onset disabilities (PoDs) represent a group of congenital or acquired chronic conditions that originate during pregnancy or in childhood and consist of impairments in behavioral, intellectual, and/ or physical functioning or are associated with abnormal growth/development or
Mayo Clin Proc. n XXX 2019;nn(n):1-9 n https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org n ª 2019 Mayo Foundation for Medical Education and Research
From the Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor (D.G.W., E.A.H., M.D.P.), Department of Kinesiology and Applied Physiology, University of Delaware, Newark (R.T.W.), Institute for Healthcare Policy and Innovation, University of Michigan, Ann Arbor (N.S.K., M.D.P.), Department of Obstetrics and Gynecology, University of Affiliations continued at the end of this article.
1
MAYO CLINIC PROCEEDINGS
physiologic processes. The population of adults with PoDs is projected to expand over the coming decades because of increases in childhood survival rates and prevalence.8-10 This trend is a major public health issue because individuals with PoDs have complex health care needs and are susceptible to an accelerated development of noncommunicable diseases and early mortality, which places a high strain on health care resources.4,5,7,11-22 Further, access to quality health care is inadequate for many individuals with PoDs, which coincides with a lack of clinical knowledge of life-course health development.23-26 The general lack of resources allocated to screening for and treating noncommunicable diseases among adults with PoDs could lead to a disproportionate increase in disease burden and health care costs in the coming decades that may otherwise be preventable. Therefore, a comprehensive understanding of the noncommunicable disease profiles among young adults with PoDs is urgently needed. This knowledge could inform early clinical decision-making processes and strategies to prevent or reduce the burden of noncommunicable diseases among these patients. Accordingly, the objective of this study was to compare the prevalence of major noncommunicable diseases that contribute to a substantial national burden of disease and mortality1,2,27,28 among young adults aged 18 to 40 years with and without PoDs. We hypothesized that young adults with PoDs would have a higher prevalence of all noncommunicable diseases compared with young adults without PoDs. PATIENTS AND METHODS Data Source Data were obtained from the Clinformatics Data Mart Database (Optum, Inc), which is a nationwide, de-identified insurance claims database of 16 million beneficiaries from a single private payer in the year 2016. This administrative database houses information of beneficiaries with both medical and pharmacy coverage. This database has been 2
Mayo Clin Proc.
n
leveraged to examine longitudinal trends of noncommunicable diseases among adults with cerebral palsy,19 a type of PoD, and other conditions and their subsequent complications.29-31 Data are de-identified, and the University of Michigan Institutional Review Board approved this study as nonregulated. This study was conducted between July 1, 2018, and February 1, 2019. Sample Selection We acquired access to the administrative claims data from the most recent available calendar year, 2016, for the analysis. Individuals between 18 and 40 years of age who had 12 months of continuous enrollment were included. All PoD conditions and noncommunicable diseases were identified using the International Classification of Diseases, Tenth Revision, Clinical Modification codes and are presented in the Supplemental Table (available online at http://www. mayoclinicproceedings.org). Using administrative claims, it is not possible to determine if some conditions developed in childhood or in adulthood (eg, mental health disorders, spinal cord injury) because of the exceptionally long enrollment time frames to track children from birth and the discontinuities in enrollment. Therefore, conditions were included in the group representing PoDs if there were known childhood origins (eg, congenital disorders) and were grouped into the following categories: (1) PoDs of the musculoskeletal system, (2) neurodevelopmental PoDs, (3) PoDs of the circulatory system, (4) PoDs of the nervous system, (5) PoDs of the genital organs, (6) other chromosomal abnormalities, (7) PoDs of the urinary system, (8) PoDs of the respiratory system and digestive system, and (9) pediatric-onset malformations of the eye, ear, face, and neck. Of the 10.3 million adults aged 18 years and older with 12 months of continuous enrollment in 2016, 1.6 million members (15.5%) did not have an inpatient, outpatient, or emergency department visit reporting a diagnostic code while enrolled in 2016. We were therefore unable to identify any XXX 2019;nn(n):1-9
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
diagnoses and excluded these cases to eliminate risk of bias. Another 991 (0.01%) individuals were excluded because of missing sex data. Finally, 6.5 million members were excluded because they were over the age of 40 years. The final sample comprised 2,227,327 individuals, including 47,077 adults with at least one PoD. Noncommunicable Diseases Noncommunicable diseases and their variations (eg, severity) were selected with guidance from the literature on disease, disability, and mortality among 1,2,4,5,27,28,32-34 adults and are represented as categories across multiple biological systems including (1) ischemic heart disease (eg, atherosclerotic heart disease), (2) cerebrovascular disease (eg, cerebral infarction), (3) hypertensive and other cardiovascular disease (eg, hypertension, heart failure), (4) type 2 diabetes mellitus, (5) malignant cancer, (6) osteoporosis, (7) mood affective disorders (eg, major depressive disorder), (8) chronic obstructive pulmonary disease (eg, emphysema), (9) chronic kidney disease (eg, kidney disease stages 1-4), and (10) liver disease (eg, cirrhosis). Multimorbidity was defined as the presence of at least 2 of the aforementioned noncommunicable diseases on at least 1 or more service dates during the beneficiaries’ full 12month enrollment. Covariates Covariates that were available in the administrative claims included sociodemographic variables: age, sex, race/ethnicity, and education level. Statistical Analyses Age was summarized as a continuous variable using mean SD, and all other variables were summarized as percentage (frequency) for those with and without PoDs, with subsequent stratification for each PoD category (eg, nervous system, musculoskeletal system). We examined each of the 10 noncommunicable disease categories and multimorbidity as outcomes, and the primary exposure Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
variable was an indicator for PoD, with the referent as without PoDs. We performed multivariable logistic regression analyses for each outcome, adjusting for age, sex, race/ ethnicity, and education level. The main effect of PoD group was interpreted. Because of the extent of missing/unknown for race/ ethnicity, we conducted a sensitivity analysis that included individuals with complete and known data for race/ethnicity to determine if it biased the main effect of PoD group for all outcomes. All analyses were performed using SAS statistical software, version 9.4 (SAS Institute). RESULTS Descriptive characteristics and prevalence of noncommunicable diseases and multimorbidity for young adults with (N¼47,077) and without (N¼2,180,250) PoDs are presented in Table 1. Young adults with PoDs had a higher prevalence of all noncommunicable diseases and multimorbidity compared with young adults without PoDs: ischemic heart disease (1.4% [663] vs 0.3% [7160]); cerebrovascular disease (2.1% [1006] vs 0.3% [5521]); hypertensive and other cardiovascular disease (14.0% [6569] vs 7.1% [155,181]); type 2 diabetes (4.5% [2108] vs 2.4% [51,752]); malignant cancer (2.0% [941] vs 0.9% [19,615]); osteoporosis (0.7% [346] vs 0.1% [2015]); mood affective disorders (18.1% [8525] vs 8.9% [193,908]); chronic obstructive pulmonary disease (0.4% [185] vs 0.1% [2334]); chronic kidney disease (2.1% [974] vs 0.3% [6391]); liver disease (3.5% [1652] vs 1.3% [29,070]); and multimorbidity (10.0% [4716] vs 3.1% [68.539]). The adjusted odds ratios (ORs; adjusting for age, sex, race/ethnicity, and education level) of noncommunicable diseases and multimorbidity are presented in Table 2. Young adults with PoDs had higher adjusted odds of all noncommunicable diseases compared with young adults without PoDs (all P<.05): ischemic heart disease (OR, 4.7; 95% CI, 4.4-5.1); cerebrovascular disease (OR, 9.0; 95% CI, 8.4-9.6); hypertensive and other cardiovascular disease (OR, 2.4; 95% CI, 2.4-2.5); type 2 diabetes (OR, 2.1;
https://doi.org/10.1016/j.mayocp.2019.07.010
3
MAYO CLINIC PROCEEDINGS
TABLE 1. Descriptive Characteristics and Prevalence of Noncommunicable Diseases and Multimorbidity in 2,227,327 Young Adults (18-40 Years) With and Without Pediatric-Onset Disabilitiesa,b Variable Descriptive characteristics Age (y) Sex Female Male Ethnicity White Black Hispanic Asian Unknown/missing Education Less than high school High school diploma More than high school Unknown/missing
With PoD (N¼47,077; 2.1%)
Without PoD (N¼2,180,250; 97.9%)
29.56.6
30.26.3
56.4 (26,544) 43.6 (20,533)
55.3 (1,205,121) 44.7 (975,129)
52.0 6.6 9.0 4.0 28.4
(24,460) (3119) (4227) (1888) (13,383)
47.8 6.8 10.4 5.3 29.7
(1,042,346) (147,412) (227,461) (115,946) (647,085)
0.4 21.4 76.1 2.1
(195) (10,052) (35,843) (987)
0.5 22.0 75.8 1.7
(11,479) (479,884) (1,652,814) (36,073)
Noncommunicable diseases Ischemic heart disease Cerebrovascular disease Hypertensive and other cardiovascular disease Type 2 diabetes Malignant cancer Osteoporosis Mood affective disorders Chronic obstructive pulmonary disease Chronic kidney disease Liver disease Multimorbidity (2 comorbidities) a
1.4 (663) 2.1 (1006) 14.0 (6569)
0.3 (7160) 0.3 (5521) 7.1 (155,181)
4.5 2.0 0.7 18.1 0.4
2.4 0.9 0.1 8.9 0.1
(2108) (941) (346) (8525) (185)
2.1 (974) 3.5 (1652) 10.0 (4716)
(51,752) (19,615) (2015) (193,908) (2334)
0.3 (6391) 1.3 (29,070) 3.1 (68,539)
PoD ¼ pediatric-onset disability. Data are presented as mean SD or percentage (No.) of patients.
b
95% CI, 2.0-2.2); malignant cancer (OR, 2.3; 95% CI, 2.2-2.5); osteoporosis (OR, 8.3; 95% CI, 7.4-9.3); mood affective disorders (OR, 2.2; 95% CI, 2.2-2.3); chronic obstructive pulmonary disease (OR, 3.8; 95% CI, 3.34.4); chronic kidney disease (OR, 7.8; 95% CI, 7.2-8.3); and liver diseases (OR, 2.9; 95% CI, 2.8-3.0). Young adults with PoDs also had higher adjusted odds of multimorbidity compared with young adults without PoDs (OR, 3.8; 95% CI, 3.7-3.9). Descriptive characteristics and prevalence of noncommunicable diseases and multimorbidity for each PoD category are 4
Mayo Clin Proc.
n
presented in Table 3. All PoD categories tended to have higher prevalences of all noncommunicable diseases and multimorbidity compared with the young adults without PoDs, except for the similar prevalence of ischemic heart disease and cerebrovascular disease among young adults with PoDs of the genital organs. There was a considerable range in the prevalence of the noncommunicable diseases across the PoD categories: ischemic heart disease (0.3% [12 of 4035, genital organs category] to 4.5% [378 of 8337, circulatory system]); cerebrovascular disease (0.3% [13, genital organs] to 6.3% [528, circulatory system]); hypertensive and other cardiovascular disease (8.0% [324, genital organs] to 30.7% [932 of 3040, urinary system); type 2 diabetes (2.6% [106, genital organs] to 6.7% [635 of 9501, neurodevelopmental]); malignant cancer (1.3% [120, neurodevelopmental] to 3.5% [107, urinary system); osteoporosis (0.4% [10 of 2262, eye/ear/face/neck] to 2.0% [105 of 5154, nervous system); mood affective disorders (10.2% [348 of 3421, chromosomal abnormalities] to 33.9% [3221, neurodevelopmental]); chronic obstructive pulmonary disease (0.1% [4, genital organs] to 0.8% [42, nervous system]); chronic kidney disease (0.7% [28, genital organs] to 17.5% [531, urinary system]); liver disease (2.2% [89, genital organs] to 9.2% [267 of 2916, respiratory or digestive systems); and multimorbidity (4.5% [181, genital organs] to 23.1% [703, urinary system]). The results of the sensitivity analysis are also presented in Table 2 and revealed that the missing/unknown data for race/ethnicity had no effect on the results for each outcome for the main effect of PoD group. DISCUSSION The chief finding of this investigation is that young adults with PoDs had a substantially higher prevalence and adjusted odds of noncommunicable diseases across multiple biological systems, as well as higher multimorbidity. Furthermore, the prevalence of these noncommunicable diseases and multimorbidity was generally higher XXX 2019;nn(n):1-9
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
TABLE 2. Multivariable Logistic Regression for Noncommunicable Diseases and Multimorbidity Between Young Adults (18-40 Years) With PoDs and Those Without PoDs (Reference)a Adjustedb OR (95% CI)
Variable
Sensitivity Analysisc OR (95% CI)
Ischemic heart disease
4.7 (4.4-5.1)
4.7 (4.3-5.2)
Cerebrovascular disease
9.0 (8.4-9.6)
8.9 (8.3-9.7)
Hypertensive and other cardiovascular disease
2.4 (2.4-2.5)
2.5 (2.4-2.5)
Type 2 diabetes
2.1 (2.0-2.2)
2.1 (2.0-2.2)
Malignant cancer
2.3 (2.2-2.5)
2.4 (2.2-2.6)
Osteoporosis
8.3 (7.4-9.3)
8.6 (7.5-9.9)
Mood affective disorders
2.2 (2.2-2.3)
2.3 (2.2-2.4)
Chronic obstructive pulmonary disease
3.8 (3.3-4.4)
4.0 (3.3-4.7)
Chronic kidney disease
7.8 (7.2-8.3)
8.0 (7.4-8.7)
Liver disease
2.9 (2.8-3.0)
3.0 (2.8-3.2)
Multimorbidity (2 comorbidities)
3.8 (3.7-3.9)
3.9 (3.7-4.0)
OR ¼ odds ratio; PoD ¼ pediatric-onset disability. Main effect for group (PoDs, without PoDs) is presented after adjusting for age, sex, race/ethnicity, and education. c With complete and known data for ethnicity, total N¼1,566,859. a
b
for all PoD categories compared with young adults without PoDs. This is a critical finding because these noncommunicable diseases are major contributors to the global and national burden of disease and mortality1,2,27,28 and are typically associated with advanced aging. Further, many noncommunicable diseases can be prevented, delayed, or managed to reduce patient and caregiver burden. These findings highlight the need for clinical and health care policy efforts to improve awareness and access to quality health care, such as screening strategies, for these populations to allow for earlier detection of these noncommunicable diseases. Individuals with PoDs have complex health care needs and are at greater risk for development of noncommunicable diseases at earlier ages and in more severe forms compared with the general population.4,5,7,11-19 At present in the United States, care coordination, knowledge transfer, and health care access are inadequate to meet the high demands of health care needs among individuals with PoDs.23-26 This issue is especially problematic as these individuals transition into and throughout their adult years, leading to missed opportunities for early detection, prevention, and treatment strategies for common noncommunicable diseases. The findings in the current Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
investigation are therefore concerning. Although we found evidence of health disparities of hypertensive and other cardiovascular diseases, type 2 diabetes, malignant cancer, mood affective disorders, chronic obstructive pulmonary disease, and liver diseases (ORs, 2.1-3.8), young adults with PoDs had even higher odds of ischemic heart disease, cerebrovascular disease, osteoporosis, and chronic kidney disease (ORs 4.7) and a multimorbidity profile that was 3.8 times higher than that of young adults without PoDs. However, interpretation for osteoporosis should be made with caution because of the lower overall number of cases with this condition (2361 overall). Higher prevalence of noncommunicable diseases supports the notion of “early aging” (see reviews11,35,36) because these diseases should not be occurring this early in adulthood. Furthermore, it is likely that many of these diseases are not being screened for among individuals with PoDs at such a young age. Therefore, study findings are likely underrepresenting the true extent of their prevalence. An important finding of our study was that the elevated noncommunicable disease and multimorbidity profile were evident across all categories of PoDs (Table 3). This finding suggests that there was not a
https://doi.org/10.1016/j.mayocp.2019.07.010
5
6 TABLE 3. Descriptive Characteristics and Prevalence of Noncommunicable Diseases and Multimorbidity of Young Adults (18-40 Years), Stratified by the Category of Pediatric-Onset Disabilitiesa,b Musculo-skeletal system Variable Neurodevelop-mental (N¼12,139) (N¼9501)
Mayo Clin Proc.
Descriptive characteristics Age (y), mean SD Sex Female Male Race/ethnicity White Black Hispanic Asian Unknown/missing Education Less than high school High school diploma More than high school Unknown/missing
Respiratory or digestive systems Eye/ear/face/neck (N¼2916) (N¼2262)
Circulatory system (N¼8337)
Nervous system (N¼5154)
Genital organs (N¼4035)
Chromosomal abnormalities (N¼3421)
Urinary system (N¼3040)
29.96.5
29.36.5
30.95.6
30.36.2
31.36.1
30.76.3
29.36.5
29.86.8
26.56.4
58.5 (7100) 41.5 (5039)
35.4 (3367) 64.6 (6134)
57.2 (4766) 57.3 (2955) 81.5 (3288) 42.8 (3571) 42.7 (2199) 18.5 (747)
67.4 (2306) 32.6 (1115)
62.8 (1910) 37.2 (1130)
55.6 (1622) 44.4 (1294)
55.3 (1250) 44.7 (1012)
52.7 6.2 8.1 3.7 29.4
(6396) (748) (986) (446) (3563)
57.4 8.0 8.3 2.8 23.5
(5454) (763) (791) (265) (2228)
51.3 5.9 8.8 3.7 30.3
(4277) 54.1 (2787) 47.0 (1898) (494) 8.3 (426) 5.7 (229) (731) 9.2 (476) 10.3 (415) (306) 2.8 (142) 6.1 (247) (2529) 25.7 (1323) 30.9 (1246)
50.5 5.8 9.2 5.6 29.0
(1726) (197) (316) (191) (991)
49.1 5.8 10.5 4.4 30.2
(1494) (175) (319) (135) (917)
49.8 5.9 10.5 5.3 28.5
(1452) (172) (306) (154) (832)
48.4 7.0 10.0 5.8 28.9
(1094) (158) (225) (131) (654)
0.4 19.5 78.5 1.7
(46) (2368) (9525) (200)
0.5 23.6 72.9 3.0
(43) (2243) (6927) (288)
0.3 20.3 77.4 1.9
(27) 0.3 (17) 0.5 (20) (1696) 26.0 (1339) 20.0 (807) (6453) 70.7 (3645) 77.8 (3141) (161) 3.0 (153) 1.7 (67)
0.4 18.2 79.0 2.5
(12) (622) (2702) (85)
0.6 23.5 74.1 1.8
(17) (714) (2254) (55)
0.7 21.3 76.5 1.5
(19) (621) (2231) (45)
0.4 21.2 76.2 2.2
(10) (479) (1723) (50)
n
XXX 2019;nn(n):1-9
Noncommunicable diseases
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
a
0.8 (101) 0.9 (110) 10.7 (1296)
0.7 (67) 2.0 (190) 12.8 (1219)
4.5 (378) 1.1 (59) 6.3 (528) 3.7 (190) 20.5 (1706) 15.3 (788)
3.7 1.6 0.9 16.1 0.5 1.0 2.6 7.5
6.7 1.3 1.1 33.9 0.3 1.3 2.9 11.8
3.9 2.2 0.5 13.4 0.6 1.6 3.6 12.6
(448) (195) (108) (1950) (57) (121) (321) (907)
(635) (120) (102) (3221) (32) (121) (279) (1122)
(322) 5.0 (255) 2.6 (106) (179) 2.5 (128) 1.8 (71) (43) 2.0 (105) 0.5 (19) (1115) 20.0 (1028) 10.9 (440) (46) 0.8 (42) 0.1 (4) (133) 1.5 (76) 0.7 (28) (301) 2.9 (148) 2.2 (89) (1051) 12.0 (620) 4.5 (181)
Data are presented as percentage (No.) of patients unless indicated otherwise. Individuals may have more than one pediatric-onset disability and can be represented across multiple categories.
b
0.3 (12) 0.3 (13) 8.0 (324)
0.7 (25) 1.0 (33) 9.9 (337) 5.5 2.4 1.4 10.2 0.3 1.6 2.9 7.1
(189) (83) (48) (348) (11) (55) (99) (242)
1.4 (41) 1.7 (53) 30.7 (932)
1.3 (38) 0.9 (26) 13.5 (394)
5.3 3.5 0.7 16.3 0.2 17.5 8.8 23.1
4.2 3.4 0.7 15.0 0.7 2.2 9.2 11.5
(162) (107) (21) (494) (7) (531) (269) (703)
(121) (99) (19) (438) (19) (63) (267) (336)
0.5 (12) 0.8 (19) 9.0 (204) 3.9 2.1 0.4 10.9 0.1 0.8 2.4 5.8
(89) (48) (10) (246) (2) (17) (54) (131)
MAYO CLINIC PROCEEDINGS
Ischemic heart disease Cerebrovascular disease Hypertensive and other cardiovascular disease Type 2 diabetes Malignant cancer Osteoporosis Mood affective disorders Chronic obstructive pulmonary disease Chronic kidney disease Liver disease Multimorbidity (2 morbidities)
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
single PoD diagnosis or subset of PoDs (eg, neurodevelopmental conditions) that were driving the prevalence estimates. Although PoDs should not be assumed to be the same because most have very specific etiologies and complications, there are several overlapping risk factors among different types of PoDs that may impede healthful growth throughout the life span. These risk factors may be directly tied to the condition (eg, executive function impairments) or as a resulting sequela of these conditions (eg, pain, low social participation, low physical activity). Taken together, these factors may largely contribute to the accelerated development of noncommunicable diseases. However, individuals with certain PoDs may be at even greater risk for specific noncommunicable diseases. Consistent with our previous study in children and adolescents,37 we found that adults with neurodevelopmental PoDs had the highest prevalence of mood affective disorders compared with other PoD categories. Our findings also suggest that individuals with congenital or pediatric-acquired conditions of a particular biological system may be at higher risk of noncommunicable diseases of that same biological system. Specifically, PoDs of the circulatory system had the highest prevalence of ischemic heart disease and cerebrovascular disease. Further, PoDs of the urinary system had the highest prevalence of chronic kidney disease. Interestingly, the prevalence of osteoporosis was similar across all PoD categories, regardless of whether that PoD is associated with impairments of physical functioning. However, this finding may be biased more than other noncommunicable diseases investigated in the current study because screening for low bone mass in individuals younger than 40 years is not common clinical practice within the United States.38 Future studies are certainly needed to investigate mechanical and nonmechanical factors (eg, low participation, comorbidities) associated with the development of osteoporosis among individuals with PoDs. A major strength of this study is the large sample of young adults with PoDs. Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
Gathering data on clinical populations is challenging, and very little is known about health disparities among individuals with PoDs across the life span. Another major strength of this study is the comprehensive assessment of several noncommunicable diseases that are major contributors to the national and global burden of disease and mortality.1,2,27,28 These findings are robust and could help to inform new research examining health disparities and health care delivery for individuals with PoDs. One limitation of this study is that the overall sample may represent a healthier portion of the US population, especially for the sample representing adults with PoDs. All participants had to be enrolled in private insurance, either by purchasing their own insurance or being covered by an employer, their parents (up to 26 years of age), or a spouse. Employment and marriage rates are lower among adults with PoDs compared with adults without PoDs.39 Further, individuals with more severe forms or more medically complex PoDs may be more likely to be receiving federally subsidized health insurance. Another limitation is the unknown sensitivity of the Optum claims data to identify individuals with PoDs. However, because we are selecting across all diagnostic code positions on the claims for these specific PoDs, we believe that we have pretty strong specificity and sensitivity to detect these conditions on the claims. Considering the aforementioned limitations, prevalence estimates among our sample of young adults with PoDs may be conservative. Future studies are required to incorporate the segment of the population with PoDs from the Medicare database to determine the magnitude of these health disparities. A final limitation of this study is not being able to include individuals with conditions that can arise throughout the life span. It is not possible using claims-based data from a single year to determine if certain conditions developed in childhood or later in life, such as spinal cord injury, cancers, or mental health disorders. This issue is important because when these conditions arise during
https://doi.org/10.1016/j.mayocp.2019.07.010
7
MAYO CLINIC PROCEEDINGS
childhood, they are associated with adverse health outcomes in adulthood, such as mental health disorders.40,41 CONCLUSION Young adults with PoDs have an early onset of several noncommunicable diseases that represent major contributors to the global and national burden of disease and mortality. The early onset of noncommunicable diseases and multimorbidity is evident across all types of PoD categories and may play a role in the “early aging” phenomenon that has been described for some forms of PoDs. Future efforts are needed to improve screening strategies and identify risk factors and early patterns of noncommunicable disease development in order to mitigate the related burden among the populations with PoDs. ACKNOWLEDGMENTS The views expressed in this article are those of the authors and do not necessarily represent those of the National Institute on Disability, Independent Living, and Rehabilitation Research, the Administration for Community Living, and the Department of Health and Human Services. SUPPLEMENTAL ONLINE MATERIAL Supplemental material can be found online at http://www.mayoclinicproceedings.org. Supplemental material attached to journal articles has not been edited, and the authors take responsibility for the accuracy of all data. Abbreviations and Acronyms: OR = odds ratio; PoD = pediatric-onset disability Affiliations (Continued from the first page of this article.): Michigan, Ann Arbor (N.S.K.), Department of Surgery, University of Michigan, Ann Arbor (N.S.K.), Department of Emergency Medicine, University of Michigan, Ann Arbor (N.S.K.).
Grant Support: This work was supported in part by grant 90RTHF0001-01-00 from the National Institute on Disability, Independent Living, and Rehabilitation Research, a center within the Administration for Community Living, Department of Health and Human Services. Potential Competing Interests: The authors report no competing interests.
8
Mayo Clin Proc.
n
Correspondence: Address to Mark D. Peterson, PhD, MS, Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, 325 E Eisenhower Pkwy #300, Ann Arbor, MI 48108 (mdpeterz@med. umich.edu).
REFERENCES 1. Murray CJ, Vos T, Lozano R, et al. Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 19902010: a systematic analysis for the Global Burden of Disease Study 2010. Lancet. 2012;380(9859):2197-2223. 2. GBD 2016 Causes of Death Collaborators. Global, regional, and national age-sex specific mortality for 264 causes of death, 1980-2016: a systematic analysis for the Global Burden of Disease Study 2016 [published correction appears in Lancet. 2017;390(10106):e38]. Lancet. 2017; 390(10100):1151-1210. 3. World Health Organization. Global Action Plan for the Prevention and Control of Noncommunicable Diseases 2013-2020. Geneva, Switzerland: World Health Organization; 2013. 4. Whitney DG, Hurvitz EA, Ryan JM, et al. Noncommunicable disease and multimorbidity in young adults with cerebral palsy. Clin Epidemiol. 2018;10:511-519. 5. Whitney DG, Hurvitz EA, Devlin MJ, et al. Age trajectories of musculoskeletal morbidities in adults with cerebral palsy. Bone. 2018;114:285-291. 6. Cremer N, Hurvitz EA, Peterson MD. Multimorbidity in middleaged adults with cerebral palsy. Am J Med. 2017;130(6). 744.e9744.e15. 7. Hosking FJ, Carey IM, Shah SM, et al. Mortality among adults with intellectual disability in England: comparisons with the general population [published correction appears in Am J Public Health. 2016;106(12):e11-e12]. Am J Public Health. 2016; 106(8):1483-1490. 8. Boyle CA, Boulet S, Schieve LA, et al. Trends in the prevalence of developmental disabilities in US children, 1997-2008. Pediatrics. 2011;127(6):1034-1042. 9. Brooks JC, Strauss DJ, Shavelle RM, Tran LM, Rosenbloom L, Wu YW. Recent trends in cerebral palsy survival, Part I: period and cohort effects. Dev Med Child Neurol. 2014; 56(11):1059-1064. 10. Global Research on Developmental Disabilities Collaborators. Developmental disabilities among children younger than 5 years in 195 countries and territories, 1990-2016: a systematic analysis for the Global Burden of Disease Study 2016 [published correction appears in Lancet Glob Health. 2018;6(12): e1287]. Lancet Glob Health. 2018;6(10):e1100-e1121. 11. Verschuren O, Smorenburg ARP, Luiking Y, Bell K, Barber L, Peterson MD. Determinants of muscle preservation in individuals with cerebral palsy across the lifespan: a narrative review of the literature. J Cachexia Sarcopenia Muscle. 2018;9(3):453-464. 12. Charlson FJ, Baxter AJ, Dua T, Degenhardt L, Whiteford HA, Vos T. Excess mortality from mental, neurological, and substance use disorders in the Global Burden of Disease Study 2010. In: Patel V, Chisholm D, Dua T, Laxminarayan R, Medina-Mora ME, eds. Mental, Neurological, and Substance Use Disorders: Disease Control Priorities. 3rd ed., Vol 4. Washington, DC: World Bank; 2016:41-66. 13. Pikora TJ, Bourke J, Bathgate K, Foley KR, Lennox N, Leonard H. Health conditions and their impact among adolescents and young adults with Down syndrome. PLoS One. 2014;9(5): e96868. 14. Sipes M, Matson JL, Belva B, Turygin N, Kozlowski AM, Horovitz M. The relationship among side effects associated with anti-epileptic medications in those with intellectual disability. Res Dev Disabil. 2011;32(5):1646-1651. XXX 2019;nn(n):1-9
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
15. Hermans H, Evenhuis HM. Factors associated with depression and anxiety in older adults with intellectual disabilities: results of the Healthy Ageing And Intellectual Disabilities Study. Int J Geriatr Psychiatry. 2013;28(7):691-699. 16. Colver A, Rapp M, Eisemann N, et al. Self-reported quality of life of adolescents with cerebral palsy: a cross-sectional and longitudinal analysis. Lancet. 2015;385(9969):705-716. 17. Groh WJ. Arrhythmias in the muscular dystrophies. Heart Rhythm. 2012;9(11):1890-1895. 18. Van Der Slot WM, Nieuwenhuijsen C, Van Den BergEmons RJ, et al. Chronic pain, fatigue, and depressive symptoms in adults with spastic bilateral cerebral palsy [published correction appears in Dev Med Child Neurol. 2012;54(11):1064]. Dev Med Child Neurol. 2012;54(9):836-842. 19. Peterson MD, Kamdar N, Hurvitz EA. Age-related trends in cardiometabolic disease among adults with cerebral palsy. Dev Med Child Neurol. 2019;61(4):484-489. 20. Smith KJ, Peterson MD, O’Connell NE, et al. Risk of depression and anxiety in adults with cerebral palsy [published correction appears in JAMA Neurol. 2019;76(8):986]. JAMA Neurol. 2019; 76(3):294-300. 21. Peterson MD, Ryan JM, Hurvitz EA, Mahmoudi E. Chronic conditions in adults with cerebral palsy. JAMA. 2015;314(21): 2303-2305. 22. Ryan JM, Peterson MD, Matthews A, et al. Non-communicable disease among adults with cerebral palsy: a matched cohort study. Neurology. 2019;93(14):e1385-e1396. https://doi.org/10. 1212/WNL.0000000000008199. 23. Berry JG, Berry SD. Caring for patients with neurological impairment: conversations between a pediatrician and geriatrician. JAMA Pediatr. 2018;172(9):796. 24. Ahmedani BK, Hock RM. Health care access and treatment for children with co-morbid autism and psychiatric conditions. Soc Psychiatry Psychiatr Epidemiol. 2012;47(11):18071814. 25. Vohra R, Madhavan S, Sambamoorthi U, St Peter C. Access to services, quality of care, and family impact for children with autism, other developmental disabilities, and other mental health conditions. Autism. 2014;18(7):815-826. 26. Aisen ML, Kerkovich D, Mast J, et al. Cerebral palsy: clinical care and neurological rehabilitation. Lancet Neurol. 2011; 10(9):844-852. 27. Whiteford HA, Degenhardt L, Rehm J, et al. Global burden of disease attributable to mental and substance use disorders: findings from the Global Burden of Disease Study 2010. Lancet. 2013;382(9904):1575-1586. 28. GBD 2017 Disease and Injury Incidence and Prevalence Collaborators. Global, regional, and national incidence, prevalence, and years lived with disabilitity for 354 diseases and injuries for 195 countries and territories, 1990-2017: a systematic analysis for the Global Burden of Disease Study
Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
29.
30.
31.
32.
33. 34.
35.
36.
37.
38.
39. 40.
41.
2017 [published correction appears in Lancet. 2019; 393(10190):e44]. Lancet. 2018;392(10159):1789-1858. Wilkinson DA, Johnson K, Garton HJ, Muraszko KM, Maher CO. Trends in surgical treatment of Chiari malformation Type I in the United States. J Neurosurg Pediatr. 2017;19(2):208216. Borza T, Jacobs BL, Montgomery JS, et al. No differences in population-based readmissions after open and robotic-assisted radical cystectomy: implications for post-discharge care. Urology. 2017;104:77-83. Stem MS, Blachley TS, Shtein RM, Herman WH, Gardner TW, Stein JD. Impact of diagnosing diabetic complications on future hemoglobin A1c levels. J Diabetes Complications. 2016;30(2): 323-328. Quiñones AR, Markwardt S, Botoseneanu A. Multimorbidity combinations and disability in older adults. J Gerontol A Biol Sci Med Sci. 2016;71(6):823-830. Salive ME. Multimorbidity in older adults. Epidemiol Rev. 2013; 35:75-83. GBD 2017 DALYs and HALE Collaborators. Global, regional, and national disability-adjusted life-years (DALYs) for 359 diseases and injuries and healthy life expectancy (HALE) for 195 countries and territories, 1990-2017: a systematic analysis for the Global Burden of Disease Study 2017 [published correction appears in Lancet. 2019;393(10190):e44]. Lancet. 2018; 392(10159):1859-1922. Peterson MD, Gordon PM, Hurvitz EA. Chronic disease risk among adults with cerebral palsy: the role of premature sarcopoenia, obesity and sedentary behaviour. Obes Rev. 2013;14(2): 171-182. Peterson MD, Gordon PM, Hurvitz EA, Burant CF. Secondary muscle pathology and metabolic dysregulation in adults with cerebral palsy. Am J Physiol Endocrinol Metab. 2012;303(9): E1085-E1093. Whitney DG, Shapiro DN, Warschausky SA, Hurvitz EA, Peterson MD. The contribution of neurological disorders to the national prevalence of depression and anxiety problems among children and adolescents. Ann Epidemiol. 2019;29:81-84.e2. US Preventive Services Task Force. Screening for osteoporosis to prevent fractures: US Preventive Services Task Force recommendation statement. JAMA. 2018;319(24):2521-2531. Tumin D. Marriage trends among Americans with childhoodonset disabilities, 1997-2013. Disabil Health J. 2016;9(4):713-718. Pine DS, Cohen P, Gurley D, Brook J, Ma Y. The risk for earlyadulthood anxiety and depressive disorders in adolescents with anxiety and depressive disorders. Arch Gen Psychiatry. 1998; 55(1):56-64. Jacobson NC, Newman MG. Anxiety and depression as bidirectional risk factors for one another: a meta-analysis of longitudinal studies. Psychol Bull. 2017;143(11):1155-1200.
https://doi.org/10.1016/j.mayocp.2019.07.010
9
Early-Onset Noncommunicable Disease and Multimorbidity Among Adults With Pediatric-Onset Disabilities Daniel G. Whitney, PhD; Rachael T. Whitney, PhD; Neil S. Kamdar, MA; Edward A. Hurvitz, MD; and Mark D. Peterson, PhD, MS Abstract Objective: To determine the prevalence of major noncommunicable diseases among young adults with pediatric-onset disabilities (PoDs) compared with young adults without PoDs. Patients and Methods: Data were obtained from the Optum Clinformatics Data Mart, a de-identified nationwide claims database of beneficiaries from a single private payer in the United States. Beneficiaries were included if they were 18 to 40 years old and had an International Classification of Diseases, Ninth Revision, Clinical Modification diagnostic code for a PoD known to originate in childhood. Diagnostic codes were used to identify high-burden noncommunicable diseases: ischemic heart disease, cerebrovascular disease, hypertensive and other cardiovascular disease, type 2 diabetes, malignant cancer, osteoporosis, mood affective disorders, chronic obstructive pulmonary disease, chronic kidney disease, and liver disease. The prevalence of noncommunicable diseases and multimorbidity (2 diseases) was compared between adults with (N¼47,077) and without (N¼2,180,250) PoDs, before and after adjusting for sociodemographic characteristics. This study was conducted between July 1, 2018, and February 1, 2019. Results: Adults with PoDs had higher prevalences and adjusted odds of all noncommunicable diseases (odds ratio, 2.1-9.0; all P<.05) and multimorbidity (odds ratio, 3.8; 95% CI, 3.7-3.9) compared with adults without PoDs. After stratifying by the type of PoD (eg, musculoskeletal, circulatory), all PoD categories had higher prevalence of all noncommunicable diseases and multimorbidity compared with young adults without PoDs, except for ischemic heart disease and cerebrovascular disease among adults with PoDs of the genital organs. Conclusion: Young adults with PoDs have an early onset of several noncommunicable diseases that represent major contributors to the global and national burden of disease and mortality. ª 2019 Mayo Foundation for Medical Education and Research
T
here has been a considerable shift in the global burden disease, with the prevalence of noncommunicable diseases now outranking injuries and communicable, maternal, neonatal, and nutritional disorders combined.1 In the United States, chronic noncommunicable diseases represent the primary causes of death2 and economic burden, with related health care costs slated to reach $7 trillion by 2030.3 Importantly, modifiable risk factors (eg, diet, physical activity, substance use) are associated with the development of many noncommunicable diseases.3 Moreover, there are segments of the population that
n
Mayo Clin Proc. 2019;nn(n):1-9
experience an elevated burden of disease throughout the life span, as well as those with disparities in access and services (eg, individuals with disabilities).4-7 Therefore, identifying at-risk populations is necessary to develop targeted strategies to prevent, treat, and manage the burden of noncommunicable diseases. In particular, pediatric-onset disabilities (PoDs) represent a group of congenital or acquired chronic conditions that originate during pregnancy or in childhood and consist of impairments in behavioral, intellectual, and/ or physical functioning or are associated with abnormal growth/development or
Mayo Clin Proc. n XXX 2019;nn(n):1-9 n https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org n ª 2019 Mayo Foundation for Medical Education and Research
From the Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor (D.G.W., E.A.H., M.D.P.), Department of Kinesiology and Applied Physiology, University of Delaware, Newark (R.T.W.), Institute for Healthcare Policy and Innovation, University of Michigan, Ann Arbor (N.S.K., M.D.P.), Department of Obstetrics and Gynecology, University of Affiliations continued at the end of this article.
1
MAYO CLINIC PROCEEDINGS
physiologic processes. The population of adults with PoDs is projected to expand over the coming decades because of increases in childhood survival rates and prevalence.8-10 This trend is a major public health issue because individuals with PoDs have complex health care needs and are susceptible to an accelerated development of noncommunicable diseases and early mortality, which places a high strain on health care resources.4,5,7,11-22 Further, access to quality health care is inadequate for many individuals with PoDs, which coincides with a lack of clinical knowledge of life-course health development.23-26 The general lack of resources allocated to screening for and treating noncommunicable diseases among adults with PoDs could lead to a disproportionate increase in disease burden and health care costs in the coming decades that may otherwise be preventable. Therefore, a comprehensive understanding of the noncommunicable disease profiles among young adults with PoDs is urgently needed. This knowledge could inform early clinical decision-making processes and strategies to prevent or reduce the burden of noncommunicable diseases among these patients. Accordingly, the objective of this study was to compare the prevalence of major noncommunicable diseases that contribute to a substantial national burden of disease and mortality1,2,27,28 among young adults aged 18 to 40 years with and without PoDs. We hypothesized that young adults with PoDs would have a higher prevalence of all noncommunicable diseases compared with young adults without PoDs. PATIENTS AND METHODS Data Source Data were obtained from the Clinformatics Data Mart Database (Optum, Inc), which is a nationwide, de-identified insurance claims database of 16 million beneficiaries from a single private payer in the year 2016. This administrative database houses information of beneficiaries with both medical and pharmacy coverage. This database has been 2
Mayo Clin Proc.
n
leveraged to examine longitudinal trends of noncommunicable diseases among adults with cerebral palsy,19 a type of PoD, and other conditions and their subsequent complications.29-31 Data are de-identified, and the University of Michigan Institutional Review Board approved this study as nonregulated. This study was conducted between July 1, 2018, and February 1, 2019. Sample Selection We acquired access to the administrative claims data from the most recent available calendar year, 2016, for the analysis. Individuals between 18 and 40 years of age who had 12 months of continuous enrollment were included. All PoD conditions and noncommunicable diseases were identified using the International Classification of Diseases, Tenth Revision, Clinical Modification codes and are presented in the Supplemental Table (available online at http://www. mayoclinicproceedings.org). Using administrative claims, it is not possible to determine if some conditions developed in childhood or in adulthood (eg, mental health disorders, spinal cord injury) because of the exceptionally long enrollment time frames to track children from birth and the discontinuities in enrollment. Therefore, conditions were included in the group representing PoDs if there were known childhood origins (eg, congenital disorders) and were grouped into the following categories: (1) PoDs of the musculoskeletal system, (2) neurodevelopmental PoDs, (3) PoDs of the circulatory system, (4) PoDs of the nervous system, (5) PoDs of the genital organs, (6) other chromosomal abnormalities, (7) PoDs of the urinary system, (8) PoDs of the respiratory system and digestive system, and (9) pediatric-onset malformations of the eye, ear, face, and neck. Of the 10.3 million adults aged 18 years and older with 12 months of continuous enrollment in 2016, 1.6 million members (15.5%) did not have an inpatient, outpatient, or emergency department visit reporting a diagnostic code while enrolled in 2016. We were therefore unable to identify any XXX 2019;nn(n):1-9
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
diagnoses and excluded these cases to eliminate risk of bias. Another 991 (0.01%) individuals were excluded because of missing sex data. Finally, 6.5 million members were excluded because they were over the age of 40 years. The final sample comprised 2,227,327 individuals, including 47,077 adults with at least one PoD. Noncommunicable Diseases Noncommunicable diseases and their variations (eg, severity) were selected with guidance from the literature on disease, disability, and mortality among 1,2,4,5,27,28,32-34 adults and are represented as categories across multiple biological systems including (1) ischemic heart disease (eg, atherosclerotic heart disease), (2) cerebrovascular disease (eg, cerebral infarction), (3) hypertensive and other cardiovascular disease (eg, hypertension, heart failure), (4) type 2 diabetes mellitus, (5) malignant cancer, (6) osteoporosis, (7) mood affective disorders (eg, major depressive disorder), (8) chronic obstructive pulmonary disease (eg, emphysema), (9) chronic kidney disease (eg, kidney disease stages 1-4), and (10) liver disease (eg, cirrhosis). Multimorbidity was defined as the presence of at least 2 of the aforementioned noncommunicable diseases on at least 1 or more service dates during the beneficiaries’ full 12month enrollment. Covariates Covariates that were available in the administrative claims included sociodemographic variables: age, sex, race/ethnicity, and education level. Statistical Analyses Age was summarized as a continuous variable using mean SD, and all other variables were summarized as percentage (frequency) for those with and without PoDs, with subsequent stratification for each PoD category (eg, nervous system, musculoskeletal system). We examined each of the 10 noncommunicable disease categories and multimorbidity as outcomes, and the primary exposure Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
variable was an indicator for PoD, with the referent as without PoDs. We performed multivariable logistic regression analyses for each outcome, adjusting for age, sex, race/ ethnicity, and education level. The main effect of PoD group was interpreted. Because of the extent of missing/unknown for race/ ethnicity, we conducted a sensitivity analysis that included individuals with complete and known data for race/ethnicity to determine if it biased the main effect of PoD group for all outcomes. All analyses were performed using SAS statistical software, version 9.4 (SAS Institute). RESULTS Descriptive characteristics and prevalence of noncommunicable diseases and multimorbidity for young adults with (N¼47,077) and without (N¼2,180,250) PoDs are presented in Table 1. Young adults with PoDs had a higher prevalence of all noncommunicable diseases and multimorbidity compared with young adults without PoDs: ischemic heart disease (1.4% [663] vs 0.3% [7160]); cerebrovascular disease (2.1% [1006] vs 0.3% [5521]); hypertensive and other cardiovascular disease (14.0% [6569] vs 7.1% [155,181]); type 2 diabetes (4.5% [2108] vs 2.4% [51,752]); malignant cancer (2.0% [941] vs 0.9% [19,615]); osteoporosis (0.7% [346] vs 0.1% [2015]); mood affective disorders (18.1% [8525] vs 8.9% [193,908]); chronic obstructive pulmonary disease (0.4% [185] vs 0.1% [2334]); chronic kidney disease (2.1% [974] vs 0.3% [6391]); liver disease (3.5% [1652] vs 1.3% [29,070]); and multimorbidity (10.0% [4716] vs 3.1% [68.539]). The adjusted odds ratios (ORs; adjusting for age, sex, race/ethnicity, and education level) of noncommunicable diseases and multimorbidity are presented in Table 2. Young adults with PoDs had higher adjusted odds of all noncommunicable diseases compared with young adults without PoDs (all P<.05): ischemic heart disease (OR, 4.7; 95% CI, 4.4-5.1); cerebrovascular disease (OR, 9.0; 95% CI, 8.4-9.6); hypertensive and other cardiovascular disease (OR, 2.4; 95% CI, 2.4-2.5); type 2 diabetes (OR, 2.1;
https://doi.org/10.1016/j.mayocp.2019.07.010
3
MAYO CLINIC PROCEEDINGS
TABLE 1. Descriptive Characteristics and Prevalence of Noncommunicable Diseases and Multimorbidity in 2,227,327 Young Adults (18-40 Years) With and Without Pediatric-Onset Disabilitiesa,b Variable Descriptive characteristics Age (y) Sex Female Male Ethnicity White Black Hispanic Asian Unknown/missing Education Less than high school High school diploma More than high school Unknown/missing
With PoD (N¼47,077; 2.1%)
Without PoD (N¼2,180,250; 97.9%)
29.56.6
30.26.3
56.4 (26,544) 43.6 (20,533)
55.3 (1,205,121) 44.7 (975,129)
52.0 6.6 9.0 4.0 28.4
(24,460) (3119) (4227) (1888) (13,383)
47.8 6.8 10.4 5.3 29.7
(1,042,346) (147,412) (227,461) (115,946) (647,085)
0.4 21.4 76.1 2.1
(195) (10,052) (35,843) (987)
0.5 22.0 75.8 1.7
(11,479) (479,884) (1,652,814) (36,073)
Noncommunicable diseases Ischemic heart disease Cerebrovascular disease Hypertensive and other cardiovascular disease Type 2 diabetes Malignant cancer Osteoporosis Mood affective disorders Chronic obstructive pulmonary disease Chronic kidney disease Liver disease Multimorbidity (2 comorbidities) a
1.4 (663) 2.1 (1006) 14.0 (6569)
0.3 (7160) 0.3 (5521) 7.1 (155,181)
4.5 2.0 0.7 18.1 0.4
2.4 0.9 0.1 8.9 0.1
(2108) (941) (346) (8525) (185)
2.1 (974) 3.5 (1652) 10.0 (4716)
(51,752) (19,615) (2015) (193,908) (2334)
0.3 (6391) 1.3 (29,070) 3.1 (68,539)
PoD ¼ pediatric-onset disability. Data are presented as mean SD or percentage (No.) of patients.
b
95% CI, 2.0-2.2); malignant cancer (OR, 2.3; 95% CI, 2.2-2.5); osteoporosis (OR, 8.3; 95% CI, 7.4-9.3); mood affective disorders (OR, 2.2; 95% CI, 2.2-2.3); chronic obstructive pulmonary disease (OR, 3.8; 95% CI, 3.34.4); chronic kidney disease (OR, 7.8; 95% CI, 7.2-8.3); and liver diseases (OR, 2.9; 95% CI, 2.8-3.0). Young adults with PoDs also had higher adjusted odds of multimorbidity compared with young adults without PoDs (OR, 3.8; 95% CI, 3.7-3.9). Descriptive characteristics and prevalence of noncommunicable diseases and multimorbidity for each PoD category are 4
Mayo Clin Proc.
n
presented in Table 3. All PoD categories tended to have higher prevalences of all noncommunicable diseases and multimorbidity compared with the young adults without PoDs, except for the similar prevalence of ischemic heart disease and cerebrovascular disease among young adults with PoDs of the genital organs. There was a considerable range in the prevalence of the noncommunicable diseases across the PoD categories: ischemic heart disease (0.3% [12 of 4035, genital organs category] to 4.5% [378 of 8337, circulatory system]); cerebrovascular disease (0.3% [13, genital organs] to 6.3% [528, circulatory system]); hypertensive and other cardiovascular disease (8.0% [324, genital organs] to 30.7% [932 of 3040, urinary system); type 2 diabetes (2.6% [106, genital organs] to 6.7% [635 of 9501, neurodevelopmental]); malignant cancer (1.3% [120, neurodevelopmental] to 3.5% [107, urinary system); osteoporosis (0.4% [10 of 2262, eye/ear/face/neck] to 2.0% [105 of 5154, nervous system); mood affective disorders (10.2% [348 of 3421, chromosomal abnormalities] to 33.9% [3221, neurodevelopmental]); chronic obstructive pulmonary disease (0.1% [4, genital organs] to 0.8% [42, nervous system]); chronic kidney disease (0.7% [28, genital organs] to 17.5% [531, urinary system]); liver disease (2.2% [89, genital organs] to 9.2% [267 of 2916, respiratory or digestive systems); and multimorbidity (4.5% [181, genital organs] to 23.1% [703, urinary system]). The results of the sensitivity analysis are also presented in Table 2 and revealed that the missing/unknown data for race/ethnicity had no effect on the results for each outcome for the main effect of PoD group. DISCUSSION The chief finding of this investigation is that young adults with PoDs had a substantially higher prevalence and adjusted odds of noncommunicable diseases across multiple biological systems, as well as higher multimorbidity. Furthermore, the prevalence of these noncommunicable diseases and multimorbidity was generally higher XXX 2019;nn(n):1-9
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
TABLE 2. Multivariable Logistic Regression for Noncommunicable Diseases and Multimorbidity Between Young Adults (18-40 Years) With PoDs and Those Without PoDs (Reference)a Adjustedb OR (95% CI)
Variable
Sensitivity Analysisc OR (95% CI)
Ischemic heart disease
4.7 (4.4-5.1)
4.7 (4.3-5.2)
Cerebrovascular disease
9.0 (8.4-9.6)
8.9 (8.3-9.7)
Hypertensive and other cardiovascular disease
2.4 (2.4-2.5)
2.5 (2.4-2.5)
Type 2 diabetes
2.1 (2.0-2.2)
2.1 (2.0-2.2)
Malignant cancer
2.3 (2.2-2.5)
2.4 (2.2-2.6)
Osteoporosis
8.3 (7.4-9.3)
8.6 (7.5-9.9)
Mood affective disorders
2.2 (2.2-2.3)
2.3 (2.2-2.4)
Chronic obstructive pulmonary disease
3.8 (3.3-4.4)
4.0 (3.3-4.7)
Chronic kidney disease
7.8 (7.2-8.3)
8.0 (7.4-8.7)
Liver disease
2.9 (2.8-3.0)
3.0 (2.8-3.2)
Multimorbidity (2 comorbidities)
3.8 (3.7-3.9)
3.9 (3.7-4.0)
OR ¼ odds ratio; PoD ¼ pediatric-onset disability. Main effect for group (PoDs, without PoDs) is presented after adjusting for age, sex, race/ethnicity, and education. c With complete and known data for ethnicity, total N¼1,566,859. a
b
for all PoD categories compared with young adults without PoDs. This is a critical finding because these noncommunicable diseases are major contributors to the global and national burden of disease and mortality1,2,27,28 and are typically associated with advanced aging. Further, many noncommunicable diseases can be prevented, delayed, or managed to reduce patient and caregiver burden. These findings highlight the need for clinical and health care policy efforts to improve awareness and access to quality health care, such as screening strategies, for these populations to allow for earlier detection of these noncommunicable diseases. Individuals with PoDs have complex health care needs and are at greater risk for development of noncommunicable diseases at earlier ages and in more severe forms compared with the general population.4,5,7,11-19 At present in the United States, care coordination, knowledge transfer, and health care access are inadequate to meet the high demands of health care needs among individuals with PoDs.23-26 This issue is especially problematic as these individuals transition into and throughout their adult years, leading to missed opportunities for early detection, prevention, and treatment strategies for common noncommunicable diseases. The findings in the current Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
investigation are therefore concerning. Although we found evidence of health disparities of hypertensive and other cardiovascular diseases, type 2 diabetes, malignant cancer, mood affective disorders, chronic obstructive pulmonary disease, and liver diseases (ORs, 2.1-3.8), young adults with PoDs had even higher odds of ischemic heart disease, cerebrovascular disease, osteoporosis, and chronic kidney disease (ORs 4.7) and a multimorbidity profile that was 3.8 times higher than that of young adults without PoDs. However, interpretation for osteoporosis should be made with caution because of the lower overall number of cases with this condition (2361 overall). Higher prevalence of noncommunicable diseases supports the notion of “early aging” (see reviews11,35,36) because these diseases should not be occurring this early in adulthood. Furthermore, it is likely that many of these diseases are not being screened for among individuals with PoDs at such a young age. Therefore, study findings are likely underrepresenting the true extent of their prevalence. An important finding of our study was that the elevated noncommunicable disease and multimorbidity profile were evident across all categories of PoDs (Table 3). This finding suggests that there was not a
https://doi.org/10.1016/j.mayocp.2019.07.010
5
6 TABLE 3. Descriptive Characteristics and Prevalence of Noncommunicable Diseases and Multimorbidity of Young Adults (18-40 Years), Stratified by the Category of Pediatric-Onset Disabilitiesa,b Musculo-skeletal system Variable Neurodevelop-mental (N¼12,139) (N¼9501)
Mayo Clin Proc.
Descriptive characteristics Age (y), mean SD Sex Female Male Race/ethnicity White Black Hispanic Asian Unknown/missing Education Less than high school High school diploma More than high school Unknown/missing
Respiratory or digestive systems Eye/ear/face/neck (N¼2916) (N¼2262)
Circulatory system (N¼8337)
Nervous system (N¼5154)
Genital organs (N¼4035)
Chromosomal abnormalities (N¼3421)
Urinary system (N¼3040)
29.96.5
29.36.5
30.95.6
30.36.2
31.36.1
30.76.3
29.36.5
29.86.8
26.56.4
58.5 (7100) 41.5 (5039)
35.4 (3367) 64.6 (6134)
57.2 (4766) 57.3 (2955) 81.5 (3288) 42.8 (3571) 42.7 (2199) 18.5 (747)
67.4 (2306) 32.6 (1115)
62.8 (1910) 37.2 (1130)
55.6 (1622) 44.4 (1294)
55.3 (1250) 44.7 (1012)
52.7 6.2 8.1 3.7 29.4
(6396) (748) (986) (446) (3563)
57.4 8.0 8.3 2.8 23.5
(5454) (763) (791) (265) (2228)
51.3 5.9 8.8 3.7 30.3
(4277) 54.1 (2787) 47.0 (1898) (494) 8.3 (426) 5.7 (229) (731) 9.2 (476) 10.3 (415) (306) 2.8 (142) 6.1 (247) (2529) 25.7 (1323) 30.9 (1246)
50.5 5.8 9.2 5.6 29.0
(1726) (197) (316) (191) (991)
49.1 5.8 10.5 4.4 30.2
(1494) (175) (319) (135) (917)
49.8 5.9 10.5 5.3 28.5
(1452) (172) (306) (154) (832)
48.4 7.0 10.0 5.8 28.9
(1094) (158) (225) (131) (654)
0.4 19.5 78.5 1.7
(46) (2368) (9525) (200)
0.5 23.6 72.9 3.0
(43) (2243) (6927) (288)
0.3 20.3 77.4 1.9
(27) 0.3 (17) 0.5 (20) (1696) 26.0 (1339) 20.0 (807) (6453) 70.7 (3645) 77.8 (3141) (161) 3.0 (153) 1.7 (67)
0.4 18.2 79.0 2.5
(12) (622) (2702) (85)
0.6 23.5 74.1 1.8
(17) (714) (2254) (55)
0.7 21.3 76.5 1.5
(19) (621) (2231) (45)
0.4 21.2 76.2 2.2
(10) (479) (1723) (50)
n
XXX 2019;nn(n):1-9
Noncommunicable diseases
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
a
0.8 (101) 0.9 (110) 10.7 (1296)
0.7 (67) 2.0 (190) 12.8 (1219)
4.5 (378) 1.1 (59) 6.3 (528) 3.7 (190) 20.5 (1706) 15.3 (788)
3.7 1.6 0.9 16.1 0.5 1.0 2.6 7.5
6.7 1.3 1.1 33.9 0.3 1.3 2.9 11.8
3.9 2.2 0.5 13.4 0.6 1.6 3.6 12.6
(448) (195) (108) (1950) (57) (121) (321) (907)
(635) (120) (102) (3221) (32) (121) (279) (1122)
(322) 5.0 (255) 2.6 (106) (179) 2.5 (128) 1.8 (71) (43) 2.0 (105) 0.5 (19) (1115) 20.0 (1028) 10.9 (440) (46) 0.8 (42) 0.1 (4) (133) 1.5 (76) 0.7 (28) (301) 2.9 (148) 2.2 (89) (1051) 12.0 (620) 4.5 (181)
Data are presented as percentage (No.) of patients unless indicated otherwise. Individuals may have more than one pediatric-onset disability and can be represented across multiple categories.
b
0.3 (12) 0.3 (13) 8.0 (324)
0.7 (25) 1.0 (33) 9.9 (337) 5.5 2.4 1.4 10.2 0.3 1.6 2.9 7.1
(189) (83) (48) (348) (11) (55) (99) (242)
1.4 (41) 1.7 (53) 30.7 (932)
1.3 (38) 0.9 (26) 13.5 (394)
5.3 3.5 0.7 16.3 0.2 17.5 8.8 23.1
4.2 3.4 0.7 15.0 0.7 2.2 9.2 11.5
(162) (107) (21) (494) (7) (531) (269) (703)
(121) (99) (19) (438) (19) (63) (267) (336)
0.5 (12) 0.8 (19) 9.0 (204) 3.9 2.1 0.4 10.9 0.1 0.8 2.4 5.8
(89) (48) (10) (246) (2) (17) (54) (131)
MAYO CLINIC PROCEEDINGS
Ischemic heart disease Cerebrovascular disease Hypertensive and other cardiovascular disease Type 2 diabetes Malignant cancer Osteoporosis Mood affective disorders Chronic obstructive pulmonary disease Chronic kidney disease Liver disease Multimorbidity (2 morbidities)
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
single PoD diagnosis or subset of PoDs (eg, neurodevelopmental conditions) that were driving the prevalence estimates. Although PoDs should not be assumed to be the same because most have very specific etiologies and complications, there are several overlapping risk factors among different types of PoDs that may impede healthful growth throughout the life span. These risk factors may be directly tied to the condition (eg, executive function impairments) or as a resulting sequela of these conditions (eg, pain, low social participation, low physical activity). Taken together, these factors may largely contribute to the accelerated development of noncommunicable diseases. However, individuals with certain PoDs may be at even greater risk for specific noncommunicable diseases. Consistent with our previous study in children and adolescents,37 we found that adults with neurodevelopmental PoDs had the highest prevalence of mood affective disorders compared with other PoD categories. Our findings also suggest that individuals with congenital or pediatric-acquired conditions of a particular biological system may be at higher risk of noncommunicable diseases of that same biological system. Specifically, PoDs of the circulatory system had the highest prevalence of ischemic heart disease and cerebrovascular disease. Further, PoDs of the urinary system had the highest prevalence of chronic kidney disease. Interestingly, the prevalence of osteoporosis was similar across all PoD categories, regardless of whether that PoD is associated with impairments of physical functioning. However, this finding may be biased more than other noncommunicable diseases investigated in the current study because screening for low bone mass in individuals younger than 40 years is not common clinical practice within the United States.38 Future studies are certainly needed to investigate mechanical and nonmechanical factors (eg, low participation, comorbidities) associated with the development of osteoporosis among individuals with PoDs. A major strength of this study is the large sample of young adults with PoDs. Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
Gathering data on clinical populations is challenging, and very little is known about health disparities among individuals with PoDs across the life span. Another major strength of this study is the comprehensive assessment of several noncommunicable diseases that are major contributors to the national and global burden of disease and mortality.1,2,27,28 These findings are robust and could help to inform new research examining health disparities and health care delivery for individuals with PoDs. One limitation of this study is that the overall sample may represent a healthier portion of the US population, especially for the sample representing adults with PoDs. All participants had to be enrolled in private insurance, either by purchasing their own insurance or being covered by an employer, their parents (up to 26 years of age), or a spouse. Employment and marriage rates are lower among adults with PoDs compared with adults without PoDs.39 Further, individuals with more severe forms or more medically complex PoDs may be more likely to be receiving federally subsidized health insurance. Another limitation is the unknown sensitivity of the Optum claims data to identify individuals with PoDs. However, because we are selecting across all diagnostic code positions on the claims for these specific PoDs, we believe that we have pretty strong specificity and sensitivity to detect these conditions on the claims. Considering the aforementioned limitations, prevalence estimates among our sample of young adults with PoDs may be conservative. Future studies are required to incorporate the segment of the population with PoDs from the Medicare database to determine the magnitude of these health disparities. A final limitation of this study is not being able to include individuals with conditions that can arise throughout the life span. It is not possible using claims-based data from a single year to determine if certain conditions developed in childhood or later in life, such as spinal cord injury, cancers, or mental health disorders. This issue is important because when these conditions arise during
https://doi.org/10.1016/j.mayocp.2019.07.010
7
MAYO CLINIC PROCEEDINGS
childhood, they are associated with adverse health outcomes in adulthood, such as mental health disorders.40,41 CONCLUSION Young adults with PoDs have an early onset of several noncommunicable diseases that represent major contributors to the global and national burden of disease and mortality. The early onset of noncommunicable diseases and multimorbidity is evident across all types of PoD categories and may play a role in the “early aging” phenomenon that has been described for some forms of PoDs. Future efforts are needed to improve screening strategies and identify risk factors and early patterns of noncommunicable disease development in order to mitigate the related burden among the populations with PoDs. ACKNOWLEDGMENTS The views expressed in this article are those of the authors and do not necessarily represent those of the National Institute on Disability, Independent Living, and Rehabilitation Research, the Administration for Community Living, and the Department of Health and Human Services. SUPPLEMENTAL ONLINE MATERIAL Supplemental material can be found online at http://www.mayoclinicproceedings.org. Supplemental material attached to journal articles has not been edited, and the authors take responsibility for the accuracy of all data. Abbreviations and Acronyms: OR = odds ratio; PoD = pediatric-onset disability Affiliations (Continued from the first page of this article.): Michigan, Ann Arbor (N.S.K.), Department of Surgery, University of Michigan, Ann Arbor (N.S.K.), Department of Emergency Medicine, University of Michigan, Ann Arbor (N.S.K.).
Grant Support: This work was supported in part by grant 90RTHF0001-01-00 from the National Institute on Disability, Independent Living, and Rehabilitation Research, a center within the Administration for Community Living, Department of Health and Human Services. Potential Competing Interests: The authors report no competing interests.
8
Mayo Clin Proc.
n
Correspondence: Address to Mark D. Peterson, PhD, MS, Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, 325 E Eisenhower Pkwy #300, Ann Arbor, MI 48108 (mdpeterz@med. umich.edu).
REFERENCES 1. Murray CJ, Vos T, Lozano R, et al. Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 19902010: a systematic analysis for the Global Burden of Disease Study 2010. Lancet. 2012;380(9859):2197-2223. 2. GBD 2016 Causes of Death Collaborators. Global, regional, and national age-sex specific mortality for 264 causes of death, 1980-2016: a systematic analysis for the Global Burden of Disease Study 2016 [published correction appears in Lancet. 2017;390(10106):e38]. Lancet. 2017; 390(10100):1151-1210. 3. World Health Organization. Global Action Plan for the Prevention and Control of Noncommunicable Diseases 2013-2020. Geneva, Switzerland: World Health Organization; 2013. 4. Whitney DG, Hurvitz EA, Ryan JM, et al. Noncommunicable disease and multimorbidity in young adults with cerebral palsy. Clin Epidemiol. 2018;10:511-519. 5. Whitney DG, Hurvitz EA, Devlin MJ, et al. Age trajectories of musculoskeletal morbidities in adults with cerebral palsy. Bone. 2018;114:285-291. 6. Cremer N, Hurvitz EA, Peterson MD. Multimorbidity in middleaged adults with cerebral palsy. Am J Med. 2017;130(6). 744.e9744.e15. 7. Hosking FJ, Carey IM, Shah SM, et al. Mortality among adults with intellectual disability in England: comparisons with the general population [published correction appears in Am J Public Health. 2016;106(12):e11-e12]. Am J Public Health. 2016; 106(8):1483-1490. 8. Boyle CA, Boulet S, Schieve LA, et al. Trends in the prevalence of developmental disabilities in US children, 1997-2008. Pediatrics. 2011;127(6):1034-1042. 9. Brooks JC, Strauss DJ, Shavelle RM, Tran LM, Rosenbloom L, Wu YW. Recent trends in cerebral palsy survival, Part I: period and cohort effects. Dev Med Child Neurol. 2014; 56(11):1059-1064. 10. Global Research on Developmental Disabilities Collaborators. Developmental disabilities among children younger than 5 years in 195 countries and territories, 1990-2016: a systematic analysis for the Global Burden of Disease Study 2016 [published correction appears in Lancet Glob Health. 2018;6(12): e1287]. Lancet Glob Health. 2018;6(10):e1100-e1121. 11. Verschuren O, Smorenburg ARP, Luiking Y, Bell K, Barber L, Peterson MD. Determinants of muscle preservation in individuals with cerebral palsy across the lifespan: a narrative review of the literature. J Cachexia Sarcopenia Muscle. 2018;9(3):453-464. 12. Charlson FJ, Baxter AJ, Dua T, Degenhardt L, Whiteford HA, Vos T. Excess mortality from mental, neurological, and substance use disorders in the Global Burden of Disease Study 2010. In: Patel V, Chisholm D, Dua T, Laxminarayan R, Medina-Mora ME, eds. Mental, Neurological, and Substance Use Disorders: Disease Control Priorities. 3rd ed., Vol 4. Washington, DC: World Bank; 2016:41-66. 13. Pikora TJ, Bourke J, Bathgate K, Foley KR, Lennox N, Leonard H. Health conditions and their impact among adolescents and young adults with Down syndrome. PLoS One. 2014;9(5): e96868. 14. Sipes M, Matson JL, Belva B, Turygin N, Kozlowski AM, Horovitz M. The relationship among side effects associated with anti-epileptic medications in those with intellectual disability. Res Dev Disabil. 2011;32(5):1646-1651. XXX 2019;nn(n):1-9
n
https://doi.org/10.1016/j.mayocp.2019.07.010 www.mayoclinicproceedings.org
CHRONIC DISEASE IN PEDIATRIC-ONSET DISABILITIES
15. Hermans H, Evenhuis HM. Factors associated with depression and anxiety in older adults with intellectual disabilities: results of the Healthy Ageing And Intellectual Disabilities Study. Int J Geriatr Psychiatry. 2013;28(7):691-699. 16. Colver A, Rapp M, Eisemann N, et al. Self-reported quality of life of adolescents with cerebral palsy: a cross-sectional and longitudinal analysis. Lancet. 2015;385(9969):705-716. 17. Groh WJ. Arrhythmias in the muscular dystrophies. Heart Rhythm. 2012;9(11):1890-1895. 18. Van Der Slot WM, Nieuwenhuijsen C, Van Den BergEmons RJ, et al. Chronic pain, fatigue, and depressive symptoms in adults with spastic bilateral cerebral palsy [published correction appears in Dev Med Child Neurol. 2012;54(11):1064]. Dev Med Child Neurol. 2012;54(9):836-842. 19. Peterson MD, Kamdar N, Hurvitz EA. Age-related trends in cardiometabolic disease among adults with cerebral palsy. Dev Med Child Neurol. 2019;61(4):484-489. 20. Smith KJ, Peterson MD, O’Connell NE, et al. Risk of depression and anxiety in adults with cerebral palsy [published correction appears in JAMA Neurol. 2019;76(8):986]. JAMA Neurol. 2019; 76(3):294-300. 21. Peterson MD, Ryan JM, Hurvitz EA, Mahmoudi E. Chronic conditions in adults with cerebral palsy. JAMA. 2015;314(21): 2303-2305. 22. Ryan JM, Peterson MD, Matthews A, et al. Non-communicable disease among adults with cerebral palsy: a matched cohort study. Neurology. 2019;93(14):e1385-e1396. https://doi.org/10. 1212/WNL.0000000000008199. 23. Berry JG, Berry SD. Caring for patients with neurological impairment: conversations between a pediatrician and geriatrician. JAMA Pediatr. 2018;172(9):796. 24. Ahmedani BK, Hock RM. Health care access and treatment for children with co-morbid autism and psychiatric conditions. Soc Psychiatry Psychiatr Epidemiol. 2012;47(11):18071814. 25. Vohra R, Madhavan S, Sambamoorthi U, St Peter C. Access to services, quality of care, and family impact for children with autism, other developmental disabilities, and other mental health conditions. Autism. 2014;18(7):815-826. 26. Aisen ML, Kerkovich D, Mast J, et al. Cerebral palsy: clinical care and neurological rehabilitation. Lancet Neurol. 2011; 10(9):844-852. 27. Whiteford HA, Degenhardt L, Rehm J, et al. Global burden of disease attributable to mental and substance use disorders: findings from the Global Burden of Disease Study 2010. Lancet. 2013;382(9904):1575-1586. 28. GBD 2017 Disease and Injury Incidence and Prevalence Collaborators. Global, regional, and national incidence, prevalence, and years lived with disabilitity for 354 diseases and injuries for 195 countries and territories, 1990-2017: a systematic analysis for the Global Burden of Disease Study
Mayo Clin Proc. n XXX 2019;nn(n):1-9 www.mayoclinicproceedings.org
n
29.
30.
31.
32.
33. 34.
35.
36.
37.
38.
39. 40.
41.
2017 [published correction appears in Lancet. 2019; 393(10190):e44]. Lancet. 2018;392(10159):1789-1858. Wilkinson DA, Johnson K, Garton HJ, Muraszko KM, Maher CO. Trends in surgical treatment of Chiari malformation Type I in the United States. J Neurosurg Pediatr. 2017;19(2):208216. Borza T, Jacobs BL, Montgomery JS, et al. No differences in population-based readmissions after open and robotic-assisted radical cystectomy: implications for post-discharge care. Urology. 2017;104:77-83. Stem MS, Blachley TS, Shtein RM, Herman WH, Gardner TW, Stein JD. Impact of diagnosing diabetic complications on future hemoglobin A1c levels. J Diabetes Complications. 2016;30(2): 323-328. Quiñones AR, Markwardt S, Botoseneanu A. Multimorbidity combinations and disability in older adults. J Gerontol A Biol Sci Med Sci. 2016;71(6):823-830. Salive ME. Multimorbidity in older adults. Epidemiol Rev. 2013; 35:75-83. GBD 2017 DALYs and HALE Collaborators. Global, regional, and national disability-adjusted life-years (DALYs) for 359 diseases and injuries and healthy life expectancy (HALE) for 195 countries and territories, 1990-2017: a systematic analysis for the Global Burden of Disease Study 2017 [published correction appears in Lancet. 2019;393(10190):e44]. Lancet. 2018; 392(10159):1859-1922. Peterson MD, Gordon PM, Hurvitz EA. Chronic disease risk among adults with cerebral palsy: the role of premature sarcopoenia, obesity and sedentary behaviour. Obes Rev. 2013;14(2): 171-182. Peterson MD, Gordon PM, Hurvitz EA, Burant CF. Secondary muscle pathology and metabolic dysregulation in adults with cerebral palsy. Am J Physiol Endocrinol Metab. 2012;303(9): E1085-E1093. Whitney DG, Shapiro DN, Warschausky SA, Hurvitz EA, Peterson MD. The contribution of neurological disorders to the national prevalence of depression and anxiety problems among children and adolescents. Ann Epidemiol. 2019;29:81-84.e2. US Preventive Services Task Force. Screening for osteoporosis to prevent fractures: US Preventive Services Task Force recommendation statement. JAMA. 2018;319(24):2521-2531. Tumin D. Marriage trends among Americans with childhoodonset disabilities, 1997-2013. Disabil Health J. 2016;9(4):713-718. Pine DS, Cohen P, Gurley D, Brook J, Ma Y. The risk for earlyadulthood anxiety and depressive disorders in adolescents with anxiety and depressive disorders. Arch Gen Psychiatry. 1998; 55(1):56-64. Jacobson NC, Newman MG. Anxiety and depression as bidirectional risk factors for one another: a meta-analysis of longitudinal studies. Psychol Bull. 2017;143(11):1155-1200.
https://doi.org/10.1016/j.mayocp.2019.07.010
9