BACKGROUND AND OBJECTIVES

Children with neurologic impairment (NI) are a growing subset of children who frequently use health care. We examined health care use and spending trends across services for children with NI during their first 5 years of life.

METHODS

This was a retrospective study of 13 947 children with NI in the multistate IBM Medicaid MarketScan Database (2009–2017). We established birth cohorts of children with NI and analyzed claims from birth to 5 years. NI, identified by using International Classification of Diseases, 9th Revision, diagnosis codes, was defined as ≥1 neurologic diagnosis that was associated with functional and/or intellectual impairment. We measured annual health care use and per-member-per-year spending by inpatient, emergency department (ED), and outpatient services. Population trends in use and spending were assessed with logistic and linear regression, respectively.

RESULTS

During their first versus fifth year, 66.8% vs 5.8% of children with NI used inpatient services, and 67.8% vs 44.4% used ED services. Annual use in both categories decreased over 0–5 years (inpatient odds ratio: 0.35, 95% confidence interval: 0.34 to 0.36; ED odds ratio: 0.78, 95% confidence interval: 0.77 to 0.79). The use of outpatient services (primary care, specialty care, home health) decreased gradually. Per-member-per-year spending on inpatient services remained the largest spending category: $83 352 (90.2% of annual spending) in the first year and $1944 (25.5%) in the fifth year.

CONCLUSIONS

For children with early-onset NI from 0–5 years, use and spending on inpatient services decreased dramatically; ED and outpatient service use decreased more gradually. These findings may help systems, clinicians, and families optimize care by anticipating and adjusting for shifting use of health care services.

What’s Known on This Subject:

Children with NI use more health care services than other children. Although their inpatient health care use is known to be recurrent and costly, little is known about their outpatient use or trends in use and spending over time.

What This Study Adds:

For children with early-onset NI, use and spending on inpatient services from 0–5 years decreased dramatically; emergency and outpatient service use decreased more gradually. Systems could optimize care by anticipating trends in shifting use of inpatient and outpatient services.

Children with neurologic impairment (NI) are a growing subset of children with chronic conditions because care advances have improved their survival and recognition.1  These children have static or progressive conditions that affect the nervous system and include children with hydrocephalus, hypoxic-ischemic injuries, genetic or metabolic disorders, and/or epilepsy. During their early years of life, the majority of children with NI develop multiple comorbidities, most commonly respiratory (eg, asthma), digestive (eg, gastroesophageal reflux), and mental and/or behavioral health diagnoses (eg, attention-deficit/hyperactivity disorder).2 

Children with NI profoundly impact the health care system with high use and costs35  and affect their families with heavy care responsibilities.6  Health systems and insurers, such as Medicaid, spend ∼50% of their budgets on the 5% of children with the highest needs, including children with NI.7,8  These children’s high health care costs appear to be driven by their inpatient care use,5,913  but little is known about their use of outpatient services (such as primary care; outpatient specialty care; occupational, physical, and speech therapy; mental health and behavioral counseling; home health; and outpatient medications).14,15  The components and trends of health care use outside the inpatient setting deserve careful evaluation because more effective outpatient services might reduce these children’s need for expensive and distressing hospitalizations.

Better understanding of health care use trends could inform policy decisions to develop more efficient care delivery systems, distribute spending more efficiently across services, and potentially improve patient outcomes. Although the ideal distribution of health care resources to best support the health of children with NI is unknown, further optimization is needed because almost one-half of families who care for children with NI report unmet health care needs or dissatisfaction with care.12  Moreover, if clinicians understood use trends for young children with NI, they could more effectively counsel new caregivers about their infant’s future health care use trajectories and help prepare them for managing changes in care as their children age.16,17 

To our knowledge, no longitudinal studies have evaluated health care use and spending for these children, taking into account both inpatient and outpatient services. To address this gap, we examined trends in health care use and spending for children with NI across age and service categories from birth to the age of 5 years. We hypothesized that the proportion of children with early-onset NI using inpatient and emergency department (ED) services would decrease over this period, and that inpatient services would remain the largest annual spending category across these years.

We performed a retrospective longitudinal cohort analysis of Medicaid-enrolled children with NI, using the IBM Medicaid MarketScan Database from years 2009–2017 (IBM Watson Health, Armonk, NY). Children were enrolled in Medicaid care plans from 12 deidentified states across the United States. The data set contains deidentified data on demographics, clinical diagnoses, and paid claims across the inpatient and outpatient care continuum.

Children were included in our sample if they received ≥1 NI diagnosis during their first 6 months of life, such as brain abnormalities, epilepsy, or intracranial hemorrhage. NI diagnoses were identified by using a previously established set of 604 International Classification of Diseases, Ninth Revision, codes that were originally identified by multiple pediatric neurologists, who reviewed and approved NI diagnoses using a modified Delphi technique.5  They used a NI definition of: “diagnosis is consistent with NI (static or progressive) and typically results in either functional and/or intellectual impairment.” We included subjects in the database from 2009 to 2012 if they had a birth hospitalization identified by using International Classification of Diseases, Ninth Revision, codes of V30-V39 and were continuously enrolled in Medicaid through age 5 (allowing for a 1-month lapse per year). This study was deemed not to be human subject research by our institutional review board.

Our primary outcomes were annual health care use (any versus none) and spending by service category across the care continuum for children with NI during their first 5 years of life. Service categories were provided by IBM Watson and grouped by health care setting (ie, inpatient, ED, and outpatient). Outpatient use was further subcategorized into 6 services: primary care; specialty care; occupational, physical, and speech therapy; mental health and behavioral counseling; home health; and outpatient medications.

Health care use was reported as the proportion of the study sample who received inpatient, ED, and outpatient subcategory services in each year of life. Because birth hospitalization was an inclusion criterion, use of inpatient services during the first year of life was defined as reuse of inpatient services after birth hospitalization, or prolonged birth hospitalization (>3 days).

Health care spending was reported as the per-member-per-year (PMPY) spending by service category.

For subjects, we report sex, reason for Medicaid enrollment, NI category, number of complex chronic conditions (CCCs), and use of NICU services. Seven NI categories were assessed: (1) static neurologic disease, (2) progressive neurologic disease, (3) anatomic abnormality, (4) epilepsy, (5) genetic or metabolic condition, (6) cerebrovascular disease, and (7) peripheral neurologic disease. NI categories were not mutually exclusive because some patients have diagnoses in multiple categories. CCCs are conditions that are expected to last ≥12 months, require specialty care, and are likely to require hospitalization because of medical diagnoses.18  Subjects were identified as using NICU services if their birth hospitalization included services billed under intensive care.

To explore trends in health care use and spending in a subset of children with NI who also had medical complexity, we performed a subanalysis of children with NI who had >2 CCCs. These children fit the generally accepted definition of children with medical complexity (CMC),19  and we refer to them as CMC with NI.

We examined the annual health care use and PMPY spending by service categories, which included inpatient, ED, and 6 outpatient subcategory services (see outcomes). To evaluate trends in use, we modeled annual use as a binary outcome for each service category, by using logistic regression with a fixed effect for year. Results are presented as an odds ratio (OR), which can be interpreted as the annual change in the odds of use for a one-year increase in age. To analyze trends in health care spending, we modeled annual spending as a continuous outcome using linear regression with a fixed effect for year. Given that annual spending was not normally distributed, we performed log transformation of the data for the trend analysis. This year-over-year analysis yielded a mean annual change (MAC), which can be interpreted as the MAC in spending for a one-year increase in age.

All analyses were completed by using SAS version 9.4 (SAS Institute, Cary, North Carolina). P values ≤0.05 were considered statistically significant.

There were 13 947 children with NI (5.0% prevalence) who were continuously enrolled in Medicaid from 0 to 5 years of age. In the cohort, 37.2% used NICU services after birth (Table 1).

TABLE 1

Demographic Characteristics and Diagnoses of Young Children With NI Enrolled in Medicaid

Characteristics(N = 13 947), n (%)
Sex, male 7546 (54.1) 
Reason for enrollment in Medicaid  
 Family income 11 604 (83.2) 
 Child disability 1518 (10.9) 
 Other or unknown 825 (5.9) 
Used NICU services during birth hospitalization 5186 (37.2) 
NI category, diagnosed by the age of 6 mo  
 Genetic or metabolic condition 3370 (24.2) 
 Anatomic abnormality 3086 (22.1) 
 Epilepsy 3056 (21.9) 
 Cerebrovascular disease 2880 (20.6) 
 Static neurologic disease 2024 (14.5) 
 Peripheral neurologic disease 187 (1.3) 
 Progressive neurologic disease 11 (0.1) 
No. CCC, diagnosed by the age of 6 mo  
 0 3825 (27.4) 
 1 5181 (37.1) 
 ≥2 4941 (35.5) 
Technology dependence 1476 (10.6) 
Characteristics(N = 13 947), n (%)
Sex, male 7546 (54.1) 
Reason for enrollment in Medicaid  
 Family income 11 604 (83.2) 
 Child disability 1518 (10.9) 
 Other or unknown 825 (5.9) 
Used NICU services during birth hospitalization 5186 (37.2) 
NI category, diagnosed by the age of 6 mo  
 Genetic or metabolic condition 3370 (24.2) 
 Anatomic abnormality 3086 (22.1) 
 Epilepsy 3056 (21.9) 
 Cerebrovascular disease 2880 (20.6) 
 Static neurologic disease 2024 (14.5) 
 Peripheral neurologic disease 187 (1.3) 
 Progressive neurologic disease 11 (0.1) 
No. CCC, diagnosed by the age of 6 mo  
 0 3825 (27.4) 
 1 5181 (37.1) 
 ≥2 4941 (35.5) 
Technology dependence 1476 (10.6) 

The 3 most common NI categories were genetic or metabolic condition (24.2%), anatomic abnormality (22.1%), and epilepsy (21.9%) (see Table 1; individual diagnoses are shown in Supplemental Table 4). Overall, 72.6% of children with early-onset NI had ≥1 CCC, and 10.6% were technology dependent (eg, using a feeding tube or tracheostomy). In the sample, 15.9% met criteria for CMC with NI.

Inpatient Services

Whereas 66.8% of children with NI used inpatient services during their first year, only 5.8% did so during their fifth year. Across their first 5 years, annual inpatient service use showed a decreasing trend (OR: 0.35; 95% confidence interval [CI]: 0.34 to 0.36), with use decreasing by 80.7% between their first and second year (Table 2). Thereafter, the rate of use continued to decrease by 15% to 30% annually. For those who used inpatient services at any point during their first 5 years, the median length of stay was 3 days (interquartile range [IQR]: 2–9 days). The 4 most common diagnosis-related groups for inpatient use were asthma and bronchitis, dehydration, seizure, and gastroenteritis.

TABLE 2

Annual Health Care Use by Year of Life of Children with NI Enrolled in Medicaid (N = 13 947)

Health Care Service CategoryUse During Each Year of Life
(Proportion Who Used Each Service, %)
Trend Analysisa
OR (95% CI)
12345
Inpatient 66.8 12.9 8.9 7.0 5.8 0.35 (0.34 to 0.36) 
EDb 67.8 62.8 55.2 49.1 44.4 0.78 (0.77 to 0.79) 
Outpatient       
 Primary care 97.7 95.2 91.2 87.8 88.7 0.69 (0.68 to 0.71) 
 Specialty care 84.8 78.1 71.2 64.4 69.0 0.79 (0.78 to 0.80) 
 Outpatient therapyc 50.7 44.3 37.5 33.8 32.1 0.82 (0.81 to 0.83) 
 Mental health and behavioral counseling 25.1 27.1 29.8 26.4 28.0 1.03 (1.01 to 1.04) 
 Home health 22.3 3.8 2.2 2.1 1.9 0.41 (0.39 to 0.42) 
 Outpatient medications 95.5 92.7 87.7 84.9 84.5 0.73 (0.72 to 0.74) 
Health Care Service CategoryUse During Each Year of Life
(Proportion Who Used Each Service, %)
Trend Analysisa
OR (95% CI)
12345
Inpatient 66.8 12.9 8.9 7.0 5.8 0.35 (0.34 to 0.36) 
EDb 67.8 62.8 55.2 49.1 44.4 0.78 (0.77 to 0.79) 
Outpatient       
 Primary care 97.7 95.2 91.2 87.8 88.7 0.69 (0.68 to 0.71) 
 Specialty care 84.8 78.1 71.2 64.4 69.0 0.79 (0.78 to 0.80) 
 Outpatient therapyc 50.7 44.3 37.5 33.8 32.1 0.82 (0.81 to 0.83) 
 Mental health and behavioral counseling 25.1 27.1 29.8 26.4 28.0 1.03 (1.01 to 1.04) 
 Home health 22.3 3.8 2.2 2.1 1.9 0.41 (0.39 to 0.42) 
 Outpatient medications 95.5 92.7 87.7 84.9 84.5 0.73 (0.72 to 0.74) 
a

Annual use (any use versus no use) for each service category was modeled by using logistic regression. Results are presented as an OR, which can be interpreted as the annual change in the odds of use for a 1 year of life increase. All OR analyses are significant at P < .001.

b

ED use not resulting in admission.

c

Includes occupational, physical, and speech therapy.

PMPY spending on inpatient services showed a decreasing trend (MAC: −$15 232; 95% CI: −$15 627 to −$14 837, Table 3). Annual spending on inpatient services was consistently the largest spending category, although the proportion of annual spending on inpatient services decreased over the time period (90.6% of annual spending in the first year, 25.5% in the fifth year).

TABLE 3

Annual Health Care Spending by Year of Life of Children With NI Enrolled in Medicaid (N = 13 947)

Health Care Service CategoryPMPY Health Care Spending During Each Year of Life, $ (Proportion of Annual Spending, %)Trend Analysisa MAC, $ (95% CI)
12345
Total annual health care spending 86 128 11 896 9131 7764 7623  
Inpatient 77 996 (90.6) 4556 (38.3) 2901 (31.8) 2172 (28.0) 1944 (25.5) −15 232 (−15 627 to −14 837) 
EDb 574 (0.7) 489 (4.1) 383 (4.2) 328 (4.2) 278 (3.6) −75 (−79 to −72) 
Outpatient       
 Primary care 994 (1.2) 673 (5.7) 398 (4.4) 323 (4.2) 351 (4.6) −164 (−166 to −161) 
 Specialty care 1307 (1.5) 1522 (12.8) 1534 (16.8) 1441 (18.6) 1463 (19.2) 23 (−15 to 61) 
 Outpatient therapyc 956 (1.1) 1126 (9.5) 980 (10.7) 886 (11.4) 810 (10.6) −53 (−66 to −40) 
 Mental health and behavioral counseling 456 (0.5) 841 (7.1) 1026 (11.2) 733 (9.4) 797 (10.5) 57 (43 to 72) 
 Home health 399 (0.5) 267 (2.2) 137 (1.5) 113 (1.5) 110 (1.4) −73 (−85 to −61) 
 Outpatient medications 2039 (2.4) 1508 (12.7) 954 (10.5) 967 (12.5) 1071 (14.0) −248 (−293 to −203) 
Other services 1407 (1.6) 915 (7.7) 817 (8.9) 801 (10.3) 800 (10.5) — 
Health Care Service CategoryPMPY Health Care Spending During Each Year of Life, $ (Proportion of Annual Spending, %)Trend Analysisa MAC, $ (95% CI)
12345
Total annual health care spending 86 128 11 896 9131 7764 7623  
Inpatient 77 996 (90.6) 4556 (38.3) 2901 (31.8) 2172 (28.0) 1944 (25.5) −15 232 (−15 627 to −14 837) 
EDb 574 (0.7) 489 (4.1) 383 (4.2) 328 (4.2) 278 (3.6) −75 (−79 to −72) 
Outpatient       
 Primary care 994 (1.2) 673 (5.7) 398 (4.4) 323 (4.2) 351 (4.6) −164 (−166 to −161) 
 Specialty care 1307 (1.5) 1522 (12.8) 1534 (16.8) 1441 (18.6) 1463 (19.2) 23 (−15 to 61) 
 Outpatient therapyc 956 (1.1) 1126 (9.5) 980 (10.7) 886 (11.4) 810 (10.6) −53 (−66 to −40) 
 Mental health and behavioral counseling 456 (0.5) 841 (7.1) 1026 (11.2) 733 (9.4) 797 (10.5) 57 (43 to 72) 
 Home health 399 (0.5) 267 (2.2) 137 (1.5) 113 (1.5) 110 (1.4) −73 (−85 to −61) 
 Outpatient medications 2039 (2.4) 1508 (12.7) 954 (10.5) 967 (12.5) 1071 (14.0) −248 (−293 to −203) 
Other services 1407 (1.6) 915 (7.7) 817 (8.9) 801 (10.3) 800 (10.5) — 

—, not applicable.

a

All MAC analyses are significant at P < .001, except specialty care (P =.2).

b

ED use not resulting in admission.

c

Includes occupational, physical, and speech therapy.

ED Visits

We found a decreasing trend in both ED service use (OR: 0.78; 95% CI: 0.77 to 0.79) and spending (MAC: −$75; 95% CI: −$79 to −$72), although the decrease was more gradual than that observed for inpatient services. The rate of ED use decreased by 7% to 12% annually. For children with early-onset NI, the mean days of ED use was 2.0 during their first year, 1.2 days during their third year, and 0.9 days during their fifth year. The 4 most common principal diagnoses for ED use were fever, upper respiratory infection, otitis media, and vomiting. PMPY spending on ED services represented ∼4% of annual spending for children >1 year of age.

Outpatient Services: Primary Care

Across their first 5 years, the majority of children with NI used primary care services (>85% of children each year) but had a decreasing trend of use (OR: 0.69; 95% CI: 0.68 to 0.71). PMPY spending on primary care services represented ∼5% of annual spending for those >1 year of age.

Outpatient Services: Specialty

Specialty services were used by >64% of children with NI each year. PMPY spending on specialty services was the second largest spending category annually and remained constant across the first 5 years (MAC: $23; 95% CI: −$15 to $61). Thus, as the PMPY spending on other services decreased, spending on specialty services represented an increasing proportion of annual spending: ∼20% by the fifth year (PMPY: $1463).

Outpatient Services: Other

Annual use of other outpatient services shifted over the 5 years (Table 2). For example, the use of home health services decreased by 82.9% after the first year, from 22.3% in the first year to 3.8% in the second. Mental health and behavioral counseling was the one outpatient service that showed a trend of increased use (OR: 1.03; 95% CI: 1.01 to 1.04).

Total Spending

In total, per member health care spending for children with early-onset NI during their entire first 5 years of life was $122 544. The median per member health care spending for the entire first 5 years was $34 394 (IQR: $12 503–$133 521). As children with NI aged from 0 to 5 years, the distribution of spending transitioned from primarily inpatient during their first year (>90% of PMPY spending) to a more even distribution across service lines in subsequent years (Table 3).

CMC with NI, who had >2 CCCs and NI, had higher health care use and spending over their first 5 years than noncomplex children with NI did but demonstrated similar decreasing trends of use and spending.

Inpatient Services

During their first year of life, 95.5% of CMC with NI used inpatient services, compared with 16.7% in their fifth year. Across their first 5 years, annual inpatient service use showed a decreasing trend (OR: 0.41, 95% CI: 0.40 to 0.43), decreasing by 65.9% between the first and second year (Supplemental Table 5). Over 5 years, PMPY spending on inpatient services showed a decreasing trend (MAC: −$43 738; 95% CI: −$45 538 to −$41 938). PMPY spending on inpatient services was $219 555 during the first year (92.0% of annual spending) and $6644 during the fifth year (31.7%).

ED services

During each year, >52% of CMC with NI used ED services, with a decreasing trend (OR: 0.80; 95% CI: 0.78 to 0.82).

Outpatient services

Over their first 5 years, the vast majority of CMC with NI consistently used primary care (>91% each year) and specialty services (>80%). Annual spending on specialty services was consistently the second largest category, representing ∼20% to 25% of annual spending for those >1 year of age.

Total spending

Total PMPY spending on health care services was $238 535 in year 1, $27 666 in year 3, and $20 952 in year 5 (Supplemental Table 6). In total, per member health care spending for CMC with NI during their entire first 5 years of life was $345 702. The median per member health care spending for the entire first 5 years was $235 860 (IQR: $97 372–$450 520).

As we hypothesized, our study of Medicaid-enrolled children with early-onset NI found decreasing use of inpatient, ED, and outpatient services as they aged from 0 to 5 years. Spending on inpatient services remained the largest annual spending category in all years but decreased dramatically after the first year, when the distribution of spending shifted to a more even distribution across inpatient and outpatient care. To our knowledge, this study is the first to examine longitudinal health care use across inpatient and outpatient care settings for a cohort of young children with NI. Our subanalysis of CMC with NI revealed similar trends from birth to 5 years, although levels of use and spending were predictably higher for this group.

This study provides a population-based overview of health care use for children with early-onset NI over their first 5 years of life. Birth cohort analyses, such as ours, help to clarify the natural history of health care use and could inform development of innovative population-based programs. Targeted efforts to reduce inpatient use during the costly first year, which accounted for >90% of that year’s health care costs, may reduce overall spending. Strategies for clinicians to consider adopting to achieve this goal might include proactively educating families about how to recognize and manage acute medical problems including respiratory and dehydration-related conditions at home, optimizing family support from outpatient services such as home health, and improving timely access to outpatient specialty care through availability of same-day or urgent appointments. This study also suggests the need for further research to examine the specific reasons for inpatient and outpatient service use in this patient group. Such efforts could identify modifiable child, family, and health system-level factors to optimize use, maximize value, and decrease unnecessary costs.

Our study highlights the relatively small amount spent on primary care for children with NI (PMPY ∼$500), representing only 5% of annual spending. Other studies have found that two-thirds of children with NI do not have access to a primary care medical home and primary care systems struggle to coordinate services across the care spectrum for these patients with limited financial resources.20  Given that more frequent well-child primary care visits have been associated with a lower likelihood of hospitalizations,21  in future studies, researchers should evaluate the optimal number of annual primary care visits for children with NI and examine enrollment in medical homes. It is telling that use of home health services decreased after these children’s first year by 82.9% to only <4% for those >1 year of age. Given home health’s potential promise to reduce expensive inpatient service use22,23  and promote family functioning,16  future work is necessary to assess if this low level of use meets the needs of these children and their families.

The only service subcategory where use increased across the first 5 years was behavioral and mental health services, used most frequently by children receiving services for developmental delay, intellectual disabilities, attention-deficit/hyperactivity disorder, conduct disorders, and depressive disorders. Given that the majority of children with NI develop behavioral and mental health comorbidities by the age of 6,2  these children could benefit from proactive screening and optimization of behavioral and mental health conditions before school age, when behavioral problems interfere with academic and social success and lead to higher health and education resource use.2427 

In our study, we found that the majority of children with NI use multiple different health services across their early years, especially in their first year, which may highlight the potential need for proactive care coordination. Care coordination is defined as a team-based effort to organize and drive service integration to address medical, social, developmental, behavioral, educational, and financial needs.28,29  Our finding that substantially fewer children with NI used inpatient services after their first year of life complements a growing body of evidence that many children with chronic conditions use fewer health care services after high-use periods.3,4  As medical home interventions and value-based care models assess their impact on use and spending,3033  they should account for our findings of decreased use of health care services over time. Although this result may reflect medical stabilization of these children’s chronic diseases,34,35  it might also be explained by more multifaceted health care processes.

Despite the decreasing trend in ED use over the first 5 years, the proportion of children with NI who used ED services each year remained substantial (>40% annually), even while use of inpatient services markedly decreased. This finding suggests that ED visits are prompted by different factors than hospitalizations. Previous studies have shown that families of children with chronic conditions such as NI are driven to seek emergency care by a complex mix of contextual factors: chronic disease decompensation, lack of access to ambulatory or specialty care, lack of caregiver self-efficacy, or absence of psychosocial supports.36,37  These multiple factors result in variable patterns of ED use that may be modifiable.38 

Our study has several limitations. Because we used a deidentified claims-based database, we could not analyze family, community, or regional attributes that might influence use and spending. Analyzing use as a binary variable did not allow for examination of specific risk factors for intensity of use. Our findings do not reflect children’s use of services for which Medicaid claims were not submitted (eg, day care or transportation). Our definition of NI, although based on the most widely used NI coding system, also included disorders with heterogeneous levels of impairment. In addition, our sample is representative of children with NI enrolled in Medicaid and may not generalize to privately insured children.

Another limitation is that some level of selection bias may have been present, given that we assessed NI diagnoses during the first 6 months of life to define our study population and required participants to be continuously enrolled in Medicaid. Thus, we did not include those who were diagnosed with NI later in childhood, those who died before 5 years of age, or those with intermittent Medicaid coverage. Given these several possible sources of bias, the direction in either over or underestimating health care use is unknown. In addition, some early-onset NI conditions may be mild in severity or improve over time, whereas others could be persistent and may result in more consistent health care use.

Our findings may help to guide the design and evaluation plans of population-health based interventions for children with early-onset NI. Health system leaders could leverage our identified patterns of health care use to incentivize a redistribution of services for this population, and consider further optimization through early intervention, primary care, and/or home health. In future studies, researchers should continue exploration of subgroups of children with NI, particularly those with higher disease acuity and multimorbidity,39  to examine how specific diagnoses, including mental and behavioral health, drive service use and spending.

We encourage providers to use our findings to counsel families of children with NI about how to plan for their child’s future health care needs. Parents appreciate receiving this information near the time of diagnosis, even if it is based on uncertain predictions, so they can prepare emotionally and logistically to navigate the health system.4044  For example, infants with NI are frequently admitted for respiratory conditions, so families of neonates with NI should be taught to recognize and manage respiratory decompensation. In addition, given our finding that approximately one-half of children with NI use ED services annually, clinicians could offer anticipatory guidance to help families implement plans for managing acute illness at home and proactively prepare for ED visits.

Children with NI enrolled in Medicaid showed trends of decreasing annual rates of use of inpatient, ED, and outpatient services (with the exception of mental and behavioral health) across their first 5 years of life. Annual use and spending on inpatient services markedly decreased after the first year but remained the largest spending category for these children each year. Health systems and policy makers should be encouraged to optimize care models for children with NI that anticipate longitudinal trends across service categories. Clinicians may leverage these findings when counseling families of children with NI to help them prepare in practical ways for shifting health care use patterns as their children age.

Constance D. Baldwin, PhD (Professor of Pediatrics, University of Rochester, Rochester, New York), offered valued mentorship and thoughtful revisions of the final manuscript.

FUNDING: Supported by the Children’s Hospital Association through the Health Services Research Academy (2020 Cohort). Dr Thomson was supported by the Agency for Healthcare Research and Quality (AHRQ K08-HS025138). Dr Feinstein was supported by the Eunice Kennedy Shriver National Institute of Child Health & Human Development of the National Institutes of Health (NIH K23-HD091295). The content is solely the responsibility of the authors and does not represent the official views of the National Institutes of Health or the US government.

Dr Bayer conceptualized and designed the study, conducted the initial analyses, drafted the initial manuscript, and reviewed and revised the manuscript; Drs Hall and Berry conceptualized and designed the study, conducted the initial analyses, and reviewed and revised the manuscript; Drs Li, Feinstein, and Thomson conceptualized and designed the study, interpreted the analyses, and reviewed and revised the manuscript; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

This study was presented in part as a virtual platform at the Pediatric Academic Societies Meeting, May 3, 2021.

CCC

complex chronic condition

CI

confidence interval

CMC

children with medical complexity

ED

emergency department

IQR

interquartile range

MAC

mean annual change

NI

neurologic impairment

OR

odds ratio

PMPY

per-member-per-year

1
Perrin
JM
,
Anderson
LE
,
Van Cleave
J
.
The rise in chronic conditions among infants, children, and youth can be met with continued health system innovations
.
Health Aff (Millwood).
2014
;
33
(
12
):
2099
2105
2
Thomson
J
,
Hall
M
,
Nelson
K
, et al
.
Timing of co-occurring chronic conditions in children with neurologic impairment
.
Pediatrics.
2021
;
147
(
2
):
e2020009217
3
Agrawal
R
,
Hall
M
,
Cohen
E
, et al
.
Trends in health care spending for children in Medicaid with high resource use
.
Pediatrics.
2016
;
138
(
4
):
e20160682
4
Cohen
E
,
Berry
JG
,
Camacho
X
,
Anderson
G
,
Wodchis
W
,
Guttmann
A
.
Patterns and costs of health care use of children with medical complexity
.
Pediatrics.
2012
;
130
(
6
):
e1463
e1470
5
Berry
JG
,
Poduri
A
,
Bonkowsky
JL
, et al
.
Trends in resource utilization by children with neurological impairment in the United States inpatient health care system: a repeat cross-sectional study
.
PLoS Med.
2012
;
9
(
1
):
e1001158
6
Foster
CC
,
Agrawal
RK
,
Davis
MM
.
Home health care for children with medical complexity: workforce gaps, policy, and future directions
.
Health Aff (Millwood).
2019
;
38
(
6
):
987
993
7
Berry
JG
,
Hall
M
,
Neff
J
, et al
.
Children with medical complexity and Medicaid: spending and cost savings
.
Health Aff (Millwood).
2014
;
33
(
12
):
2199
2206
8
Kuo
DZ
,
Hall
M
,
Agrawal
R
, et al
.
Comparison of health care spending and utilization among children with Medicaid insurance
.
Pediatrics.
2015
;
136
(
6
):
e1521
e1529
9
Berry
JG
,
Hall
DE
,
Kuo
DZ
, et al
.
Hospital utilization and characteristics of patients experiencing recurrent readmissions within children’s hospitals
.
JAMA.
2011
;
305
(
7
):
682
690
10
Murphy
NA
,
Hoff
C
,
Jorgensen
T
,
Norlin
C
,
Young
PC
.
Costs and complications of hospitalizations for children with cerebral palsy
.
Pediatr Rehabil.
2006
;
9
(
1
):
47
52
11
Simon
TD
,
Berry
J
,
Feudtner
C
, et al
.
Children with complex chronic conditions in inpatient hospital settings in the United States
.
Pediatrics.
2010
;
126
(
4
):
647
655
12
Bitsko
RH
,
Visser
SN
,
Schieve
LA
,
Ross
DS
,
Thurman
DJ
,
Perou
R
.
Unmet health care needs among CSHCN with neurologic conditions
.
Pediatrics.
2009
;
124
(
suppl 4
):
S343
S351
13
Boulet
SL
,
Boyle
CA
,
Schieve
LA
.
Health care use and health and functional impact of developmental disabilities among US children, 1997-2005
.
Arch Pediatr Adolesc Med.
2009
;
163
(
1
):
19
26
14
Barnert
ES
,
Coller
RJ
,
Nelson
BB
, et al
.
Experts’ perspectives toward a population health approach for children with medical complexity
.
Acad Pediatr.
2017
;
17
(
6
):
672
677
15
Coller
RJ
,
Berry
JG
,
Kuo
DZ
, et al
.
Health system research priorities for children and youth with special health care needs
.
Pediatrics.
2020
;
145
(
3
):
e20190673
16
Kuo
DZ
,
Cohen
E
,
Agrawal
R
,
Berry
JG
,
Casey
PH
.
A national profile of caregiver challenges among more medically complex children with special health care needs
.
Arch Pediatr Adolesc Med.
2011
;
165
(
11
):
1020
1026
17
Edwards
JD
.
Anticipatory guidance on the risks for unfavorable outcomes among children with medical complexity
.
J Pediatr.
2017
;
180
:
247
250
18
Feudtner
C
,
Hays
RM
,
Haynes
G
,
Geyer
JR
,
Neff
JM
,
Koepsell
TD
.
Deaths attributed to pediatric complex chronic conditions: national trends and implications for supportive care services
.
Pediatrics.
2001
;
107
(
6
):
E99
19
Berry
JG
,
Hall
M
,
Cohen
E
,
O’Neill
M
,
Feudtner
C
.
Ways to identify children with medical complexity and the importance of why
.
J Pediatr.
2015
;
167
(
2
):
229
237
20
Phelps
RA
,
Pinter
JD
,
Lollar
DJ
,
Medlen
JG
,
Bethell
CD
.
Health care needs of children with Down syndrome and impact of health system performance on children and their families
.
J Dev Behav Pediatr.
2012
;
33
(
3
):
214
220
21
Shumskiy
I
,
Richardson
T
,
Brar
S
, et al
.
Well-child visits of Medicaid-insured children with medical complexity
.
J Pediatr.
2018
;
199
:
223
230.e2
22
Gay
JC
,
Thurm
CW
,
Hall
M
, et al
.
Home health nursing care and hospital use for medically complex children
.
Pediatrics.
2016
;
138
(
5
):
e20160530
23
Maynard
R
,
Christensen
E
,
Cady
R
, et al
.
Home health care availability and discharge delays in children with medical complexity
.
Pediatrics.
2019
;
143
(
1
):
e20181951
24
Hysing
M
,
Elgen
I
,
Gillberg
C
,
Lie
SA
,
Lundervold
AJ
.
Chronic physical illness and mental health in children. Results from a large-scale population study
.
J Child Psychol Psychiatry.
2007
;
48
(
8
):
785
792
25
Mattson
G
,
Kuo
DZ
;
Committee on Psychosocial Aspects of Child and Family Health; Council on Children with Disabilities
.
Psychosocial factors in children and youth with special health care needs and their families
.
Pediatrics.
2019
;
143
(
1
):
e20183171
26
Torio
CM
,
Encinosa
W
,
Berdahl
T
,
McCormick
MC
,
Simpson
LA
.
Annual report on health care for children and youth in the United States: national estimates of cost, utilization and expenditures for children with mental health conditions
.
Acad Pediatr.
2015
;
15
(
1
):
19
35
27
Howell
EM
,
Teich
J
.
Variations in Medicaid mental health service use and cost for children
.
Adm Policy Ment Health.
2008
;
35
(
3
):
220
228
28
Antonelli
RC
,
McAllister
JW
,
Popp
J
.
Making Care Coordination a Critical Component of the Pediatric Health System: a Multidisciplinary Framework.
New York, NY
:
The Commonwealth Fund
;
2009
29
Kuo
DZ
,
McAllister
JW
,
Rossignol
L
,
Turchi
RM
,
Stille
CJ
.
Care coordination for children with medical complexity: whose care is it, anyway?
Pediatrics.
2018
;
141
(
Suppl 3
):
S224
S232
30
Coller
RJ
,
Nelson
BB
,
Sklansky
DJ
, et al
.
Preventing hospitalizations in children with medical complexity: a systematic review
.
Pediatrics.
2014
;
134
(
6
):
e1628
e1647
31
Ehlenbach
ML
,
Coller
RJ
.
Growing evidence for successful care management in children with medical complexity
.
Pediatrics.
2020
;
145
(
4
):
e20193982
32
Bergman
DA
,
Keller
D
,
Kuo
DZ
, et al
.
Costs and use for children with medical complexity in a care management program
.
Pediatrics.
2020
;
145
(
4
):
e20192401
33
Simon
TD
,
Whitlock
KB
,
Haaland
W
, et al
.
Effectiveness of a comprehensive case management service for children with medical complexity
.
Pediatrics.
2017
;
140
(
6
):
e20171641
34
Homer
CJ
,
Klatka
K
,
Romm
D
, et al
.
A review of the evidence for the medical home for children with special health care needs
.
Pediatrics.
2008
;
122
(
4
):
e922
e937
35
Mosquera
RA
,
Avritscher
EBC
,
Samuels
CL
, et al
.
Effect of an enhanced medical home on serious illness and cost of care among high-risk children with chronic illness: a randomized clinical trial
.
JAMA.
2014
;
312
(
24
):
2640
2648
36
Berry
JG
,
Agrawal
R
,
Kuo
DZ
, et al
.
Characteristics of hospitalizations for patients who use a structured clinical care program for children with medical complexity
.
J Pediatr.
2011
;
159
(
2
):
284
290
37
Berry
JG
,
Rodean
J
,
Hall
M
, et al
.
Impact of chronic conditions on emergency department visits of children using Medicaid
.
J Pediatr.
2017
;
182
:
267
274
38
Coller
RJ
,
Rodean
J
,
Linares
DE
, et al
.
Variation in hospitalization rates following emergency department visits in children with medical complexity
.
J Pediatr.
2019
;
214
:
113
120.e1
39
Thomson
JE
,
Feinstein
JA
,
Hall
M
,
Gay
JC
,
Butts
B
,
Berry
JG
.
Identification of children with high-intensity neurological impairment
.
JAMA Pediatr.
2019
;
173
(
10
):
989
991
40
Baird
G
,
McConachie
H
,
Scrutton
D
.
Parents’ perceptions of disclosure of the diagnosis of cerebral palsy
.
Arch Dis Child.
2000
;
83
(
6
):
475
480
41
Hasnat
MJ
,
Graves
P
.
Disclosure of developmental disability: a study of parent satisfaction and the determinants of satisfaction
.
J Paediatr Child Health.
2000
;
36
(
1
):
32
35
42
Davis
E
,
Reddihough
D
,
Murphy
N
, et al
.
Exploring quality of life of children with cerebral palsy and intellectual disability: what are the important domains of life?
Child Care Health Dev.
2017
;
43
(
6
):
854
860
43
Rosenbaum
P
.
Classification of abnormal neurological outcome
.
Early Hum Dev.
2006
;
82
(
3
):
167
171
44
Milner
J
,
Bungay
C
,
Jellinek
D
,
Hall
DM
.
Needs of disabled children and their families
.
Arch Dis Child.
1996
;
75
(
5
):
399
404

Competing Interests

FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.

POTENTIAL CONFLICT OF INTEREST: The authors have no conflicts of interest relevant to this article to disclose.

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