DISCUSSION
The present descriptive study demonstrated that expenditures for
patients with SCD in the CHECK High and Medium utilizer tiers decreased
over time. However, the small size of the High utilizer group (n=32) may
have reduced the statistical power to detect a true effect. The SCD
patients in the Low utilizer tier showed significant rise in expenditure
levels over time. The Low utilizer tier group increase in expenditures
after the second year of CHECK may have been due to receiving
appropriate, but formerly unused, services introduced by the CHECK CHWs.
Chronic health conditions effect an estimated 25% of children and
adolescents in the United States and account for a disproportionate
share of pediatric healthcare utilization and expenditures, with the
majority of the $110 billion annual expenditures attributed to hospital
care34. Similarly, while comprising a small proportion
of children and adolescents with chronic diseases, those with SCD have a
disproportionately large effect on health care
expenditures41.
In developing the CHECK program, the hope was that a comprehensive
health care coordination program would reduce health care expenditures.
We did not expect this program would impact all patients equally. While
the overall analysis of the CHECK program showed no change in costs
related to the program33, this exploration specific to
the children with SCD demonstrated a likely impact on healthcare costs
for SCD children.
The comparisons between Low and High, Medium and High utilization risk
groups support the expectation that the odds of zero expenditures were
significantly different from Baseline Year in the predicted direction.
We can speculate that care coordination was associated with some of
these changes, but the study was designed as a demonstration project and
has limitations (discussed below) that preclude definitive conclusion. A
null hypothesis that High and Low utilization are simply exhibiting
regression to the mean cannot be completely excluded. The high odds
ratio of 240.54 for the comparison at Baseline year is likely due to
cells with few patients. Finally, the bimodal distributions of
utilization for all the risk groups (Figure 1) show striking similarity
in Year 2. This longitudinal pattern suggests that one year of
utilization data was not enough to stratification patients for
utilization.
A longitudinal study of 101 children with SCD in Houston in 2011-2013
found that 25% of parents reported receiving care coordination help and
another 20% reported a need for extra care
coordination42. However, multivariate regression did
not detect an association between care coordination and acute care
utilization. The Houston findings are consistent with the stratification
we used. Together, the CHECK program data and Houston data suggest that
much a larger sample size and a rigorous study design might be required
to determine a benefit of care coordination for children with SCD. This
is difficult to do because SCD is a rare disease.
The longitudinal pattern of acute care utilization highlights that some
SCD patients have complications that vary from year to year. These data
suggest that future stratification by utilization risk groups should use
baseline data that spans at least 3 years, and the intervention period
probably should also span multiple years.
Limitations
This study has several limitations. First, because the CHECK program was
designed as a healthcare demonstration project and not as a randomized
clinical trial, there is no control group; patients receiving CHECK
services were targeted for services for specific reasons. The SCD sample
is small, illustrating the small proportion of chronically ill patients
with the disorder but also limiting the statistical power necessary for
introducing other variables in the analyses. Using these heterogeneous
cost data, a sample size estimate for 3 utilization categories with
repeated differences in expenditures across time and 2-3 covariates,
alpha=.05 and power=.80 would require n=3800 SCD patients in each
utilization category to detect a 10% cost difference. The SCD patients
in this study were not selected using randomized sampling procedures and
outliers were removed, which weakens the ability to generalize findings
to the wider SCD population.
Only 52% of SCD patients were fully “engaged” in the CHECK program,
meaning they received full CHECK services tailored to individual needs.
This means our analysis does include some who may have needed services
but did not choose to participate or could not be reached. Some acute
hospitalizations in SCD are avoidable (e.g., hospitalization for sickle
cell pain that was triggered by bad behavioral choices or failure to
fill prescriptions for home pain medications), but other
hospitalizations are unavoidable (e.g., fever or Acute chest syndrome).
This makes determination of the appropriateness of hospitalizations
difficult. New guidelines recommending pediatric use of the
disease-modifying therapy hydroxyurea were released in 2014 during the
CHECK study period and could have reduced the acute care utilization
with or without CHECK services43. CHECK SCD patients
received medical services, care coordination, or education from other
programs, confounding the coordinated care in CHECK. Most of the CHECK
SCD patients were outside of UI Health, receiving a variety of services
in other pediatric SCD programs in the Chicago region. The study design
does not allow untangling a CHECK treatment effect from these other
services. Finally, the longitudinal observational study design leaves
open the possibility that the pattern simply represents the phenomenon
that data at the extremes of a distribution might show “regression
toward the mean” through random changes.
Conclusion
In conclusion, this analysis provides lessons regarding care
coordination service needs for children. The heterogeneity of SCD care
utilization was revealed in the skewed bimodal distribution of
expenditures that emerged in each tier over the three years of data.
Stratification showed a trend of reduced expenditures in the High risk
and Medium risk groups. The CHECK care coordination intervention was a
tailored “precision medicine” approach delivered by CHWs. With this,
the care coordination tried to match the intensity of intervention to
the multiple modifiers and barriers to SCD acute and chronic care. The
benefit of CHECK might have been obscured by low sample size, uneven
intensity of care coordination intervention, duration of intervention
shorter than the natural variability of SCD expenditures (possibly 3
years), and the inability to distinguish the appropriateness of
healthcare utilization for SCD. Future studies of care coordination in
SCD should build upon these lessons learned in CHECK: stratify by tiers
of healthcare utilization, focus on the highest utilizers, design more
than three years of data collection, and add outcomes for
appropriateness of healthcare utilization.