Determinants of Health Care Use in a Population-Based Leukodystrophy Cohort

Abstract

Objectives To determine the costs for children with leukodystrophies, and whether high costs were associated with characteristic clinical features or resources utilization. Study design We determined health care costs in a population cohort of 122 patients with leukodystrophy including inpatient, outpatient, and emergency department use, over a 9 year period. We analyzed differences in patients with high costs (>85th percentile) and their health care utilization. Results Patients with leukodystrophy had significant variability in resource utilization, with the top 15th percentile of patients accounting for 73% of costs ($9.6 million). The majority of costs, 81% ($10.8 million), arose from inpatient hospitalization. High-cost patients had more and longer hospitalizations, increased requirements for intensive unit care and mechanical ventilation, and significantly more infections. Importantly, bone marrow transplantation did not solely account for the difference between high-cost and low-cost groups. Conclusion Inpatient hospitalization is the greatest source of health care resource utilization in patients with leukodystrophy. A minority of patients account for the majority of costs, primarily due to an increased volume of hospitalization. Strategies to improve care and reduce costs will need to reduce inpatient stays and target modifiable reasons for hospitalization.

Costs of the diagnostic odyssey in children with inherited leukodystrophies

“…In comparison, overall costs of care for patients with leukodystrophy averaged more than $107,000, and in some patients more than $1 million. 1,4,8 Thus, financial costs for the diagnostic odyssey and diagnostic testing are relatively low compared to the overall costs of care for patients with leukodystrophy. It would seem that financial costs of the diagnostic odyssey should not limit pursuit of diagnosis.…”

“…Health care costs for patients with leukodystrophy average more than $100,000, and in some patients are higher than $1 million. 4 However, a definitive diagnosis is reached in only approximately 50% of patients. 1,5 Newer testing approaches based on next-generation sequencing may offer higher testing yields in a shorter time.…”

“…Complete cost data were extracted for each patient from IH's activity-based cost accounting system and were standardized to 2013 constant US dollars. 4 We did not include costs for hospital care or professional fees.…”

Comprehensive population-based determination of pediatric multiple sclerosis health care costs

“…The definition of a high-cost group is based on prior cost studies with skewed (non-normal) distributions. 7,9 High-cost patients averaged higher costs per encounter, more MRIs, a greater number of days spent receiving care, and had a longer average length of time in the study. It is unclear whether extending the study period would result in the same disparities.…”

“…The EDW was queried using unique identifiers for each patient to obtain comprehensive cost information pertaining to imaging, laboratory, pharmacy, clinic visits, hospital stays, and amount of time spent receiving care. Costs were categorized by type (diagnostics, therapeutics, room overhead) as well as by setting (outpatient, inpatient, emergency department), and were adjusted to 2014 US dollars (see Nelson et al 7 for detailed definitions of cost groups). The EDW does not maintain data for professional practitioner fees, which were therefore not included in total costs.…”