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J Pediatr. 2013 Mar;162(3):624-628.e1. doi: 10.1016/j.jpeds.2012.08.046. Epub 2012 Oct 13.

Determinants of health care use in a population-based leukodystrophy cohort.

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  • 1Department of Pediatrics, Division of Pediatric Neurology, University of Utah School of Medicine, Salt Lake City, UT 84108, USA.



To determine the costs for children with leukodystrophies and whether high costs are associated with characteristic clinical features or resources use.


We determined health care costs in a population cohort of 122 patients with leukodystrophies, including inpatient, outpatient, and emergency department use, during a 9-year period. We analyzed differences in patients with high costs (>85th percentile) and their health care use.


Patients with leukodystrophy had significant variability in resource use, 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.


Inpatient hospitalization is the greatest source of health care resource use in patients with leukodystrophies. A minority of patients account for the majority of costs, primarily attributable 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.

Copyright © 2013 Mosby, Inc. All rights reserved.

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