Jackson Richards1, E Kent Korgenski1, Rajendu Srivastava1, Joshua L Bonkowsky2. 1. From the Division of Inpatient Medicine, Department of Pediatrics (R.S.), and Division of Pediatric Neurology, Department of Pediatrics (J.L.B.), University of Utah School of Medicine (J.R.), Salt Lake City; Institute for Health Care Delivery Research (R.S.), Intermountain Healthcare (E.K.K.), Salt Lake City, UT. 2. From the Division of Inpatient Medicine, Department of Pediatrics (R.S.), and Division of Pediatric Neurology, Department of Pediatrics (J.L.B.), University of Utah School of Medicine (J.R.), Salt Lake City; Institute for Health Care Delivery Research (R.S.), Intermountain Healthcare (E.K.K.), Salt Lake City, UT. joshua.bonkowsky@hsc.utah.edu.
Abstract
OBJECTIVES: Our objective was to determine the extent of testing and costs solely related to diagnosis (the diagnostic odyssey) in a cohort of children with inherited leukodystrophies. METHODS: We determined all inpatient and outpatient laboratory testing, including brain MRIs obtained for the purpose of diagnosis, over an 8-year time period in a retrospective population cohort of children with inherited leukodystrophies. Costs were determined from an activity-based cost accounting system and were standardized to 2013 constant US dollars. RESULTS: Each patient had on average 20 tests (range 2-42 tests), with costs of $4,200 (range $357-$15,611). Diagnostic yield plateaued after 25 tests, and costs increased significantly after 32 tests. Fifty-three percent of patients were diagnosed in 20 or fewer tests, compared with 17% if more than 20 tests were performed. CONCLUSIONS: Our findings provide details on the amount and costs of testing in children who often undergo a diagnostic odyssey. Our results suggest that diagnostic testing is a relatively modest contributor to the overall health care costs in patients with leukodystrophy, and offer insights into the diagnostic odyssey of children with neurologic impairment.
OBJECTIVES: Our objective was to determine the extent of testing and costs solely related to diagnosis (the diagnostic odyssey) in a cohort of children with inherited leukodystrophies. METHODS: We determined all inpatient and outpatient laboratory testing, including brain MRIs obtained for the purpose of diagnosis, over an 8-year time period in a retrospective population cohort of children with inherited leukodystrophies. Costs were determined from an activity-based cost accounting system and were standardized to 2013 constant US dollars. RESULTS: Each patient had on average 20 tests (range 2-42 tests), with costs of $4,200 (range $357-$15,611). Diagnostic yield plateaued after 25 tests, and costs increased significantly after 32 tests. Fifty-three percent of patients were diagnosed in 20 or fewer tests, compared with 17% if more than 20 tests were performed. CONCLUSIONS: Our findings provide details on the amount and costs of testing in children who often undergo a diagnostic odyssey. Our results suggest that diagnostic testing is a relatively modest contributor to the overall health care costs in patients with leukodystrophy, and offer insights into the diagnostic odyssey of children with neurologic impairment.
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