Costs of the diagnostic odyssey in children with inherited leukodystrophies

Neurology. 2015 Sep 29;85(13):1167-70. doi: 10.1212/WNL.0000000000001974. Epub 2015 Aug 28.

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.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Child
  • Female
  • Health Care Costs*
  • Hereditary Central Nervous System Demyelinating Diseases / diagnosis*
  • Hereditary Central Nervous System Demyelinating Diseases / economics*
  • Humans
  • Leukodystrophy, Globoid Cell / diagnosis
  • Leukodystrophy, Globoid Cell / economics
  • Leukodystrophy, Metachromatic / diagnosis
  • Leukodystrophy, Metachromatic / economics
  • Male
  • Pelizaeus-Merzbacher Disease / diagnosis
  • Pelizaeus-Merzbacher Disease / economics
  • Retrospective Studies