The collective impact of rare diseases in Western Australia: an estimate using a population-based cohort - Sorbonne Université Access content directly
Journal Articles Genetics in Medicine Year : 2017

The collective impact of rare diseases in Western Australia: an estimate using a population-based cohort

Caroline E Walker
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Trinity Mahede
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Geoff Davis
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Laura J Miller
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Jennifer Girschik
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Kate Brameld
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Wenxing Sun
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Ana Rath
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Stephen R Zubrick
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Gareth S Baynam
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Caron Molster
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Hugh Dawkins
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Tarun S Weeramanthri
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Hugh J.S. Dawkins
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Abstract

Purpose: It has been argued that rare diseases should be recognized as a public health priority. However, there is a shortage of epidemiological data describing the true burden of rare diseases. This study investigated hospital service use to provide a better understanding of the collective health and economic impacts of rare diseases. Methods: Novel methodology was developed using a carefully constructed set of diagnostic codes, a selection of rare disease cohorts from hospital administrative data, and advanced data-linkage technologies. Outcomes included health-service use and hospital admission costs. Results: In 2010, cohort members who were alive represented approximately 2.0% of the Western Australian population. The cohort accounted for 4.6% of people discharged from hospital and 9.9% of hospital discharges, and it had a greater average length of stay than the general population. The total cost of hospital discharges for the cohort represented 10.5% of 2010 state inpatient hospital costs. Conclusions: This population-based cohort study provides strong new evidence of a marked disparity between the proportion of the population with rare diseases and their combined health-system costs. The methodology will inform future rare-disease studies, and the evidence will guide government strategies for managing the service needs of people living with rare diseases.Genet Med advance online publication 22 September 2016.
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Dates and versions

hal-04412584 , version 1 (23-01-2024)

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Caroline E Walker, Trinity Mahede, Geoff Davis, Laura J Miller, Jennifer Girschik, et al.. The collective impact of rare diseases in Western Australia: an estimate using a population-based cohort. Genetics in Medicine, 2017, 19 (5), pp.546-552. ⟨10.1038/gim.2016.143⟩. ⟨hal-04412584⟩
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