Hummel, S.R., Sadler, S. orcid.org/0000-0001-5935-8507, Whitaker, M.J. et al. (3 more authors) (2016) A Model for Measuring the Health Burden of Classic Congenital Adrenal Hyperplasia in Adults. Clinical Endocrinology. ISSN 0300-0664
Abstract
Aim Patients with classic congenital adrenal hyperplasia (CAH)have poor health outcomes. In the absence of a comprehensive observational study, this manuscript provides a model to esti-mate the lifetime disease burden of adults with classic CAH. Methods The model, built in Excel, comprises subdomains addressing the health consequences of CAH and synthesises evidence from clinical and epidemiological studies on health out-comes.Results The model estimates that adults with classic CAH will implement ‘sick day rules’ (doubling or tripling glucocorticoid and/or use of parenteral therapy) 171 times over their lifetime and attend hospital for adrenal crisis on 11 occasions. In a population of 1000, over 200 will die of a condition complicated by adrenal crisis resulting, on average, in a loss of 7 years of life.Patients with CAH may also suffer from excess CVD events.Treatment with glucocorticoids almost doubles the risk of bone fractures in patients with CAH compared to the general popula-tion, leading on average to an additional 08 fractures perpatient with CAH over their lifetime.Conclusions The disease burden model highlights gaps in evidence, particularly regarding intensity of care and adrenal crisis,and the relationship between control of CAH and risks of CVD, osteoporosis, diabetes and infertility. The model can be used for research on the impact of new clinical pathways and therapeutic interventions in terms of clinical events and cost.
Metadata
Item Type: | Article |
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Authors/Creators: |
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Copyright, Publisher and Additional Information: | © 2016 The Authors. Clinical Endocrinology published by John Wiley & Sons Ltd. This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License (https://creativecommons.org/licenses/by-nc-nd/4.0/), which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made. |
Dates: |
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Institution: | The University of Sheffield |
Academic Units: | The University of Sheffield > Faculty of Medicine, Dentistry and Health (Sheffield) > Department of Human Metabolism (Sheffield) The University of Sheffield > Faculty of Medicine, Dentistry and Health (Sheffield) > School of Health and Related Research (Sheffield) > ScHARR - Sheffield Centre for Health and Related Research The University of Sheffield > Sheffield Teaching Hospitals |
Depositing User: | Symplectic Sheffield |
Date Deposited: | 20 Jul 2016 09:29 |
Last Modified: | 20 Jul 2016 09:29 |
Published Version: | http://dx.doi.org/10.1111/cen.13060 |
Status: | Published |
Publisher: | Wiley |
Refereed: | Yes |
Identification Number: | 10.1111/cen.13060 |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:101557 |
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