A conceptual framework for a long-term economic model for the treatment of attention-deficit/hyperactivity disorder

ABSTRACT

Background: Models incorporating long-term outcomes (LTOs) are not available to assess the health economic impact of attention-deficit/hyperactivity disorder (ADHD).

Objective: Develop a conceptual modelling framework capable of assessing long-term economic impact of ADHD therapies.

Methods: Literature was reviewed; a conceptual structure for the long-term model was outlined with attention to disease characteristics and potential impact of treatment strategies.

Results: The proposed model has four layers: i) multi-state short-term framework to differentiate between ADHD treatments; ii) multiple states being merged into three core health states associated with LTOs; iii) series of sub-models in which particular LTOs are depicted; iv) outcomes collected to be either used directly for economic analyses or translated into other relevant measures.

Conclusions: This conceptual model provides a framework to assess relationships between short- and long-term outcomes of the disease and its treatment, and to estimate the economic impact of ADHD treatments throughout the course of the disease.

KEYWORDS:

• ADHD
• long-term model
• conceptual model framework
• long-term disease outcomes
• ADHD therapy

Acknowledgments

Editorial assistance in formatting, proofreading and copy editing the manuscript, and coordination and collation of comments, was provided by Caudex (Oxford, UK), funded by Shire International GmbH.

Declaration of interest

J Setyawan was an employee of Shire and owned stock/stock options at the time of the study. B Nagy, T Soroncz-Szabo and Z Kaló are employed or contracted by Syreon Research Institute, which received funding for this research from Shire, including costs of travelling to an advisory board meeting and giving a lecture, and manuscript preparation. D Coghill reports receiving grants/grants pending from Shire and the European Union, consulting fees/honararia from Shire, Novartis and Sandoz, support to travel to meetings for the study from Shire, provision of writing assistance, medicines, equipment or administrative support from Shire, payment for lectures including Speakers Bureaus from Shire, Eli Lilly, Janssen Cilag and Novartis, expert testimony from GlaxoSmithKline, and royalties from Oxford University Press. JA Doshi has received research funding from Pfizer, Janssen, PhRMA, Sanofi, Amgen, Biogen, Regeneron, Biogen, Regeneron, National Pharmaceutical Council and Humana; and received consulting fees from Merck & Co. Inc, Boehringer Ingelheim, Shire, Alkermes, Vertex, Allergan, and Ironwood Pharmaceuticals; and reports spouse owning stock in Merck & Co. Inc and Pfizer Inc. The authors have no other relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript apart from those disclosed.

Supplemental data

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Additional information

Funding

This research was funded by Shire Development, LLC. Although employees of Shire were involved in the design, collection, analysis, interpretation, and fact checking of information, the content of this manuscript, the interpretation of the data, and the decision to submit the manuscript for publication was made by the authors independently. Publication of study results was not contingent on Shire’s approval or censorship.

Notes on contributors

Balázs Nagy

B. Nagy, T. Soroncz-Szabo and Z. Kalo reviewed the literature, synthesized the evidence and prepared the conceptual structure of the model. J. Setyawan, J.A. Doshi and D. Coghill conceived the research, provided inputs for the conceptual framework during the development process, and were members of the expert panel. Every author participated in drafting and/or critically reviewing the manuscript, and approved the final submitted version. Z. Kalo is the overall guarantor for the work.