Issues, Challenges and Opportunities for Economic Evaluations of Orphan Drugs in Rare Diseases: An Umbrella Review

Author:

Grand Tobias SydendalORCID,Ren ShijieORCID,Hall JamesORCID,Åström Daniel OudinORCID,Regnier StephaneORCID,Thokala PraveenORCID

Funder

Innovationsfonden

Publisher

Springer Science and Business Media LLC

Reference83 articles.

1. Grand T, Ren S, Thokala P, Oudin Åström D, Regnier S, Hall J. Issues, challenges and opportunities for economic evaluation of orphan drugs: an umbrella review protocol. 2023. The University of Sheffield’s research data repository. 2023. https://doi.org/10.15131/shef.data.23390060.v1.

2. Richter T, Nestler-Parr S, Babela R, Khan ZM, Tesoro T, Molsen E, Hughes DA. Rare disease terminology and definitions: a systematic global review: report of the ISPOR Rare Disease Special Interest Group. Value Health. 2015;18(6):906–14. https://doi.org/10.1016/j.jval.2015.05.008.

3. The European Parliament. Regulation (EC) No 141/2000 of the European Parliament and of the Council of 16 December 1999 on orphan medicinal products. 2000. https://eur-lex.europa.eu/legal-content/EN/TXT/PDF/?uri=CELEX:32000R0141&from=EN. Accessed 20 Oct 2022.

4. Federal Food, Drug and Cosmetic Act. Orphan Drug Act. In: Senate and house of representatives of the United States of America in Congress, editor. 97th Congress: Public Law 97–114; 1983.

5. Thomas S, Caplan A. The orphan drug act revisited. JAMA. 2019;321(9):833–4. https://doi.org/10.1001/jama.2019.0290.

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