{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,29]],"date-time":"2025-12-29T19:07:55Z","timestamp":1767035275631},"reference-count":38,"publisher":"Springer Science and Business Media LLC","issue":"2","license":[{"start":{"date-parts":[[2019,6,7]],"date-time":"2019-06-07T00:00:00Z","timestamp":1559865600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/www.springer.com\/tdm"},{"start":{"date-parts":[[2019,6,7]],"date-time":"2019-06-07T00:00:00Z","timestamp":1559865600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.springer.com\/tdm"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Scientometrics"],"published-print":{"date-parts":[[2019,8]]},"DOI":"10.1007\/s11192-019-03129-5","type":"journal-article","created":{"date-parts":[[2019,6,7]],"date-time":"2019-06-07T17:28:28Z","timestamp":1559928508000},"page":"361-374","update-policy":"http:\/\/dx.doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Comparative analysis of correlations of research and development indicators for rare diseases among Japan, the US, and Europe"],"prefix":"10.1007","volume":"120","author":[{"given":"Hirokuni","family":"Mizoguchi","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shingo","family":"Kano","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2019,6,7]]},"reference":[{"key":"3129_CR1","doi-asserted-by":"publisher","first-page":"1299","DOI":"10.1038\/nature04226","volume":"437","author":"D Altshuler","year":"2005","unstructured":"Altshuler, D., Brooks, L. D., Chakravarti, A., Collins, F. S., Daly, M. J., Donnelly, P., et al. (2005). A haplotype map of the human genome. Nature,\n                           437, 1299\u20131320.","journal-title":"Nature"},{"issue":"1","key":"3129_CR2","doi-asserted-by":"publisher","first-page":"41","DOI":"10.1007\/s11192-016-1952-z","volume":"108","author":"CB Amat","year":"2016","unstructured":"Amat, C. B., & Perruchas, F. (2016). Evolving cohesion metrics of a research network on rare diseases: a longitudinal study over 14 years. Scientometrics,\n                           108(1), 41\u201356.","journal-title":"Scientometrics"},{"key":"3129_CR3","doi-asserted-by":"crossref","unstructured":"Ayme, S., & Schmidtke, J. (2007). Networking for rare diseases: a necessity for Europe. Bundesgesundheitsblatt-Gesundheitsforschung-Gesundheitsschutz, 50(12), 1477-1483.","DOI":"10.1007\/s00103-007-0381-9"},{"key":"3129_CR4","doi-asserted-by":"crossref","unstructured":"Bell, S.A., & Smith, C.T. (2014). A comparison of interventional clinical trials in rare versus non-rare diseases: an analysis of ClinicalTrials.gov. Orphanet Journal of Rare Diseases, 9, 170.","DOI":"10.1186\/s13023-014-0170-0"},{"issue":"1","key":"3129_CR5","first-page":"298","volume":"57","author":"Y Benjamini","year":"1995","unstructured":"Benjamini, Y., & Hochberg, Y. (1995). Controlling the false discovery rate: a practical and powerful approach to multiple testing. Journal of the Royal Statistical Society: Series B (Methodological),\n                           57(1), 298\u2013300.","journal-title":"Journal of the Royal Statistical Society: Series B (Methodological)"},{"issue":"7","key":"3129_CR6","doi-asserted-by":"publisher","first-page":"519","DOI":"10.1038\/nrd3160","volume":"9","author":"MM Braun","year":"2010","unstructured":"Braun, M. M., Farag-El-Massah, S., Xu, K., & Cote, T. R. (2010). Emergence of orphan drugs in the United States: a quantitative assessment of the first 25 years. Nature Reviews Drug Discovery,\n                           9(7), 519\u2013522.","journal-title":"Nature Reviews Drug Discovery"},{"key":"3129_CR7","volume-title":"Open innovation: The new imperative for creating and profiting from technology","author":"H Chesbrough","year":"2003","unstructured":"Chesbrough, H. (2003). Open innovation: The new imperative for creating and profiting from technology. Boston: Harvard Business School Press."},{"key":"3129_CR8","doi-asserted-by":"publisher","first-page":"56","DOI":"10.1016\/j.socscimed.2013.06.019","volume":"94","author":"AS Desser","year":"2013","unstructured":"Desser, A. S. (2013). Prioritizing treatment of rare diseases: A survey of preferences of Norwegian doctors. Social Science and Medicine,\n                           94, 56\u201362.","journal-title":"Social Science and Medicine"},{"issue":"2","key":"3129_CR9","doi-asserted-by":"publisher","first-page":"338","DOI":"10.1096\/fj.07-9492LSF","volume":"22","author":"ME Falagas","year":"2008","unstructured":"Falagas, M. E., Pitsouni, E. I., Malietzis, G. A., & Pappas, G. (2008). Comparison of PubMed, Scopus, Web of Science, and Google Scholar: Strengths and weaknesses. The FASEB Journal,\n                           22(2), 338\u2013342.","journal-title":"The FASEB Journal"},{"key":"3129_CR10","doi-asserted-by":"publisher","first-page":"53","DOI":"10.3389\/fmed.2017.00053","volume":"4","author":"AM Farkas","year":"2017","unstructured":"Farkas, A. M., Mariz, S., Stoyanova-Beninska, V., Celis, P., Vamvakas, S., Larsson, K., et al. (2017). Advanced therapy medicinal products for rare diseases: State of play of incentives supporting development in Europe. Frontiers in Medicine,\n                           4, 53.","journal-title":"Frontiers in Medicine"},{"key":"3129_CR11","volume-title":"Rare diseases and orphan products: Accelerating research and development","year":"2010","unstructured":"Field, M. J., & Boat, T. F. (Eds.). (2010). Rare diseases and orphan products: Accelerating research and development. Washington, D.C.: Institute of Medicine, The National Academies Press."},{"issue":"3\u20134","key":"3129_CR12","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1016\/j.drudis.2012.08.009","volume":"18","author":"P Franco","year":"2013","unstructured":"Franco, P. (2013). Orphan drugs: The regulatory environment. Drug Discovery Today,\n                           18(3\u20134), 163\u2013172.","journal-title":"Drug Discovery Today"},{"issue":"6","key":"3129_CR13","first-page":"840","volume":"43","author":"F Galati","year":"2016","unstructured":"Galati, F., & Bigliardi, B. (2016). The unintended effect of the Orphan Drug Act on the adoption of open innovation. Science and Public Policy,\n                           43(6), 840\u2013848.","journal-title":"Science and Public Policy"},{"issue":"1","key":"3129_CR14","doi-asserted-by":"publisher","first-page":"64","DOI":"10.1186\/s13023-017-0617-1","volume":"12","author":"V Giannuzzi","year":"2017","unstructured":"Giannuzzi, V., Conte, R., Landi, A., Ottomano, S. A., Bonifazi, D., Baiardi, P., et al. (2017a). Orphan medicinal products in Europe and United States to cover needs of patients with rare diseases: An increased common effort is to be foreseen. Orphanet Journal of Rare Diseases,\n                           12(1), 64.","journal-title":"Orphanet Journal of Rare Diseases"},{"issue":"9","key":"3129_CR15","first-page":"e017358","volume":"7","author":"V Giannuzzi","year":"2017","unstructured":"Giannuzzi, V., Landi, A., Bosone, E., Giannuzzi, F., Nicotri, S., Torrent-Farnell, J., et al. (2017b). Failures to further developing orphan medicinal products after designation granted in Europe: An analysis of marketing authorisation failures and abandoned drugs. British Medical Journal Open,\n                           7(9), e017358.","journal-title":"British Medical Journal Open"},{"issue":"8","key":"3129_CR16","doi-asserted-by":"publisher","first-page":"850","DOI":"10.1016\/j.jclinepi.2014.03.012","volume":"67","author":"ME Glickman","year":"2014","unstructured":"Glickman, M. E., Rao, S. R., & Schultz, M. R. (2014). False discovery rate control is a recommended alternative to Bonferroni-type adjustments in health studies. Journal of Clinical Epidemiology,\n                           67(8), 850\u2013857.","journal-title":"Journal of Clinical Epidemiology"},{"issue":"15\u201316","key":"3129_CR17","doi-asserted-by":"publisher","first-page":"670","DOI":"10.1016\/j.drudis.2008.05.001","volume":"13","author":"HE Heemstra","year":"2008","unstructured":"Heemstra, H. E., de Vrueh, R. L. A., van Weely, S., Buller, H. A., & Leufkens, H. G. M. (2008). Orphan drug development across Europe: Bottlenecks and opportunities. Drug Discovery Today,\n                           13(15\u201316), 670\u2013676.","journal-title":"Drug Discovery Today"},{"issue":"23\u201324","key":"3129_CR18","doi-asserted-by":"publisher","first-page":"1166","DOI":"10.1016\/j.drudis.2009.09.008","volume":"14","author":"HE Heemstra","year":"2009","unstructured":"Heemstra, H. E., van Weely, S., Buller, H. A., Leufkens, H. G. M., & de Vrueh, R. L. A. (2009). Translation of rare disease research into orphan drug development: Disease matters. Drug Discovery Today,\n                           14(23\u201324), 1166\u20131173.","journal-title":"Drug Discovery Today"},{"key":"3129_CR19","doi-asserted-by":"publisher","DOI":"10.1533\/9781908818393","volume-title":"Orphan drugs: Understanding the rare disease market and its dynamics","author":"E Hernberg-St\u00e5hl","year":"2013","unstructured":"Hernberg-St\u00e5hl, E., & Reljanovi\u0107, M. (2013). Orphan drugs: Understanding the rare disease market and its dynamics. Sawston: Woodhead Publishing."},{"issue":"1","key":"3129_CR20","doi-asserted-by":"publisher","first-page":"383","DOI":"10.1007\/s11192-015-1648-9","volume":"106","author":"M Igami","year":"2016","unstructured":"Igami, M., & Saka, A. (2016). Decreasing diversity in Japanese science, evidence from in-depth analyses of science maps. Scientometrics,\n                           106(1), 383\u2013403.","journal-title":"Scientometrics"},{"key":"3129_CR21","unstructured":"Japan Agency for Medical Research and Development. (2017). Support program for orphan drug prior to the designation. \n                              https:\/\/www.amed.go.jp\/en\/program\/list\/06\/03\/001_03-01.html\n                              \n                           . Accessed Apr 2018."},{"issue":"4","key":"3129_CR22","doi-asserted-by":"publisher","first-page":"1009","DOI":"10.1007\/s00228-012-1423-2","volume":"69","author":"R Joppi","year":"2013","unstructured":"Joppi, R., Bertele, V., & Garattini, S. (2013). Orphan drugs, orphan diseases. The first decade of orphan drug legislation in the EU. European Journal of Clinical Pharmacology,\n                           69(4), 1009\u20131024.","journal-title":"European Journal of Clinical Pharmacology"},{"issue":"9","key":"3129_CR23","doi-asserted-by":"publisher","first-page":"562","DOI":"10.1038\/gt.2017.29","volume":"24","author":"D Julkowska","year":"2017","unstructured":"Julkowska, D., Austin, C. P., Cutillo, C. M., Gancberg, D., Hager, C., Halftermeyer, J., et al. (2017). The importance of international collaboration for rare diseases research: A European perspective. Gene Therapy,\n                           24(9), 562\u2013571.","journal-title":"Gene Therapy"},{"issue":"1","key":"3129_CR24","first-page":"41","volume":"4","author":"K Kaneko","year":"2014","unstructured":"Kaneko, K., Yamada, T., Kashitani, Y., Sato, Y., Arai, M., & Satou, H. (2014). Proposals from the pharmaceutical industry on the development and promotion of orphan drugs. Regulatory Science of Medical Products,\n                           4(1), 41\u201348. (in Japanese).","journal-title":"Regulatory Science of Medical Products"},{"issue":"11","key":"3129_CR25","doi-asserted-by":"publisher","first-page":"867","DOI":"10.1038\/nrd3251","volume":"9","author":"R Kneller","year":"2010","unstructured":"Kneller, R. (2010). The importance of new companies for drug discovery: Origins of a decade of new drugs. Nature Reviews Drug Discovery,\n                           9(11), 867\u2013882.","journal-title":"Nature Reviews Drug Discovery"},{"issue":"1","key":"3129_CR26","doi-asserted-by":"publisher","first-page":"343","DOI":"10.1007\/s11192-012-0776-8","volume":"94","author":"B Lander","year":"2013","unstructured":"Lander, B. (2013). Sectoral collaboration in biomedical research and development. Scientometrics,\n                           94(1), 343\u2013357.","journal-title":"Scientometrics"},{"issue":"9970","key":"3129_CR27","doi-asserted-by":"publisher","first-page":"750","DOI":"10.1016\/S0140-6736(15)60297-5","volume":"385","author":"L Luzzatto","year":"2015","unstructured":"Luzzatto, L., Hollak, C. E. M., Cox, T. M., Schieppati, A., Licht, C., Kaariainen, H., et al. (2015). Rare diseases and effective treatments: Are we delivering? The Lancet,\n                           385(9970), 750\u2013752.","journal-title":"The Lancet"},{"issue":"1","key":"3129_CR28","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1016\/S0048-7333(99)00030-X","volume":"29","author":"GS McMillan","year":"2000","unstructured":"McMillan, G. S., Narin, F., & Deeds, D. L. (2000). An analysis of the critical role of public science in innovation: The case of biotechnology. Research Policy,\n                           29(1), 1\u20138.","journal-title":"Research Policy"},{"issue":"10","key":"3129_CR29","doi-asserted-by":"publisher","first-page":"1681","DOI":"10.1016\/j.drudis.2016.06.014","volume":"21","author":"H Mizoguchi","year":"2016","unstructured":"Mizoguchi, H., Yamanaka, T., & Kano, S. (2016). Research and drug development activities in rare diseases: Differences between Japan and Europe regarding influence of prevalence. Drug Discovery Today,\n                           21(10), 1681\u20131689.","journal-title":"Drug Discovery Today"},{"issue":"5","key":"3129_CR30","doi-asserted-by":"publisher","first-page":"599","DOI":"10.1248\/yakushi.13-00256-4","volume":"134","author":"H Mizushima","year":"2014","unstructured":"Mizushima, H., Tanabe, M., & Kanatani, Y. (2014). Patient database and orphan drug development. Yakugaku Zasshi,\n                           134(5), 599\u2013605. (in Japanese).","journal-title":"Yakugaku Zasshi"},{"key":"3129_CR31","first-page":"130","volume":"31","author":"Y Murakami","year":"2016","unstructured":"Murakami, Y. (2016). Achievement of research on international research collaboration and suggestion to policy in Japan. The Journal of Science Policy and Research Management,\n                           31, 130\u2013144. (in Japanese).","journal-title":"The Journal of Science Policy and Research Management"},{"issue":"4","key":"3129_CR32","doi-asserted-by":"publisher","first-page":"544","DOI":"10.1016\/j.drudis.2016.02.016","volume":"21","author":"M Murakami","year":"2016","unstructured":"Murakami, M., & Narukawa, M. (2016). Matched analysis on orphan drug designations and approvals: Cross regional analysis in the United States, the European Union, and Japan. Drug Discovery Today,\n                           21(4), 544\u2013549.","journal-title":"Drug Discovery Today"},{"key":"3129_CR33","doi-asserted-by":"publisher","first-page":"94","DOI":"10.1186\/s12984-016-0205-y","volume":"13","author":"G Pal","year":"2016","unstructured":"Pal, G., O\u2019Keefe, J., Robertson-Dick, E., Bernard, B., Anderson, S., & Hall, D. (2016). Global cognitive function and processing speed are associated with gait and balance dysfunction in Parkinson\u2019s disease. Journal of NeuroEngineering and Rehabilitation,\n                           13, 94.","journal-title":"Journal of NeuroEngineering and Rehabilitation"},{"issue":"2","key":"3129_CR34","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1517\/21678707.2013.749181","volume":"1","author":"S Patel","year":"2013","unstructured":"Patel, S., & Needleman, K. I. M. (2013). Funding grants for rare diseases at the food and drug administration. Expert Opinion on Orphan Drugs,\n                           1(2), 99\u2013102.","journal-title":"Expert Opinion on Orphan Drugs"},{"issue":"1","key":"3129_CR35","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1080\/21678707.2013.752128","volume":"1","author":"MI Phillips","year":"2013","unstructured":"Phillips, M. I. (2013). Big Pharma\u2019s new model in orphan drugs and rare diseases. Expert Opinion on Orphan Drugs,\n                           1(1), 1\u20133.","journal-title":"Expert Opinion on Orphan Drugs"},{"issue":"3","key":"3129_CR36","doi-asserted-by":"publisher","first-page":"191","DOI":"10.1038\/nrd3681","volume":"11","author":"JW Scannell","year":"2012","unstructured":"Scannell, J. W., Blanckley, A., Boldon, H., & Warrington, B. (2012). Diagnosing the decline in pharmaceutical R&D efficiency. Nature Reviews Drug Discovery,\n                           11(3), 191\u2013200.","journal-title":"Nature Reviews Drug Discovery"},{"issue":"12","key":"3129_CR37","doi-asserted-by":"publisher","first-page":"921","DOI":"10.1038\/nrd3275","volume":"9","author":"E Tambuyzer","year":"2010","unstructured":"Tambuyzer, E. (2010). Rare diseases, orphan drugs and their regulation: Questions and misconceptions. Nature Reviews Drug Discovery,\n                           9(12), 921\u2013929.","journal-title":"Nature Reviews Drug Discovery"},{"key":"3129_CR38","first-page":"8","volume":"32","author":"K Tatsumi","year":"2011","unstructured":"Tatsumi, K. (2011). Development trends of orphan drugs in Japan, the US and the EU and issues for rare disease innovation in Japan. OPIR News,\n                           32, 8\u201323. (in Japanese).","journal-title":"OPIR News"}],"container-title":["Scientometrics"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s11192-019-03129-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1007\/s11192-019-03129-5\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s11192-019-03129-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2020,6,5]],"date-time":"2020-06-05T23:20:02Z","timestamp":1591399202000},"score":1,"resource":{"primary":{"URL":"http:\/\/link.springer.com\/10.1007\/s11192-019-03129-5"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,6,7]]},"references-count":38,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2019,8]]}},"alternative-id":["3129"],"URL":"https:\/\/doi.org\/10.1007\/s11192-019-03129-5","relation":{},"ISSN":["0138-9130","1588-2861"],"issn-type":[{"value":"0138-9130","type":"print"},{"value":"1588-2861","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,6,7]]},"assertion":[{"value":"1 May 2018","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"7 June 2019","order":2,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}}]}}