Title |
Reducing selection bias in case-control studies from rare disease registries
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Published in |
Orphanet Journal of Rare Diseases, September 2011
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DOI | 10.1186/1750-1172-6-61 |
Pubmed ID | |
Authors |
J Alexander Cole, John S Taylor, Thomas N Hangartner, Neal J Weinreb, Pramod K Mistry, Aneal Khan |
Abstract |
In clinical research of rare diseases, where small patient numbers and disease heterogeneity limit study design options, registries are a valuable resource for demographic and outcome information. However, in contrast to prospective, randomized clinical trials, the observational design of registries is prone to introduce selection bias and negatively impact the validity of data analyses. The objective of the study was to demonstrate the utility of case-control matching and the risk-set method in order to control bias in data from a rare disease registry. Data from the International Collaborative Gaucher Group (ICGG) Gaucher Registry were used as an example. |
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Unknown | 2 | 100% |
Demographic breakdown
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Members of the public | 2 | 100% |
Mendeley readers
Geographical breakdown
Country | Count | As % |
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Germany | 2 | 3% |
Unknown | 65 | 97% |
Demographic breakdown
Readers by professional status | Count | As % |
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Student > Master | 16 | 24% |
Researcher | 12 | 18% |
Student > Bachelor | 7 | 10% |
Student > Ph. D. Student | 4 | 6% |
Student > Doctoral Student | 3 | 4% |
Other | 10 | 15% |
Unknown | 15 | 22% |
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Mathematics | 6 | 9% |
Pharmacology, Toxicology and Pharmaceutical Science | 6 | 9% |
Agricultural and Biological Sciences | 3 | 4% |
Other | 15 | 22% |
Unknown | 16 | 24% |