Printed in U.S.A. EFFICIENT SELECTION OF CONTROLS FOR MULTI-CENTERED COLLABORATIVE STUDIES OF RARE DISEASES1

Abstract

WA 98104), A. Whitehead, and E. Noonan. Efficient selection of controls for multi-centered collaborative studies of rare diseases. Am J Epidemiol 1986;123:901-904. An increasing number of epidemiologic studies of rare diseases are being conducted as collaborative ventures among several institutions in different geo-graphic areas. Geographic area must be accounted for in the analysis, along with age and other potential confounders. This paper shows that it may not be possible to accurately predict the distribution of persons with rare diseases by age and geographic area. Therefore, selecting controls by matching on age group and geographic area is more efficient than selecting controls in all age and geographic area strata based on prior estimates of expected numbers of cases. epidemiologic methods; retrospective studies; sampling studies In recent years, epidemiologists from in-stitutions in diverse geographic areas have collaborated to investigate causes of rare diseases. Since a large number of years of case accrual would otherwise be necessary to obtain an adequate sample size for a case-control study of a rare disease, the collaborative study offers the benefit of ex-pediency. The collaborative study design, however, brings with it an additional prob-lem in efficiency in control selection. Spe-cifically, it is not possible to accurately predict the age-geographic area distribution Received for publication April 26,1985, and in fina

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