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Retrospective estimation of the birth prevalence for delayed onset disorders: application to cystic fibrosis in Nova Scotia
Author(s) -
Chong Mei Yin,
Hamilton David C.,
Cole David E. C.
Publication year - 2000
Publication title -
statistics in medicine
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.996
H-Index - 183
eISSN - 1097-0258
pISSN - 0277-6715
DOI - 10.1002/(sici)1097-0258(20000315)19:5<743::aid-sim338>3.0.co;2-f
Subject(s) - nova scotia , medicine , cystic fibrosis , pediatrics , maximum likelihood , estimation , demography , statistics , geography , mathematics , management , archaeology , sociology , economics
Maximum likelihood methods are used to estimate the birth prevalence, the age‐at‐diagnosis distribution, and the number of infants born with CF in Nova Scotia over a 20 year period. The data were taken from a registry of CF cases, and, because of the delayed onset of symptoms, some CF affected individuals were not diagnosed by the end of the study period. A goodness‐of‐fit test for assessing whether the age‐at‐diagnosis distribution is stable over the 20 years is provided. These methods could be applied equally well to data on any genetic disorder with delayed onset. Copyright © 2000 John Wiley & Sons, Ltd.