Farrer L A, Cupples L A
Department of Neurology, Boston University School of Medicine, MA 02118.
Am J Hum Genet. 1994 Feb;54(2):374-83.
Alzheimer disease (AD) is neuropsychiatric illness caused by multiple etiologies. Prediction of whether AD is genetically based in a given family is problematic because of censoring bias among unaffected relatives as a consequence of the late onset of the disorder, diagnostic uncertainties, heterogeneity, and limited information in a single family. We have developed a method based on Bayesian probability to compute values for a continuous variable that ranks AD families as having a major gene form of AD (MGAD). In addition, we have compared the Bayesian method with a maximum-likelihood approach. These methods incorporate sex- and age-adjusted risk estimates and allow for phenocopies and familial clustering of age at onset. Agreement is high between the two approaches for ranking families as MGAD (Spearman rank [r] = .92). When either method is used, the numerical outcomes are sensitive to assumptions of the gene frequency and cumulative incidence of the disease in the population. Consequently, risk estimates should be used cautiously for counseling purposes; however, there are numerous valid applications of these procedures in genetic and epidemiological studies.
阿尔茨海默病(AD)是一种由多种病因引起的神经精神疾病。由于该疾病发病较晚,未受影响亲属中存在截尾偏倚、诊断不确定性、异质性以及单个家庭信息有限等原因,预测AD在特定家庭中是否基于遗传存在问题。我们开发了一种基于贝叶斯概率的方法,用于计算一个连续变量的值,该变量将AD家族列为具有AD的主要基因形式(MGAD)。此外,我们还将贝叶斯方法与最大似然法进行了比较。这些方法纳入了性别和年龄调整后的风险估计,并考虑了表型模拟和发病年龄的家族聚集性。两种将家族列为MGAD的方法之间的一致性很高(斯皮尔曼等级相关系数[r]=0.92)。当使用任何一种方法时,数值结果对人群中基因频率和疾病累积发病率的假设都很敏感。因此,出于咨询目的,应谨慎使用风险估计;然而,这些程序在遗传和流行病学研究中有许多有效的应用。