Liang K Y, Hsu F C, Beaty T H, Barnes K C
Department of Biostatistics, Johns Hopkins University, Baltimore, MD 21205, USA.
Am J Hum Genet. 2001 Apr;68(4):937-50. doi: 10.1086/319504. Epub 2001 Mar 15.
In the present study we propose a multipoint approach, for the mapping of genes, that is based on the case-parent trio design. We first derive an expression for the expected preferential-allele-transmission statistics for transmission, from either parent to an affected child, for an arbitrary location within a chromosomal region demarcated by several genetic markers. No assumption about genetic mechanism is needed in this derivation, beyond the assumption that no more than one disease gene lies in the region framed by the markers. When one builds on this representation, the way in which one may maximize the genetic information from multiple markers becomes obvious. This proposed method differs from the popular transmission/disequilibrium test (TDT) approach for fine mapping, in the following ways: First, in contrast with the TDT approach, all markers contribute information, regardless of whether the parents are heterozygous at any one marker, and incomplete trio data can be utilized in our approach. Second, rather than performing the TDT at each marker separately, we propose a single test statistic that follows a chi(2) distribution with 1 df, under the null hypothesis of no linkage or linkage disequilibrium to the region. Third, in the presence of linkage evidence, we offer a means to estimate the location of the disease locus along with its sampling uncertainty. We illustrate the proposed method with data from a family study of asthma, conducted in Barbados.
在本研究中,我们提出了一种基于病例-父母三联体设计的多点基因定位方法。我们首先推导了在由多个遗传标记划定的染色体区域内任意位置,从父母任一方传递给患病子女的预期优先等位基因传递统计量的表达式。在这个推导过程中,除了假设标记所界定区域内不超过一个致病基因外,无需对遗传机制做任何假设。基于这种表示形式,如何从多个标记中最大化遗传信息就变得显而易见了。所提出的这种方法与用于精细定位的流行的传递/不平衡检验(TDT)方法在以下方面有所不同:第一,与TDT方法不同,所有标记都能提供信息,无论父母在任一标记处是否为杂合子,并且我们的方法可以利用不完整的三联体数据。第二,我们不是在每个标记处分别进行TDT,而是提出一个单一的检验统计量,在与该区域无连锁或连锁不平衡的零假设下,它服从自由度为1的卡方分布。第三,在存在连锁证据的情况下,我们提供了一种方法来估计疾病位点的位置及其抽样不确定性。我们用在巴巴多斯进行的一项哮喘家族研究的数据说明了所提出的方法。