Division of Medical Genetics, Department of Medicine, University of Washington, Seattle, WA, USA.
J Lipid Res. 2011 Oct;52(10):1837-46. doi: 10.1194/jlr.P016576. Epub 2011 Jul 13.
Phospholipid transfer protein activity (PLTPa) is associated with insulin levels and has been implicated in atherosclerotic disease in both mice and humans. Variation at the PLTP structural locus on chromosome 20 explains some, but not all, heritable variation in PLTPa. In order to detect quantitative trait loci (QTLs) elsewhere in the genome that affect PLTPa, we performed both oligogenic and single QTL linkage analysis on four large families (n = 227 with phenotype, n = 330 with genotype, n = 462 total), ascertained for familial combined hyperlipidemia. We detected evidence of linkage between PLTPa and chromosome 19p (lod = 3.2) for a single family and chromosome 2q (lod = 2.8) for all families. Inclusion of additional marker and exome sequence data in the analysis refined the linkage signal on chromosome 19 and implicated coding variation in LASS4, a gene regulated by leptin that is involved in ceramide synthesis. Association between PLTPa and LASS4 variation was replicated in the other three families (P = 0.02), adjusting for pedigree structure. To our knowledge, this is the first example for which exome data was used in families to identify a complex QTL that is not the structural locus.
磷脂转移蛋白活性(PLTPa)与胰岛素水平相关,并且在小鼠和人类的动脉粥样硬化疾病中都有涉及。20 号染色体上的 PLTP 结构基因座的变异可以解释部分,但不是全部,PLTPa 的遗传变异。为了检测影响 PLTPa 的基因组其他部位的数量性状基因座(QTL),我们对四个大家族(表型 n = 227,基因型 n = 330,总计 n = 462)进行了多基因和单 QTL 连锁分析,这些家族是为家族性混合性高脂血症而确定的。我们在单个家族中检测到 PLTPa 与 19p 染色体(lod = 3.2)之间存在连锁证据,在所有家族中检测到 PLTPa 与 2q 染色体(lod = 2.8)之间存在连锁证据。在分析中纳入额外的标记和外显子序列数据,使 19 号染色体上的连锁信号得到了细化,并暗示了 LASS4 编码变异,LASS4 是一种受瘦素调节的基因,参与神经酰胺合成。PLTPa 与 LASS4 变异之间的关联在其他三个家族中得到了复制(调整家系结构后,P = 0.02)。据我们所知,这是首次在家族中使用外显子数据来识别不是结构基因座的复杂 QTL 的例子。