Craniofacial variability and morphological integration in mice susceptible to cleft lip and palate
Craniofacial variability and morphological integration in mice susceptible to cleft lip and palate
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DOI：
10.1111/j.0021-8782.2004.00356.x
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发表时间：
2004-12-01
影响因子：
2.4
通讯作者：
German, RZ
中科院分区：
文献类型：
--
作者：
Hallgrímsson, B;Dorval, CJ;German, RZ
A/WySnJ小鼠是一种近交系，发生唇裂伴或不伴腭裂(CL/P)，发生率为25%-30%，主要为单侧表达模式。与人类一样，不完全外显的模式以及可变和频繁的单边表达表明，在生长过程中，可变性(发育稳定性、管道化和发育整合)的调节发生了变化。我们比较了A/WySnJ小鼠、一种不发育CL/P(C57BL/6J)的品系和它们的F1杂交后代的颅面形状和大小的平均值和变异性参数。我们发现，与C57BL/6J小鼠和F1杂交小鼠相比，不表现出唇裂的成年A/WySnJ小鼠表现出颅骨的形态整合减少，整体形状和大小变化的协调受到破坏。整合度的下降最明显的是在上颚和面部。F_1代没有这种模式，说明它是由隐性遗传因素决定的。相比之下，被认为使A/WySnJ小鼠易患CL/P的品系之间的形状差异显示出一定范围的显性，这表明存在多基因基础。我们认为，颅面生长整合性降低可能是A/WySnJ小鼠CL/P的病因之一。
A/WySnJ mice are an inbred strain that develops cleft lip with or without cleft palate (CL/P) with a frequency of 25-30% and a predominantly unilateral expression pattern. As in humans, the pattern of incomplete penetrance, and variable and frequent unilateral expression suggests a role for altered regulation of variability (developmental stability, canalization and developmental integration) during growth. We compared both mean and variability parameters for craniofacial shape and size among A/WySnJ mice, a strain that does not develop CL/P (C57BL/6J) and their F1 cross. We show that adult A/WySnJ mice that do not express cleft lip exhibit decreased morphological integration of the cranium and that the co-ordination of overall shape and size variation is disrupted compared with both C57BL/6J mice and the F1 cross. The decrease in integration is most pronounced in the palate and face. The absence of this pattern in the F1 cross suggests that it is determined by recessive genetic factors. By contrast, the shape differences between the strains, which are thought to predispose A/WySnJ mice to CL/P, show a range of dominance which suggests a polygenic basis. We suggest that decreased integration of craniofacial growth may be an aetiological factor for CL/P in A/WySnJ mice.