Experimental Studies on Development of Cleft LIp and/or Palate. -Analysis and Breeding of CL/P Model Mouse with Anemia (A/J- W^v, CL/Fr- W^v) -

唇腭裂(CL/P)是人类最常见的先天性畸形之一，其病因尚不清楚。为探讨CL/P的致病因素，以近交系A/J和CL/Fr小鼠为实验模型。CL/P的发病机制被认为既涉及母体环境因素，也涉及胚胎遗传因素。从流行病学研究结果可以看出，CL/P可能是由贫血母亲作为母体环境因素诱发的。为了研究CL/P与贫血的关系，我们要求同时具有CL/P和贫血特征的同源小鼠研究CL/P与贫血的关系。本研究的目的是：一是开发具有W^v突变基因的新的同源品系A/J-W^v和CL/Fr-W^v，它们在小鼠中引起轻微的贫血；二是分析其胎儿的血细胞形成(RBC、Hb、Ht、MCV、MCHC)和CL/P频率。在这一时期，我们可以利用小鼠育种的方法(直接回交)培育出已经传到N12代的两个新的同源品系A/J-W^v和CL/Fr-W^v。现在，这些菌株通过近亲交配保持了它们的特征。从造血分析数据可以证实，与+/+小鼠相比，W^v/+小鼠的RBC、Hb、Ht和MCHC明显降低。并分别观察了贫血组(W^v/+)和正常母亲组(+/+)胎儿的CL/P频率。从这一结果看来，贫血母亲的胎儿CL/P频率(W^v/+)明显高于正常母亲(+/+)。

Cleft lip and/or palate (CL/P) is one of the most common congenital malformations in human, but the cause of that is still unclear. To study the causal factor of CL/P,inbred mouse strains A/J and CL/Fr have been used as experimental model. The pathogenesis of CL/P have been thought to involve both the maternal environmental factor and the enbryonic genetic facotr. From the result of the epidemiologic study, we noticed that CL/P may be induced by anemic mother as the maternal environmental factor. To investigate that, we required the congenic mice that have both characters of CL/P and anemia to study the relation between CL/P and anemia.The purposes of this study are : first, the developing new congenic strains A/J-W^v and CL/Fr-W^v that have W^v mutant genes that invite a slight anemia in mice and : second, the analyzing blood formation (RBC,Hb, Ht, MCV,MCHC) and CL/P frequency of their fetuses.In this term, we could develop two new congenic strains A/J- W^v and CL/Fr- W^v that already attained to N12 generation using mouse breeding method (direct backcross). Now these strains are keeping their character by inbreeding. From the data of analyzing blood formation, we could confirm that the decreases of RBC,Hb, Ht and MCHC in W^v /+mice compare with +/+ mice. And we observed the CL/P frequencies of fetuses that developed in anemic mothres (W^v /+) and in mormal mothers (+/+), respectively. From that results, it seems that CL/P frequencies of fetuses are more frquent in anemic mothers (W^v /+) than in mormal mothers (+/+) .