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（Cleft lip and/or palate，CL/P）是人类最常见的先天性畸形之一，但其病因尚不清楚。为了研究CL/P的致病因素，本研究以A/J和CL/Fr近交系小鼠为实验模型。CL/P的发病机制被认为是母体环境因素和胚胎遗传因素共同作用的结果。从流行病学研究结果来看，母亲贫血可能是诱发CL/P的母体环境因素。本研究的目的是：首先，通过建立具有CL/P和贫血两种特征的同源系小鼠模型，筛选出具有导致小鼠轻度贫血的W^v突变基因的新同源系A/J-W^v和CL/Fr-W^v;第二，分析血液形成，（RBC、Hb、Ht、MCV、MCHC）和CL/P频率。利用小鼠直接回交育种方法，可以选育出已达到N12代的两个新的同源品系A/J-W ^v和CL/Fr-W ^v。现在这些品种通过近亲繁殖保持了它们的特性。从血液形成分析数据可以证实，与+/+小鼠相比，W^v /+小鼠的RBC、Hb、Ht和MCHC降低。并分别观察了贫血母亲（W^v /+）和正常母亲（+/+）所产胎儿的CL/P频率。结果表明，贫血母亲（W^v /+）的胎儿CL/P频率高于正常母亲（+/+）。

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 (+/+) .