Copp A J, Seller M J, Polani P E
J Embryol Exp Morphol. 1982 Jun;69:151-67.
A dye-injection technique has been used to determine the developmental stage at which posterior neuropore (PNP) closure occurs in normal and mutant curly tail mouse embryos. In vivo, the majority of non-mutant embryos undergo PNP closure between 30 and 34 somites whereas approximately 50% of all mutant embryos show delayed closure, and around 20% maintain an open PNP even at advanced stages of development. A similar result has been found for embryos developing in vitro from the headfold stage. Later in development, 50--60% of mutant embryos in vivo develop tail flexion defects, and 15--20% lumbosacral myeloschisis. This supports the view that delayed PNP closure is the main developmental lesion leading to the appearance of caudal neural tube defects in curly tail mice. The neural tube is closed in the region of tail flexion defects, but it is locally over-expanded and abnormal in position. The significance of these observations is discussed in relation to possible mechanisms of development of lumbosacral and caudal neural tube defects. This paper constitutes the first demonstration of the development of a genetically induced malformation in vitro.
一种染料注射技术已被用于确定正常和突变型卷尾小鼠胚胎中后神经孔（PNP）闭合发生的发育阶段。在体内，大多数非突变胚胎在30至34体节之间发生PNP闭合，而大约50%的所有突变胚胎显示闭合延迟，并且约20%即使在发育后期也保持PNP开放。对于从头褶期开始体外发育的胚胎也发现了类似的结果。在发育后期，体内50 - 60%的突变胚胎出现尾部弯曲缺陷，15 - 20%出现腰骶部脊髓裂。这支持了这样一种观点，即PNP闭合延迟是导致卷尾小鼠尾部神经管缺陷出现的主要发育病变。神经管在尾部弯曲缺陷区域是闭合的，但局部过度扩张且位置异常。结合腰骶部和尾部神经管缺陷可能的发育机制讨论了这些观察结果的意义。本文首次证明了体外遗传诱导畸形的发育过程。
