Schorle H, Meier P, Buchert M, Jaenisch R, Mitchell P J
Whitehead Institute for Biomedical Research, Cambridge, Massachusetts 02142, USA.
Nature. 1996 May 16;381(6579):235-8. doi: 10.1038/381235a0.
During closure of the neural tube in the mouse, transcription factor AP-2 is expressed in ectoderm and in neural-crest cells migrating from the cranial neural folds. Cranial neural crest cells provide patterning information for craniofacial morphogenesis, generate most of the skull bones, and together with placodal ectoderm, form the cranial ganglia. To study the role of AP-2 during embryogenesis, we undertook a targeted mutagenesis of the AP-2 gene in the mouse. Here we report that AP-2(-/-) mice died perinatally with cranio-abdominoschisis and severe dismorphogenesis of the face, skull, sensory organs and cranial ganglia. Failure of cranial closure between 9 and 9.5 days postcoitum coincided with increased apoptosis in the midbrain, anterior hindbrain and proximal mesenchyme of the first branchial arch, but did not involve loss of expression of twist or Pax-3, two other regulatory genes known to be required for cranial closure.
在小鼠神经管闭合期间，转录因子AP - 2在外胚层以及从颅神经褶迁移而来的神经嵴细胞中表达。颅神经嵴细胞为颅面形态发生提供模式信息，生成大部分颅骨，并与基板外胚层一起形成颅神经节。为了研究AP - 2在胚胎发育过程中的作用，我们对小鼠的AP - 2基因进行了定向诱变。在此我们报告，AP - 2(-/-)小鼠在围产期死亡，伴有颅腹裂以及面部、颅骨、感觉器官和颅神经节的严重畸形。怀孕后9至9.5天颅部闭合失败与中脑、后脑前部和第一鳃弓近端间充质中凋亡增加同时发生，但不涉及已知颅部闭合所需的另外两个调节基因twist或Pax - 3表达的缺失。
