Martínez-Frías M L, Frías J L, Opitz J M
ECEMC and Departamento de Farmacologia, Facultad de Medicina, Universidad Complutense de Madrid, Spain.
Am J Med Genet. 1998 Apr 1;76(4):291-6.
Field theory provides a rational basis for birth defects terminology. During blastogenesis in higher metazoa, pattern formation in the primary field leads to the establishment of upstream expression domains of growth and transcription factors, which, in various permutations and at specific sites and times, lay down the pattern of progenitor fields. Further spatially coordinated, temporally synchronized, and epimorphically hierarchical morphogenetic events, mostly during organogenesis, lead to the attainment of final form in the secondary, epimorphic fields. Because of shared molecular determinants, spatial contiguity, and close timing of morphogenetic events during blastogenesis, most malformations arising during blastogenesis are polytopic, i.e., involving two or more progenitor fields, e.g., acrorenal, cardiomelic, gastromelic, or splenomelic anomalies. Defects of organogenesis tend to be monotopic malformations, e.g., cleft palate or postaxial polydactyly. We suggest that what were called "associations" (e.g., VATER, schisis) be designated primary polytopic developmental field defects, or simply polytopic field defects, and that the term "association" be reserved for the original definition of a statistical combination of anomalies (mostly of organogenesis) [Spranger et al. (1982): J Pediatr 100:160-165]. If genetically caused or predisposed, all structures involved in a polytopic or monotopic malformation are genetically abnormal, whereas the parts secondarily affected as a consequence of a malformation sequence (e.g., spina bifida) are genetically normal. Polytopic field anomalies, per se, must be distinguished from pleiotropy, although such anomalies may constitute a part of pleiotropy (e.g., in trisomy 18). Because they are downstream from pattern-forming events in the primary field, multiple anomalies of organogenesis more likely represent syndromal pleiotropy.
场论为出生缺陷术语提供了合理依据。在高等后生动物的胚泡形成过程中,初级场中的模式形成导致生长和转录因子上游表达域的建立,这些表达域以各种排列方式并在特定的位点和时间,奠定了祖场的模式。进一步在空间上协调、时间上同步且形态发生层次上逐步递进的形态发生事件,大多发生在器官发生过程中,导致次生的、形态建成场中最终形态的形成。由于在胚泡形成过程中存在共同的分子决定因素、空间连续性以及形态发生事件的紧密时间关系,大多数在胚泡形成过程中出现的畸形是多部位的,即涉及两个或更多祖场,例如肢端-肾、心-肢、胃-肢或脾-肢异常。器官发生缺陷往往是单部位畸形,例如腭裂或轴后多指畸形。我们建议将所谓的“关联”(例如VATER、裂)指定为原发性多部位发育场缺陷,或简称为多部位场缺陷,而“关联”一词应保留用于异常(大多为器官发生异常)的统计组合的原始定义[施普朗格等人(1982年):《儿科学杂志》100:160 - 165]。如果是由遗传引起或具有遗传易感性,多部位或单部位畸形中涉及的所有结构在遗传上都是异常的,而作为畸形序列后果而继发受影响的部分(例如脊柱裂)在遗传上是正常的。多部位场异常本身必须与多效性区分开来,尽管此类异常可能构成多效性的一部分(例如在18三体综合征中)。由于它们处于初级场模式形成事件的下游,器官发生的多种异常更可能代表综合征性多效性。