Journal ArticleDOI
Effect of the uterine environment on the frequency of spontaneous cleft lip in CL/FR mice.
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TLDR
The demonstration of a uterine effect on cleft lip susceptibility suggests that it might be possible to modify the environment in the direction of increased resistance.Abstract:
It was previously shown (Davidson et al., '69) that the A/J female mouse predisposes her offspring to congenital cleft lip, either by providing a uterine environment or cytoplasmic factors that increase susceptibility, or by not providing a cytoplasmically transmitted factor, present in the C57BL/6J strain, that decreases susceptibility to cleft lip. In the present experiment males of the CL/Fr strain, which has a relatively high frequency of cleft lip, were crossed to females of the C57BL/6J strain, and the F1 offspring were backcrossed to CL/Fr animals. Three second backcrosses were made such that embryos of comparable genotype developed in CL/Fr mothers or in hybrid mothers with or without the postulated cytoplasmic factors. The results showed that the maternal effect on cleft lip susceptibility was present in the CL/Fr strain, and was not mediated through the cytoplasm. Maternal age, parity, litter size, and sex and pigmentation of the offspring showed no consistent associations with frequency of cleft lip. The demonstration of a uterine effect on cleft lip susceptibility suggests that it might be possible to modify the environment in the direction of increased resistance.read more
Citations
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Journal ArticleDOI
Genetics of nonsyndromic oral clefts revisited.
TL;DR: The main features of published studies pointing out their strengths and limitations are described and insight is given into current methods for detecting the presence of interaction between genetic markers and environmental exposures in the etiology of oral clefts.
Journal ArticleDOI
Prenatal Craniofacial Development: New Insights On Normal and Abnormal Mechanisms
M.C. Johnston,P.T. Bronsky +1 more
TL;DR: Technical advances are radically altering the authors' concepts of normal prenatal craniofacial development, including concepts of germ layer formation, the establishment of the initial head plan in the neural plate, and the manner in which head segmentation is controlled by regulatory gene activity in neuromeres and their derived neural crest cells.
Journal ArticleDOI
Mouse genetic models of cleft lip with or without cleft palate.
TL;DR: Previously unexamined genes are identified as strong candidate genes for causative roles in human nonsyndromic CLP: BMP4, BMPR1B, TFAP2A, SOX4, WNT9B, W NT3, and SP8.
Journal ArticleDOI
Phenotypic variability and craniofacial dysmorphology: increased shape variance in a mouse model for cleft lip.
Trish E. Parsons,Erika Kristensen,Lynnette Hornung,Virginia M. Diewert,Steven K. Boyd,Rebecca Z. German,Benedikt Hallgrímsson +6 more
TL;DR: Three‐dimensional geometric morphometric and novel high throughput morphometric techniques based on three‐dimensional computed microtomography of mouse embryos are used to analyze craniofacial shape variation during primary palate formation to confirm previous findings that the midface in A/WySn embryos, and the maxillary prominence in particular, is relatively reduced in size and appears to be developmentally delayed.
Journal ArticleDOI
Unravelling the complex genetics of cleft lip in the mouse model.
TL;DR: Specific combinations of marker genotypes associated with cleft lip risk indicated that high risk in A/WySnJ mice is caused by epistatic interaction between clf1 and clf2 in the context of a genetic maternal effect.
References
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Journal ArticleDOI
Genetical studies on the skeleton of the mouse
TL;DR: The tail anomalies are produced mechanically either by spina bifida which persists, or by a rudimentary form of it (delayed closure of the posterior neuropore) which is transitory as mentioned in this paper.
Journal ArticleDOI
A maternal effect on the frequency of spontaneous cleft lip in the A-J mouse.
TL;DR: The A/J mother contributes to the fetal susceptibility to Cleft lip, either by providing an appropriate uterine environment or by not providing a factor, present in the C57BL strain, that decreases susceptibility to cleft lip.
Journal ArticleDOI
Influence of uterine site on occurrence of spontaneous cleft lip in mice.
TL;DR: It has been found that within the A/Jax strain, embryos in the uterine site nearest the ovary develop cleft lip significantly more often than embryos in other positions in the uterus.
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