Knockout mouse lines that show embryonic and perinatal lethality are valuable models to investigate the genetic pathways that are associated with human congenital diseases. To elucidate the relationship between a specific gene and a developmental defect, detailed screening of knockout phenotypes is pivotal. Here, Wolfgang Weninger and colleagues used high-resolution episcopic microscopy (HREM) to screen the morphological phenotypes of embryonic day 14.5 (E14.5) embryos of 34 mouse strains that produce prenatally lethal offspring. The authors developed a reliable and ergonomic screening protocol to efficiently and comprehensively score structural abnormalities in those embryos. Their approach enabled them to detect a total of 58 defects that might be missed by employing alternative three-dimensional imaging methods and scoring systems. Many of these defects might be causal to embryonic or perinatal mortality. The results demonstrate that HREM combined with a systematic screening protocol enables more efficient phenotyping of E14.5 mouse embryos than any alternative approach. Such a method will contribute to advancing our knowledge of normal tissue and organ development, and of the causality of congenital diseases. Page 1143
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IN THIS ISSUE| 01 October 2014
Advancing congenital defect phenotyping using HREM imaging
Online Issn: 1754-8411
Print Issn: 1754-8403
© 2014. Published by The Company of Biologists Ltd
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution and reproduction in any medium provided that the original work is properly attributed.
Dis Model Mech (2014) 7 (10): e1002.
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Advancing congenital defect phenotyping using HREM imaging. Dis Model Mech 1 October 2014; 7 (10): e1002. doi:
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