Genetically determined absence of an initiation site of cranial neural tube closure is causally related to exencephaly in SELH/Bc mouse embryos

Teratology
T M GunnM J Harris

Abstract

The SELH/Bc mouse strain (SELH) has a high frequency of the lethal neural tube closure defect, exencephaly, in newborns and embryos. Previous work has shown that all SELH mouse embryos have an abnormal mechanism of rostral neural tube closure. They lack initiation of contact and fusion of the cranial neural tube at the prosencephalon/mesencephalon boundary [Closure 2), and undergo closure by extension of a more rostral site of fusion. This process fails in 10-20% of embryos, where the mesencephalic folds remain unelevated, resulting in exencephaly. Previous work has also shown that the cause of liability to exencephaly in SELH mice is multigenic, involving a small number of loci. The purpose of the present study was to test the hypothesis that the genes causing the lack of Closure 2 also cause the liability to exencephaly in SELH, by observation of their joint transmission from genetically segregating animals. A concurrent mapping study provided the necessary genetic material, a segregating F2 generation from a cross of SELH with the normal LM/Bc strain. The genetic liability to exencephaly transmitted by individual F2 sires had been measured by the frequencies of exencephalic day 14 embryos they produced in test-crosses with S...Continue Reading

References

May 3, 1979·Anatomy and Embryology·J A Geelen, J Langman
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Citations

May 29, 2001·Developmental Dynamics : an Official Publication of the American Association of Anatomists·J F Colas, G C Schoenwolf
Oct 13, 2001·Teratology·D M JuriloffS L Dewell
Jun 22, 2005·Birth Defects Research. Part A, Clinical and Molecular Teratology·Muriel J Harris, Diana M Juriloff
Sep 19, 2008·Birth Defects Research. Part A, Clinical and Molecular Teratology·Katharine L StoateDiana M Juriloff
Oct 15, 2013·American Journal of Medical Genetics. Part C, Seminars in Medical Genetics·Claudia Kappen
Nov 7, 2019·BMC Surgery·Jayant Kumar YadavSajid Nazir Bhatti

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