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PredicateObject
rdf:type
lifeskim:mentions
pubmed:dateCreated
2007-3-12
pubmed:abstractText
The mouse Engrailed genes, En1 and En2, play an important role in the development of the cerebellum from its inception at the mid/hindbrain boundary in early embryonic development through cell type specification events and beyond. In the absence of En1, the cerebellum and caudal midbrain fail to develop normally--a phenotype that we have previously reported to be strain dependent. On the 129/S1 strain background, En1 null alleles lead to mid/hindbrain failure, whereas on the C57BL/6 background, En1 deficiency is compatible with near normal cerebellar development. We have pursued this dramatic effect of genetic background by performing a genetic modifier screen through F1 backcross and F1 intercross matings. The backcross has yielded two strong candidate intervals with suggestive linkage to a third region. Moreover, variations in rescue frequency among subgroups within the backcross indicate gender and parent of origin influences on rescue penetrance. The intercross data reveal locus heterogeneity of the En1 modifiers, with more than one compliment of C57BL/6 and 129/S1 alleles capable of mediating the rescue phenotype. These findings highlight the complexity and plasticity of gene networks involved in brain development.
pubmed:language
eng
pubmed:journal
pubmed:citationSubset
IM
pubmed:chemical
pubmed:status
MEDLINE
pubmed:month
Apr
pubmed:issn
0006-8993
pubmed:author
pubmed:issnType
Print
pubmed:day
6
pubmed:volume
1140
pubmed:owner
NLM
pubmed:authorsComplete
Y
pubmed:pagination
170-8
pubmed:meshHeading
pubmed:year
2007
pubmed:articleTitle
A genetic study of the suppressors of the Engrailed-1 cerebellar phenotype.
pubmed:affiliation
Alzheimer Research Laboratory, Department of Neuroscience, Case Western Reserve University, Cleveland, OH 44106, USA.
pubmed:publicationType
Journal Article