SIP1 expression patterns in brain investigated by generating a SIP1-EGFP reporter knock-in mouse.
A loss of function of SIP1 (Smad interacting protein 1) in the mouse as well as in human of Mowat-Wilson syndrome results in severe and multiple defects in neural tissue development, especially in the brain. However, no detailed expression analysis of SIP1 during brain development has been previously reported. In this study, we describe the generation of an EGFP knock-in reporter mouse for the Sip1 locus and our subsequent analysis of SIP1-EGFP fusion protein expression during brain development. SIP1-EGFP expression was observed in the pyramidal neurons of the hippocampus, the dentate gyrus, and the postmitotic neurons in the cerebral cortex. In layer 5 of the cerebral cortex, SIP1-EGFP expression was complementary to the Ctip2-expressing neurons, most of which are thought to be the cortico-spinal neurons. This suggested that SIP1-EGFP expressing cells might have the specific trajectory targets other than the spinal region. We further observed SIP1-EGFP expression in oligodendrocytes of the corpus callosum and fimbria, Bergmann glial cells of the cerebellum, the olfactory bulb, and in the serotonergic and dopaminergic neurons of the raphe nuclei in the brainstem. These findings may help to clarify the unknown roles of SIP1 in these cells and the pathoetiology of Mowat-Wilson syndrome.
Department of Perinatology, Institute for Developmental Research, Aichi Human Service Center, Kasugai, Aichi, Japan.
Yuriko
Y
Nishizaki
Tsuyoshi
T
Takagi
Fumiko
F
Matsui
Yujiro
Y
Higashi
D000818:Animals; D001921:Brain / Q000254:growth & development / Q000378:metabolism*; D002540:Cerebral Cortex / Q000254:growth & development / Q000378:metabolism; D003337:Corpus Callosum / Q000378:metabolism; D018891:Dentate Gyrus / Q000254:growth & development / Q000378:metabolism; D019066:Facies; D055879:Gene Knock-In Techniques; D017930:Genes, Reporter; D049452:Green Fluorescent Proteins / Q000378:metabolism*; D006627:Hirschsprung Disease / Q000235:genetics; D018398:Homeodomain Proteins / Q000235:genetics / Q000378:metabolism*; D006801:Humans; D008607:Intellectual Disability / Q000235:genetics; D051379:Mice; D008810:Mice, Inbred C57BL; D008831:Microcephaly / Q000235:genetics; D017966:Pyramidal Cells / Q000378:metabolism; D011993:Recombinant Fusion Proteins / Q000235:genetics / Q000378:metabolism*; D012097:Repressor Proteins / Q000235:genetics / Q000378:metabolism*; D025521:Tumor Suppressor Proteins / Q000378:metabolism*; D000074165:Zinc Finger E-box Binding Homeobox 2
C511143:Bcl11b protein, mouse; D018398:Homeodomain Proteins; D011993:Recombinant Fusion Proteins; D012097:Repressor Proteins; D025521:Tumor Suppressor Proteins; C120602:ZEB2 protein, mouse; D000074165:Zinc Finger E-box Binding Homeobox 2; C485184:enhanced green fluorescent protein; D049452:Green Fluorescent Proteins
Mowat-Wilson syndrome; zeb2; zfhx1b