Developmental brain abnormalities accompanied with the retarded production of S-100β protein in genetic polydactyly mice

Naruse, Ichiro; Kato, Kanefusa; Asano, Taku; Suzuki, Fujiko; Kameyama, Yoshiro

doi:10.1016/0165-3806(90)90283-5

Cited by 44 publications

(48 citation statements)

References 21 publications

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“…Pdn/Pdn Brains Neurofilament, Tuj1 and L1 immunhistochemical stainings, and cortical DiI labeling confirmed a previous description of CC malformation in Pdn mutants (Naruse et al 1990),…”

Section: Midline Guidepost Cells Are Severely Disorganized Insupporting

confidence: 87%

“…The mutant mouse lines Pdn, τGFP, Slit2, Sprouty1, and Sprouty2 and mating strategies have been described previously (Naruse et al 1990;Pratt et al 2000;Plump et al 2002;Basson et al 2005;Shim et al 2005;Simrick et al 2011). All experimental procedures involving mice were performed in accordance with local guidelines.…”

Section: Micementioning

confidence: 99%

“…Moreover, Gli3 functions in axon pathfinding in the forebrain. The Gli3 hypomorphic mouse mutant Polydactyly Nagoya (Pdn) shows defects in the corticothalamic and thalamocortical tracts (Magnani et al 2010) and lacks the CC (Naruse et al 1990) though for unknown reasons. Using transplantation experiments, we here demonstrate that midline abnormalities are primarily responsible for agenesis of the corpus callosum (ACC).…”

Section: Introductionmentioning

confidence: 99%

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Gli3 Controls Corpus Callosum Formation by Positioning Midline Guideposts During Telencephalic Patterning

et al. 2012

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The corpus callosum (CC) represents the major forebrain commissure connecting the 2 cerebral hemispheres. Midline crossing of callosal axons is controlled by several glial and neuronal guideposts specifically located along the callosal path, but it remains unknown how these cells acquire their position. Here, we show that the Gli3 hypomorphic mouse mutant Polydactyly Nagoya (Pdn) displays agenesis of the CC and mislocation of the glial and neuronal guidepost cells. Using transplantation experiments, we demonstrate that agenesis of the CC is primarily caused by midline defects. These defects originate during telencephalic patterning and involve an upregulation of Slit2 expression and altered Fgf and Wnt/β-catenin signaling. Mutations in sprouty1/2 which mimic the changes in these signaling pathways cause a disorganization of midline guideposts and CC agenesis. Moreover, a partial recovery of midline abnormalities in Pdn/Pdn;Slit2 −/− embryos mutants confirms the functional importance of correct Slit2 expression levels for callosal development. Hence, Gli3 controlled restriction of Fgf and Wnt/β-catenin signaling and of Slit2 expression is crucial for positioning midline guideposts and callosal development.

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“…Pdn/Pdn Brains Neurofilament, Tuj1 and L1 immunhistochemical stainings, and cortical DiI labeling confirmed a previous description of CC malformation in Pdn mutants (Naruse et al 1990),…”

Section: Midline Guidepost Cells Are Severely Disorganized Insupporting

confidence: 87%

Section: Micementioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Gli3 Controls Corpus Callosum Formation by Positioning Midline Guideposts During Telencephalic Patterning

et al. 2012

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“…The origin of the commissure defects observed in Dmrt5 −/− embryos, also found in Emx2 and Gli3 mutants (Naruse et al 1990;Yoshida et al 1997), is unknown. Disturbance of commissure formation is likely to result from intrinsic defects in cortical neurons or from failure of the environment to produce an extracellular environment that is suitable for navigation (Lindwall et al 2007).…”

Section: Mild Cortical Lamination Defects and Severe Forebrain Axonalmentioning

confidence: 99%

The Doublesex Homolog Dmrt5 is Required for the Development of the Caudomedial Cerebral Cortex in Mammals

et al. 2012

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Regional patterning of the cerebral cortex is initiated by morphogens secreted by patterning centers that establish graded expression of transcription factors within cortical progenitors. Here, we show that Dmrt5 is expressed in cortical progenitors in a high-caudomedial to low-rostrolateral gradient. In its absence, the cortex is strongly reduced and exhibits severe abnormalities, including agenesis of the hippocampus and choroid plexus and defects in commissural and thalamocortical tracts. Loss of Dmrt5 results in decreased Wnt and Bmp in one of the major telencephalic patterning centers, the dorsomedial telencephalon, and in a reduction of Cajal-Retzius cells. Expression of the dorsal midline signaling center-dependent transcription factors is downregulated, including Emx2, which promotes caudomedial fates, while the rostral determinant Pax6, which is inhibited by midline signals, is upregulated. Consistently, Dmrt5 −/− brains exhibit patterning defects with a dramatic reduction of the caudomedial cortex. Dmrt5 is increased upon the activation of Wnt signaling and downregulated in Gli3 xt/xt mutants. We conclude that Dmrt5 is a novel Wnt-dependent transcription factor required for early cortical development and that it may regulate initial cortical patterning by promoting dorsal midline signaling center formation and thereby helping to establish the graded expression of the other transcription regulators of cortical identity.

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“…They also exhibit various brain malformations, including absence of olfactory bulb and corpus callosum [9]. Pdn/+ (heterozygote of Polydactyly Nagoya mouse) exhibits preaxial polydactyly of the distal phalangeal type in the hind limbs, Hongo/Hakuba/Shiota/Naruse but no brain malformations or behavioral dysfunctions [7,9].…”

Section: Introductionmentioning

confidence: 99%

Suckling Dysfunction Caused by Defects in the Olfactory System in Genetic Arhinencephaly Mice

et al. 2000

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Mouse newborns find their mother’s nipples and suckle milk by themselves. It has been argued which sense organ they use when locating their mother’s nipples to suckle milk. Olfactory or tactile sensory systems are primary candidates. In the present study, we investigated the trigeminal-whisker sensory and olfactory systems in genetic arhinencephaly mouse embryos (Pdn/Pdn). Pdn/Pdn newborns do not suckle milk and die within 1 day after birth. Dysfunction of nipple-searching behavior was clear in Pdn/Pdn newborns. Pdn/Pdn newborns had a complete developmental failure in the olfactory nerve projection to the central nervous system and no olfactory bulb architecture. The trigeminal-whisker system was intact in this strain. From the results of these experiments, it was suggested that the olfactory system is essential for nipple-searching behavior and suckling milk and that the trigeminal-whisker system is not able to substitute for the lack of olfactory input in mouse newborns.

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Developmental brain abnormalities accompanied with the retarded production of S-100β protein in genetic polydactyly mice

Cited by 44 publications

References 21 publications

Gli3 Controls Corpus Callosum Formation by Positioning Midline Guideposts During Telencephalic Patterning

Gli3 Controls Corpus Callosum Formation by Positioning Midline Guideposts During Telencephalic Patterning

The Doublesex Homolog Dmrt5 is Required for the Development of the Caudomedial Cerebral Cortex in Mammals

Suckling Dysfunction Caused by Defects in the Olfactory System in Genetic Arhinencephaly Mice

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