A role for primary cilia in glutamatergic synaptic integration of adult-born neurons

Kumamoto, Natsuko; Gu, Yan; Wang, J; Jenoschka, S; Takemaru, Ken Ichi; Levine, Joel M.; Ge, Shaoyu

doi:10.1186/2046-2530-1-s1-p31

Cited by 25 publications

(40 citation statements)

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“…The role of the cilium in the transduction of key signaling pathways such as Wnt and Shh is undoubtedly relevant to understand their function in neurons but cilia are being shown to play additional roles. For example, conditional ablation of cilia has been shown to result in disrupted dendritic arborization and the establishment of neuronal networks [118]. In addition, cilia are being shown to participate in diverse aspects of neuronal biology such as maintenance of progenitor pools, neurogenesis and neuronal migration, findings that will promote a better understanding of the cellular basis of the different CNS defects that characterize BBS and other ciliopathies (for thorough reviews on the role of cilia in the CNS see [119,120]).…”

Section: Central Nervous Systemmentioning

confidence: 99%

Bardet–Biedl syndrome: Is it only cilia dysfunction?

et al. 2015

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a b s t r a c tBardet-Biedl syndrome (BBS) is a genetically heterogeneous, pleiotropic disorder, characterized by both congenital and late onset defects. From the analysis of the mutational burden in patients to the functional characterization of the BBS proteins, this syndrome has become a model for both understanding oligogenic patterns of inheritance and the biology of a particular cellular organelle: the primary cilium. Here we briefly review the genetics of BBS to then focus on the function of the BBS proteins, not only in the context of the cilium but also highlighting potential extra-ciliary roles that could be relevant to the etiology of the disorder. Finally, we provide an overview of how the study of this rare syndrome has contributed to the understanding of cilia biology and how this knowledge has informed on the cellular basis of different clinical manifestations that characterize BBS and the ciliopathies.

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Section: Central Nervous Systemmentioning

confidence: 99%

Bardet–Biedl syndrome: Is it only cilia dysfunction?

et al. 2015

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“…Our data presented here however do not address whether the primary cilium plays a role in the Shh-induced presynaptic growth. Nevertheless, a recent in vivo study demonstrated that deletion of the primary cilium disrupts synapse formation in adult-born dentate granule cells of the hippocampus (Kumamoto et al, 2012). This raises the possibility that the primary cilium might serve a functional role in the Shh signaling-mediated synapse development in hippocampal neurons in addition to that of the adult-born dentate granule cells.…”

Section: Shh Signaling Changes the Physiology Of Hippocampal Neuronsmentioning

confidence: 99%

Sonic hedgehog regulates presynaptic terminal size, ultrastructure and function in hippocampal neurons

Mitchell

Petralia

Currier

et al. 2012

Journal of Cell Science

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SummarySonic hedgehog (Shh) signaling is essential to the patterning of the embryonic neural tube, but its presence and function in the postmitotic differentiated neurons in the brain remain largely uncharacterized. We recently showed that Shh and its signaling components, Patched and Smoothened, are expressed in postnatal and adult hippocampal neurons. We have now examined whether Shh signaling has a function in these neurons. Using cultured hippocampal neurons as a model system, we found that presynaptic terminals become significantly larger in response to the application of Shh. Ultrastructural examination confirmed the enlarged presynaptic profiles and also revealed variable increases in the size of synaptic vesicles, with a resulting loss of uniformity. Furthermore, electrophysiological analyses showed significant increases in the frequency, but not the amplitude, of spontaneous miniature excitatory postsynaptic currents (mEPSCs) in response to Shh, providing functional evidence of the selective role of Shh in presynaptic terminals. Thus, we conclude that Shh signaling regulates the structure and functional properties of presynaptic terminals of hippocampal neurons.

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“…Primary cilia are required for the expansion of progenitor pool during cerebellar development (10,11) and in the formation of neural stem cells (NSCs) and progenitors in the adult hippocampus (12)(13)(14). Moreover, it has been shown that primary cilia regulate dendritic refinement and synaptic integration of adultborn hippocampal neurons (15). Recent evidence also shows that Arl13b in primary cilia is essential for the early polarization of the neuroepithelium and the formation of radial glia (16).…”

mentioning

confidence: 99%

Primary cilia are required in a unique subpopulation of neural progenitors

Tong

Han

Shah

et al. 2014

Proc. Natl. Acad. Sci. U.S.A.

125

122

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The apical domain of embryonic (radial glia) and adult (B1 cells) neural stem cells (NSCs) contains a primary cilium. This organelle has been suggested to function as an antenna for the detection of morphogens or growth factors. In particular, primary cilia are essential for Hedgehog (Hh) signaling, which plays key roles in brain development. Their unique location facing the ventricular lumen suggests that primary cilia in NSCs could play an important role in reception of signals within the cerebrospinal fluid. Surprisingly, ablation of primary cilia using conditional alleles for genes essential for intraflagellar transport [kinesin family member 3A (Kif3a) and intraflagellar transport 88 (Ift88)] and Cre drivers that are activated at early [Nestin; embryonic day 10.5 (E10.5)] and late [human glial fibrillary acidic protein (hGFAP); E13.5] stages of mouse neural development resulted in no apparent developmental defects. Neurogenesis in the ventricular-subventricular zone (V-SVZ) shortly after birth was also largely unaffected, except for a restricted ventral domain previously known to be regulated by Hh signaling. However, Kif3a and Ift88 genetic ablation also disrupts ependymal cilia, resulting in hydrocephalus by postnatal day 4. To directly study the role of B1 cells' primary cilia without the confounding effects of hydrocephalus, we stereotaxically targeted elimination of Kif3a from a subpopulation of radial glia, which resulted in ablation of primary cilia in a subset of B1 cells. Again, this experiment resulted in decreased neurogenesis only in the ventral V-SVZ. Primary cilia ablation led to disruption of Hh signaling in this subdomain. We conclude that primary cilia are required in a specific Hh-regulated subregion of the postnatal V-SVZ.adult neurogenesis | subependyma | ventricular zone | olfactory bulb | Gli1

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A role for primary cilia in glutamatergic synaptic integration of adult-born neurons

Cited by 25 publications

References 0 publications

Bardet–Biedl syndrome: Is it only cilia dysfunction?

Bardet–Biedl syndrome: Is it only cilia dysfunction?

Sonic hedgehog regulates presynaptic terminal size, ultrastructure and function in hippocampal neurons

Primary cilia are required in a unique subpopulation of neural progenitors

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