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Neuronal Primary Cilia: an Underappreciated Signaling and Sensory Organelle in the Brain

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Neuronal Primary Cilia: an Underappreciated Signaling and Sensory Organelle in the Brain HOT TOPICS ............................................................................................................................................................... 244 Neuronal Primary Cilia: highlighting the importance of pri- in the same neuronal cilium and form mary cilia function within the brain heteromers (Green et al, 2012). As GPCR An Underappreciated (Figure 1). heteromerization can lead to changes Signaling and Sensory Although it was discovered over 50 in ligand binding properties, receptor Organelle in the Brain years ago that central neurons possess desensitization and internalization, or primary cilia, their precise functions the generation of a completely unique The human brain is comprised of remain unclear and the roles primary signal, this adds an additional layer of billions of neurons that communicate cilia play in overall neuronal function complexity to neuronal ciliary signaling. with each other through an intricate are unknown. The first insights into Thus, neuronal cilia may represent a web of axons and dendrites. However, their functions were provided by the specialized signaling platform that facil- more and more research suggests that discovery that certain G protein- itates interactions between receptors neurons may also sense and respond coupled receptors (GPCRs), including and/or signaling cascades to provide an to their environment via a specialized somatostatin receptor 3 and serotonin exclusive ciliary signal. organelle called a primary cilium receptor 6, selectively localize to Studies are needed to further unveil (Green and Mykytyn, 2010). Primary neuronal cilia (Brailov et al, 2000; the molecular composition of neuro- cilia are typically solitary, microtu- Handel et al, 1999). Subsequently, nal primary cilia and determine their bule-based appendages that project neuronal cilia were found to be precise roles in neuronal signaling. from the surface of nearly every cell enriched for type 3 adenylyl cyclase Understanding ciliary GPCR signaling, type in the human body, including (Bishop et al, 2007). As primary cilia generated from homomers or hetero- most neurons throughout the brain. are restricted compartments and their mers, will shed light on the function The primary cilium is thought to act function is defined by the complement of these understudied organelles and as an antenna surveying the extra- of proteins enriched within them, create a model for non-synaptic cellular milieu, receiving various sig- these seminal studies suggested that neuronal signaling. The future of drug nals, and transmitting those signals neuronal primary cilia have the cap- development for ciliopathies, and into the cell. For example, the outer ability to act as non-synaptic sensory likely other neurological disorders, segment of photoreceptors, which is a and signaling organelles. A critical will rely on elucidating neuronal modified cilium, mediates vision by question is why neurons have devel- ciliary signaling. sensing light. Olfactory cilia mediate oped ciliary signaling. One intriguing olfaction by sensing and responding explanation is neuronal cilia are spe- cialized signaling compartments that to odorants. Defective formation or ACKNOWLEDGEMENTS function of primary cilia is implicated generate a unique signal. The basis of in the pathogenesis of many human this uniqueness may be the complement This research was supported by developmental disorders and diseases, of GPCRs on the ciliary membrane, the GM083120 to KM. collectively termed ciliopathies. Cilio- precise signaling transduction pathways pathy patients display a range of neuro- coupled to these receptors, and/or the Jill A Green1 and Kirk Mykytyn1 1 logical disorders (Lee and Gleeson, regulation of these signaling cascades. Wexner Medical Center, Department of Pharmacology, The Ohio State University, Columbus, 2011), including cognitive deficits Moreover, we recently discovered that OH, USA and behavioral phenotypes, thereby different GPCRs can colocalize with- E-mail: [email protected] Figure 1. (a) A schematic of a network of neurons. Primary cilia (red) protrude from the neuronal cell body. (b) A schematic of a neuronal primary cilium. GPCRs selectively localize to the membrane of the cilium where they may reside as monomeric units and/or heteromers. An intraflagellar transport particle that mediates protein trafficking within cilia is indicated in green. (c) A hypothalamic neuron labeled for the ciliary GPCR, melanin-concentrating hormone receptor 1 (Mchr1, green). Scale bar ¼ 10 mm. .............................................................................................................................................. Neuropsychopharmacology HOT TOPICS ............................................................................................................................................................... 245 FUNDING AND DISCLOSURE nucleus in a stimulus-dependent man- The authors declare no conflict of interest. When Synaptic Proteins Meet the Genome: ner, where they can regulate via their ........................................................................................... nuclear target interactions very specific Bishop GA, Berbari NF, Lewis J, Mykytyn K (2007). Type III adenylyl cyclase localizes to primary cilia Transcriptional aspects of gene expression (Jordan and throughout the adult mouse brain. J Comp Neurol Regulation in Cell Death Kreutz, 2009). However, this type of 505: 562–571. signaling is challenged by space and Brailov I, Bancila M, Brisorgueil MJ, Miquel MC, and Plasticity by the Hamon M, Verge D (2000). Localization of time constraints. Synapses are localized 5-HT(6) receptors at the plasma membrane Synapto–Nuclear far away from the nucleus and synaptic of neuronal cilia in the rat brain. Brain Res 872: 271–275. Messenger Jacob signals are very labile and rapidly Green JA, Gu C, Mykytyn K (2012). Heteromerization degraded. On top the minute quantities of ciliary G protein-coupled receptors in the mouse of signaling molecules that can be brain. PLoS ONE 7: e46304. Neurons express more genes than any Green JA, Mykytyn K (2010). Neuronal ciliary signaling other cell type and it is therefore released from synaptic sites are greatly in homeostasis and disease. Cell Mol Life Sci 67: unlikely that synapto–nuclear Ca2 þ diluted in relation to the entire volume 3287–3297. of the dendritic arborization or nu- Handel M, Schulz S, Stanarius A, Schreff M, signaling alone can explain the specific Erdtmann-Vourliotis M, Schmidt H et al (1999). genomic response to the plethora of cleus. A precise machinery is therefore Selective targeting of somatostatin receptor 3 to extracellular stimuli that control gene required to deliver the signal to nuclear neuronal cilia. Neuroscience 89: 909–926. Lee JE, Gleeson JG (2011). Cilia in the nervous expression. Possible candidates for target sites, but unfortunately very little system: linking cilia function and neurodevelop- encoding of signals at the origin and is known on just how protein messen- mental disorders. Curr Opin Neurol 24: 98–105. later decoding at a nuclear destination ger can translocate several hundred Neuropsychopharmacology Reviews (2014) 39, 244–245; are synapto–nuclear protein messen- micrometers, avoid degradation, retain doi:10.1038/npp.2013.203 ger. These proteins translocate to the signal and do this in a reliable manner. Figure 1. Schematic illustrating the encoding of the origin of synaptic and extrasynaptic NMDAR signals by Jacob. Neuronal activity leads to Calpain- mediated cleavage of a-internexin and the myristoylated N terminus of Jacob. High but not low frequency stimulation of synaptic NMDAR results in ERK activation, ERK binding, and phosphorylation of the serine180 in Jacob. This is then followed by recruitment of C-terminal fragments of a-internexin or a tighter assembly of an already preformed complex, which is then efficiently protected against phosphatase activity analogous to a mechanism demonstrated by Perlson et al (2005) for the retrograde transport of pERK after axon injury. In the nucleus, this complex can dock to CREB and potentially other sites and will enhance plasticity and cell survival promoting gene expression. Following the extrasynaptic NMDAR activation, ERK is not activated and remains outside of the nucleus, while non-phosphorylated Jacob will dock yet unknown protein components to CREB that eventually induce CREB shut-off and cell death. .............................................................................................................................................. Neuropsychopharmacology.
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