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Maneshi, M. M.

Publications and source records attributed to Maneshi, M. M..

3 recordsLinked to original sources

NGN2 Expression and Regional Patterning Allow Rapid Differentiation from hiPSCs to DRG-Like Neurons Responsive to Type 2 Cytokines

Itch or pruritus, is a sensation that elicits scratching behaviour and is a major symptom and cause of morbidity in skin diseases such as atopic dermatitis (AD), allergic contact dermatitis (ACD), prurigo nodularis (PN), and urticaria. Itch is often triggered by inflammatory stimuli in the skin including type 2 cytokines such as IL-4, IL-13, and/or IL-31. Several therapies targeting type 2 immune pathways have been developed to treat pruritus; however, itch improvement in many patients remains to be improved. Thus, additional approaches to modulate sensory neuron activity are needed. Ex vivo or even in vitro study of the molecular mechanisms underlying primary sensory neuron activation is challenging since harvesting neurons from dorsal root ganglia (DRG) in patients can only be done from cadavers. Herein, we describe rapid human sensory neurons generation (2 days to precursor cells) by in vitro differentiation of human induced pluripotent stem cells (hiPSC) from simultaneous application of patterning factors with NGN2 overexpression. We show that these hiPSC-derived sensory neurons possess key characteristics of primary sensory neurons. They express key neuronal markers, such as TRKA receptors, TRPV1 and TRPA1 channels, and functionally respond to the TRPV1 agonist capsaicin. In addition, they express key type 2 cytokine receptors such as interleukin (IL)-4R and IL31-R, known to promote itch in AD and PN. Moreover, these cells are functional as our sensory neurons respond to IL-4, IL-13 and IL-31 stimulation. Collectively, these data demonstrate that our protocol generates a phenotypic profile consistent with native somatosensory neurons that can facilitate development of novel approaches to model and treat pruritic disease.

neuroscience↗

GDF15 is a dynamic biomarker of the Integrated Stress Response in the central nervous system

AimCharacterize Growth Differentiation Factor 15 (GDF15) as a secreted biomarker of the Integrated Stress Response (ISR) within the Central Nervous System (CNS). MethodsWe determined GDF15 levels utilizing in vitro and in vivo neuronal systems wherein the ISR was activated. Primarily, we used the murine model of Vanishing White Matter disease (VWMD), a neurological disease driven by persistent ISR in the CNS, to establish a link between levels of GDF15 in the cerebrospinal fluid (CSF) and ISR gene expression signature in the CNS. GDF15 was also determined in the CSF of VWM patients. ResultsGDF15 expression was increased concomitant to ISR activation in stress-induced primary astrocytes as well as in retinal ganglion cells following optic nerve crush, while treatment with 2Bact, a specific eIF2B activator, suppressed both the ISR and GDF15. In the VWMD model, CSF GDF15 levels corresponded with the magnitude of the ISR and were reduced by 2BAct. In VWM patients, mean CSF GDF15 was elevated >20-fold as compared to healthy controls, whereas plasma GDF15 was undifferentiated. ConclusionsThese data suggest that CSF GDF15 is a dynamic marker of ISR activation in the CNS and may serve as a pharmacodynamic biomarker for ISR-modulating therapies.

neuroscience↗

Regulation of neuropathic pain by microglial Orai1 channels

Microglia are important mediators of neuroinflammation that underlies neuropathic pain. However, the molecular checkpoints controlling microglial reactivity are not well-understood. We investigated the role of Orai1 channels for microglia-mediated neuroinflammation following nerve injury and find that deletion of Orai1 in microglia attenuates Ca2+ signaling and the production of inflammatory cytokines by proalgesic agonists. Conditional deletion of Orai1 attenuated microglia proliferation in the dorsal horn, spinal cytokines levels, and potentiation of excitatory neurotransmission following peripheral nerve injury. These cellular effects were accompanied by mitigation of pain hyperalgesia in Orai1 knockout mice. A small-molecule Orai1 inhibitor, CM4620, similarly mitigated allodynia in male mice. Surprisingly, these protective effects were not seen in female mice, revealing striking sexual dimorphism in Orai1 regulation of microglial reactivity and hyperalgesia. These findings indicate that Orai1 channels are key regulators of the sexually dimorphic role of microglia for the neuroinflammation that underlies neuropathic pain.

neuroscience↗