GPR56/ADGRG1 regulates development and maintenance of peripheral myelin

Ackerman, Sarah D.; Luo, Rong; Poitelon, Yannick; Mogha, Amit; Harty, Breanne L.; D’Rozario, Mitchell; Sánchez, Nicolás Fuster; Lakkaraju, Asvin K. K.; Gamble, Paul; Li, Jun; Qu, Jun; MacEwan, Matthew R.; Ray, Wilson Zachary; Aguzzi, Adriano; Feltri, M. Laura; Piao, Xiangshu; Monk, Kelly R.

doi:10.1084/jem.20161714

Cited by 53 publications

(61 citation statements)

References 68 publications

(160 reference statements)

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“…Mutations in the plectin gene are responsible for human diseases denominated plectinopathies [247]. Among these, the most common is epidermolysis bullosa simplex, with muscular dystrophy and neuropathy-like symptoms due to defects in the peripheral myelin ultrastructure [246,279].…”

Section: Mutations In Cytoskeletal Genes Associated With Neurodegenermentioning

confidence: 99%

Much More Than a Scaffold: Cytoskeletal Proteins in Neurological Disorders

Muñoz-Lasso

Romá‐Mateo

Pallardó

et al. 2020

Cells

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Recent observations related to the structure of the cytoskeleton in neurons and novel cytoskeletal abnormalities involved in the pathophysiology of some neurological diseases are changing our view on the function of the cytoskeletal proteins in the nervous system. These efforts allow a better understanding of the molecular mechanisms underlying neurological diseases and allow us to see beyond our current knowledge for the development of new treatments. The neuronal cytoskeleton can be described as an organelle formed by the three-dimensional lattice of the three main families of filaments: actin filaments, microtubules, and neurofilaments. This organelle organizes well-defined structures within neurons (cell bodies and axons), which allow their proper development and function through life. Here, we will provide an overview of both the basic and novel concepts related to those cytoskeletal proteins, which are emerging as potential targets in the study of the pathophysiological mechanisms underlying neurological disorders.

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Section: Mutations In Cytoskeletal Genes Associated With Neurodegenermentioning

confidence: 99%

Much More Than a Scaffold: Cytoskeletal Proteins in Neurological Disorders

Muñoz-Lasso

Romá‐Mateo

Pallardó

et al. 2020

Cells

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“…Hence, Gpr56 deficiency prompts the premature cell cycle exit of OPC, leading to less mature oligodendrocytes ( 23 , 31 ). In a more recent study, GPR56 was shown to be also involved in myelin development and maintenance in the peripheral nervous system by interacting with plectin ( 33 ).…”

Section: Biological and Cellular Functions Of Gpr56mentioning

confidence: 99%

The Activation and Signaling Mechanisms of GPR56/ADGRG1 in Melanoma Cell

Huang

Lin

2018

Front. Oncol.

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Adhesion G protein-coupled receptors (aGPCRs) constitute the second largest GPCR subfamily. GPR56/ADGRG1 is a member of the ADGRG subgroup of aGPCRs. Although GPR56 is best known for its pivotal role in the cerebral cortical development, it is also important for tumor progression. Numerous studies have revealed that GPR56 is expressed in various cancer types with a role in cancer cell adhesion, migration and metastasis. In a recent study, we found that the immobilized GPR56-specific CG4 antibody enhanced IL-6 production and migration ability of melanoma cells. In this review, we will summarize the current understanding of GPR56 function and discuss the activation and signaling mechanisms of GPR56 in melanoma cells.

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“…These structural features make aGPCRs plausible transducers for cellular responses to ECM interactions, and specific aGPCRs have emerged as mechanical receptors and as transducers of cell-matrix interactions ( Scholz et al, 2015 ; Petersen et al, 2015 ; Langenhan et al, 2016 ). Prior work has defined functions for several aGPCRs in the development of myelinating glial cells in both the peripheral and central nervous systems ( Monk et al, 2009 ; Petersen et al, 2015 ; Ackerman et al, 2015 ; Giera et al, 2015 ; Langenhan et al, 2016 ; Shin et al, 2013 ; Ackerman et al, 2018 ). In particular, the aGPCR ADGRG1 (also known as GPR56) has been shown to regulate OL development and CNS myelination ( Ackerman et al, 2015 ; Giera et al, 2015 ; Salzman et al, 2016 ).…”

Section: Introductionmentioning

confidence: 99%

Microglial transglutaminase-2 drives myelination and myelin repair via GPR56/ADGRG1 in oligodendrocyte precursor cells

Giera

Luo

Ying

et al. 2018

eLife

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111

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In the central nervous system (CNS), myelin formation and repair are regulated by oligodendrocyte (OL) lineage cells, which sense and integrate signals from their environment, including from other glial cells and the extracellular matrix (ECM). The signaling pathways that coordinate this complex communication, however, remain poorly understood. The adhesion G protein-coupled receptor ADGRG1 (also known as GPR56) is an evolutionarily conserved regulator of OL development in humans, mice, and zebrafish, although its activating ligand for OL lineage cells is unknown. Here, we report that microglia-derived transglutaminase-2 (TG2) signals to ADGRG1 on OL precursor cells (OPCs) in the presence of the ECM protein laminin and that TG2/laminin-dependent activation of ADGRG1 promotes OPC proliferation. Signaling by TG2/laminin to ADGRG1 on OPCs additionally improves remyelination in two murine models of demyelination. These findings identify a novel glia-to-glia signaling pathway that promotes myelin formation and repair, and suggest new strategies to enhance remyelination.

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GPR56/ADGRG1 regulates development and maintenance of peripheral myelin

Abstract: This study identifies GPR56/ADGRG1 as a conserved regulator of peripheral nervous system myelination and defines a novel interacting partner of GPR56: plectin. This study is directly relevant to human health because mutations in GPR56 and PLECTIN cause diseases with neuropathic symptoms.

Cited by 53 publications

References 68 publications

Much More Than a Scaffold: Cytoskeletal Proteins in Neurological Disorders

Much More Than a Scaffold: Cytoskeletal Proteins in Neurological Disorders

The Activation and Signaling Mechanisms of GPR56/ADGRG1 in Melanoma Cell

Microglial transglutaminase-2 drives myelination and myelin repair via GPR56/ADGRG1 in oligodendrocyte precursor cells

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