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. 2021 Jun 29;15:689208. doi: 10.3389/fnint.2021.689208

TABLE 1.

Summary of altered proteins and mRNAs involved in local translation in different nervous system pathologies.

Disease Protein/mRNA Altered mechanism on local translation Model system References
ALS/FTD TDP-43 Abnormal trafficking of futsch/Map1b mRNAs to neurites leading to cytoskeleton defects Drosophila model of ALS in vivo, ALS vs. control postmortem brain tissue Coyne et al., 2014
FUS Stress-mediated inhibition of intra-axonal translation HEK-293 cell line and zebrafish embryo spinal cords; mutant FUS mice and mouse hippocampal neuron culture Bosco et al., 2010; Lopez-Erauskin et al., 2018
C9orf72 mRNP granules mislocalization to neuritic compartments affecting to branching Rat spinal cord neuron culture, Drosophila model in vivo Burguete et al., 2015
SMA SMN Actb, Nrn1, Gap43, Anxa2 mislocalization altering neurite growth, presynaptic function, and cytoskeleton Repression of axonal mTOR through miR-183 NSC-34 cell line; mouse motor neuron culture Rat hippocampal, cortical, and motor neuron culture Rage et al., 2013; Fallini et al., 2014, 2016; Kye et al., 2014; Rihan et al., 2017
AD Mapt Mapt mislocalization to dendrites and hyperphosphorylation Mouse hippocampal neuron culture, WT vs. Tau KO mice; hippocampal neuron culture from WT and Fyn KO mice, HEK-293 cell line, WT, APP23 and FynCA Tg mouse models in vivo Kobayashi et al., 2017; Li and Gotz, 2017
Atf4 Atf4 recruitment to axons induced by Aβ oligomers, leading to neuronal death Rat hippocampal neuron culture, mouse model in vivo, AD and control postmortem human brain tissue Baleriola et al., 2014; Walker et al., 2018
PD UPR genes UPR altered. A compartment-dependent UPR involving local translation? α-synuclein Tg mice Hetz and Saxena, 2017
LRRK2 Deregulation of global eIF4E/4E-BP. Defects in axonal 4E-BP dependent translation? Axonal microRNAs deregulation? HEK-293 and SH-SY5Y cell lines; rat superior cervical ganglia neuron culture; rat cortical neuron culture, WTvs. LRRK2 KO mice Imai et al., 2008; Kumar et al., 2010; Natera-Naranjo et al., 2010; Herzig et al., 2011; Trancikova et al., 2012
PINK1/PARK2 Pumilio and Glorund/hnRNP-F displacement: altered translation in the mitochondrion surface (within axons?) due to PINK1/PARK2 mutations. Drosophila PINK1 model and HEK 293T cell line Mouton-Liger et al., 2017; Martinez et al., 2019
HD HTT Impaired dendritic levels of Actb mRNA, Ago2 protein and P-bodies Altered axonal BDNF delivery leading to neurotoxicity Homozygous Htt mutant cell lines (109Q/109Q); HeLa S3 cells Flagged-Htt590; rat cortical neuron culture and brain sections Gauthier et al., 2004; Savas et al., 2008; Ma et al., 2010, 2011; Savas et al., 2010
ASD and FXS FMRP Deregulation of local mRNAs linked to abnormal spine morphology and plasticity Upregulation of dendritic Dlg4 translation through inhibition of miR-125a with spine morphology defects Dendritic mTOR upregulation Rat hippocampal neuron culture; cortical neuron culture from Fmr1 KO mice; in vivo Fmr1 KO mouse model; Organotypic hippocampal slice cultures from Fmr1 KO mice Antar et al., 2004; Muddashetty et al., 2011; Pyronneau et al., 2017; Banerjee et al., 2018; Feuge et al., 2019
DS Dscam Upregulation of dendritic mRNA and protein levels with defects in dendrite branching Mouse hippocampal neuron culture from Ts1Cje mice, DS mouse model Alves-Sampaio et al., 2010
Bdnf Upregulation within dendrites leading to an aberrant activation of dendritic translation In vitro hippocampal dendrites from Ts1Cje mice Troca-Marin et al., 2011; Swanger and Bassell, 2013
Other Bdnf (depression and bipolar disorder) Dendritic Bdnf localization to dendrites disrupted Ts1 Cje mouse hippocampal neuron culture Swanger and Bassell, 2013
Pumilio-2 (epilepsy) Erroneous mRNAs localization to axons Upregulation of overall translation in axons with branching defects In vivo 5 month Pum2 gene-trap mice; Pum2 knockdown in primary dorsal root ganglion rat neuron culture and mouse model in vivo Follwaczny et al., 2017; Martinez et al., 2019

ALS, amyotrophic lateral sclerosis; FTD, frontotemporal dementia; SMA, spinal muscular atrophy; AD, Alzheimer’s disease; PD, Parkinson’s disease; HD, Huntington’s disease; ASD, autism spectrum disorders; FXS, fragile-X syndrome; DS: down syndrome.