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Synapse formation and growth are tightly controlled processes. How synaptic growth is terminated after reaching proper size remains unclear. Here, we show that Leon, the Drosophila USP5 deubiquitinase, controls postsynaptic growth. In leon mutants, postsynaptic specializations of neuromuscular junctions are dramatically expanded, including the subsynaptic reticulum, the postsynaptic density, and the glutamate receptor cluster. Expansion of these postsynaptic features is caused by a disruption of ubiquitin homeostasis with accumulation of free ubiquitin chains and ubiquitinated substrates in the leon mutant. Accumulation of Ubiquilin (Ubqn), the ubiquitin receptor whose human homolog ubiquilin 2 is associated with familial amyotrophic lateral sclerosis, also contributes to defects in postsynaptic growth and ubiquitin homeostasis. Importantly, accumulations of postsynaptic proteins cause different aspects of postsynaptic overgrowth in leon mutants. Thus, the deubiquitinase Leon maintains ubiquitin homeostasis and proper Ubqn levels, preventing postsynaptic proteins from accumulation to confine postsynaptic growth.
Pubmed ID: 28489002 RIS Download
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View all literature mentionsThis monoclonal targets Glutamate receptor subunit, DGluR-IIA
View all literature mentionsThis monoclonal targets Human UBQLN2
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View all literature mentionsThis monoclonal targets cysteine string protein (CSP), Drosophila common
View all literature mentionsThis monoclonal targets Mouse Drosophila synapsin
View all literature mentionsThis monoclonal targets Discs large (Drosophila)
View all literature mentionsDrosophila melanogaster with name w[*]; P{w[+mW.hs]=GawB}D42 from BDSC.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD4689}v41136/TM3 from Flybase.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD6741}v17567 from Flybase.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD4245}v30497 from Flybase.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD11310}v40512 from Flybase.
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View all literature mentionsCell line Schneider 2 is a Spontaneously immortalized cell line with a species of origin Drosophila melanogaster
View all literature mentionsDrosophila melanogaster with name Pak[6]/TM3, Sb[1] Ser[1] from BDSC.
View all literature mentionsDrosophila melanogaster with name w[*]; P{w[+mW.hs]=GAL4-da.G32}UH1, Sb[1]/TM6B, Tb[1] from BDSC.
View all literature mentionsDrosophila melanogaster with name w[*]; P{w[+mW.hs]=GawB}how[24B] from BDSC.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD4689}v41136/TM3 from Flybase.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD11310}v40512 from Flybase.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD4245}v30497 from Flybase.
View all literature mentionsDrosophila melanogaster with name y[1] w[*]; P{w[+mC]=UAS-mCD8::GFP.L}LL5, P{UAS-mCD8::GFP.L}2 from BDSC.
View all literature mentionsDrosophila melanogaster with name w[1118]; P{GD6741}v17567 from Flybase.
View all literature mentionsDrosophila melanogaster with name w[*]; P{w[+mW.hs]=GawB}D42 from BDSC.
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