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August 22, 2024FEBS Letters

Structural basis of sugar recognition by SCFFBS2 ubiquitin ligase involved in NGLY1 deficiency

Tadashi Satoh Maho Yagi‐Utsumi Nozomi Ishii Tsunehiro Mizushima Hirokazu Yagi Ryuichi Kato Yuriko Tachida Hiroaki Tateno Ichiro Matsuo Koichi Kato Tadashi Suzuki Yukiko Yoshida

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Abstract

The cytosolic peptide:N-glycanase (PNGase) is involved in the quality control of N-glycoproteins via the endoplasmic reticulum-associated degradation (ERAD) pathway. Mutations in the gene encoding cytosolic PNGase (NGLY1 in humans) cause NGLY1 deficiency. Recent findings indicate that the F-box protein FBS2 of the SCF

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Satoh et al. (2024) studied this question.

synapsesocial.com/papers/68e5b4e9b6db64358754dc6f https://doi.org/10.1002/1873-3468.15003

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Synapse has enriched 5 closely related papers on similar clinical questions. Consider them for comparative context:

1The role of N-glycans and their processing in ER-to-lysosome-associated degradation of disease-causing mutant NeuroserpinOriginal Research · 2026
2Mouse Models Reveal the Physiological Functions of Cytosolic Peptide:<i>N</i>-Glycanase, NGLY12024
3Functional and transcriptomic analyses in <i>Neurospora crassa</i> reveal a crucial role for the N‐glycoprotein deglycosylation process in fungal homeostasisOriginal Research · 2026
4The conserved NUS-1/DHDDS-1 complex links N-glycosylation to lipid and lysosomal homeostasis in C. elegansOriginal Research · 2026
5Development of a fluorescence and quencher-based FRET assay for detection of endogenous peptide:N-glycanase/NGLY1 activity2024 · 14 citations

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Molecular Modeling of N-Acetylglucosamine Binding to the I154R Mutant of NAGLU: Pathogenic Insights into Sanfilippo Syndrome Type B2026

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