Lysosomal oligosaccharide catabolism (Homo sapiens)

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3, 16, 18711, 148, 12, 132, 9, 10, 15, 196cytosollysosomal lumenManGlcNAc:ManManMAN2B2:Zn2+Zn2+ MAN2C1 alpha-D-Man-(1->3)-MANBAMAN2C1:Zn2+MAN2B1 H2OH2OMAN2B1:Zn2+Zn2+ alpha-D-Man-(1->3)-alpha-D-Man-(1->3)-MAN2B2 alpha-D-Man-(1->2)-alpha-D-Man-(1->2)-alpha-D-Man-(1->3)-GlcNAcAsparagine N-linkedglycosylationZn2+ H2O1, 4, 5, 17


Description

N-Glycosylation is one of the most common co- and posttranslational modifications of eukaryotic proteins occurring in the ER lumen. N-glycosylation plays pivotal roles in protein folding and intra- or inter-cellular trafficking of N-glycosylated proteins. Quality control mechanisms in the ER sift out incorrectly-folded proteins from correctly-folded proteins, the former then destined for degradation. Incorrectly-folded N-glycans are exported to the cytosol where the process of degradation begins. Once the unfolded protein is cleaved from the oligosaccharide (forming free oligosaccharides, fOS), step-wise degradation of mannose moieties, both in the cytosol (Suzuki & Harada 2014) and then in the lysosome (Aronson & Kuranda 1989, Winchester 2005), results in complete degradation. Breakdown must be complete to avoid lysosomal storage diseases that occur when fragments as small as dimers are left undigested. View original pathway at Reactome.

Comments

Reactome-Converter 
Pathway is converted from Reactome ID: 8853383
Reactome-version 
Reactome version: 75
Reactome Author 
Reactome Author: Jassal, Bijay

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Bibliography

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  1. Apweiler R, Hermjakob H, Sharon N.; ''On the frequency of protein glycosylation, as deduced from analysis of the SWISS-PROT database.''; PubMed Europe PMC Scholia
  2. Liao YF, Lal A, Moremen KW.; ''Cloning, expression, purification, and characterization of the human broad specificity lysosomal acid alpha-mannosidase.''; PubMed Europe PMC Scholia
  3. Winchester B.; ''Lysosomal metabolism of glycoproteins.''; PubMed Europe PMC Scholia
  4. Hossler P, Goh LT, Lee MM, Hu WS.; ''GlycoVis: visualizing glycan distribution in the protein N-glycosylation pathway in mammalian cells.''; PubMed Europe PMC Scholia
  5. Shental-Bechor D, Levy Y.; ''Folding of glycoproteins: toward understanding the biophysics of the glycosylation code.''; PubMed Europe PMC Scholia
  6. Saint-Pol A, Bauvy C, Codogno P, Moore SE.; ''Transfer of free polymannose-type oligosaccharides from the cytosol to lysosomes in cultured human hepatocellular carcinoma HepG2 cells.''; PubMed Europe PMC Scholia
  7. Suzuki T, Hara I, Nakano M, Shigeta M, Nakagawa T, Kondo A, Funakoshi Y, Taniguchi N.; ''Man2C1, an alpha-mannosidase, is involved in the trimming of free oligosaccharides in the cytosol.''; PubMed Europe PMC Scholia
  8. Alkhayat AH, Kraemer SA, Leipprandt JR, Macek M, Kleijer WJ, Friderici KH.; ''Human beta-mannosidase cDNA characterization and first identification of a mutation associated with human beta-mannosidosis.''; PubMed Europe PMC Scholia
  9. Borgwardt L, Lund AM, Dali CI.; ''Alpha-mannosidosis - a review of genetic, clinical findings and options of treatment.''; PubMed Europe PMC Scholia
  10. Beccari T, Stinchi S, Orlacchio A.; ''Lysosomal alpha-D-mannosidase.''; PubMed Europe PMC Scholia
  11. Venkatesan M, Kuntz DA, Rose DR.; ''Human lysosomal alpha-mannosidases exhibit different inhibition and metal binding properties.''; PubMed Europe PMC Scholia
  12. Percheron F, Foglietti MJ, Bernard M, Ricard B.; ''Mammalian beta-D-mannosidase and beta-mannosidosis.''; PubMed Europe PMC Scholia
  13. Molho-Pessach V, Bargal R, Abramowitz Y, Doviner V, Ingber A, Raas-Rothschild A, Ne'eman Z, Zeigler M, Zlotogorski A.; ''Angiokeratoma corporis diffusum in human beta-mannosidosis: Report of a new case and a novel mutation.''; PubMed Europe PMC Scholia
  14. Park C, Meng L, Stanton LH, Collins RE, Mast SW, Yi X, Strachan H, Moremen KW.; ''Characterization of a human core-specific lysosomal {alpha}1,6-mannosidase involved in N-glycan catabolism.''; PubMed Europe PMC Scholia
  15. Berg T, King B, Meikle PJ, Nilssen Ø, Tollersrud OK, Hopwood JJ.; ''Purification and characterization of recombinant human lysosomal alpha-mannosidase.''; PubMed Europe PMC Scholia
  16. Harada Y, Masahara-Negishi Y, Suzuki T.; ''Cytosolic-free oligosaccharides are predominantly generated by the degradation of dolichol-linked oligosaccharides in mammalian cells.''; PubMed Europe PMC Scholia
  17. Lederkremer GZ.; ''Glycoprotein folding, quality control and ER-associated degradation.''; PubMed Europe PMC Scholia
  18. Aronson NN, Kuranda MJ.; ''Lysosomal degradation of Asn-linked glycoproteins.''; PubMed Europe PMC Scholia
  19. Riise Stensland HM, Frantzen G, Kuokkanen E, Buvang EK, Klenow HB, Heikinheimo P, Malm D, Nilssen Ø.; ''amamutdb.no: A relational database for MAN2B1 allelic variants that compiles genotypes, clinical phenotypes, and biochemical and structural data of mutant MAN2B1 in α-mannosidosis.''; PubMed Europe PMC Scholia

History

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CompareRevisionActionTimeUserComment
114730view16:21, 25 January 2021ReactomeTeamReactome version 75
113174view11:24, 2 November 2020ReactomeTeamReactome version 74
112402view15:34, 9 October 2020ReactomeTeamReactome version 73
101306view11:19, 1 November 2018ReactomeTeamreactome version 66
100843view20:50, 31 October 2018ReactomeTeamreactome version 65
100384view19:25, 31 October 2018ReactomeTeamreactome version 64
99931view16:09, 31 October 2018ReactomeTeamreactome version 63
99486view14:41, 31 October 2018ReactomeTeamreactome version 62 (2nd attempt)
99138view12:40, 31 October 2018ReactomeTeamreactome version 62
93284view11:19, 9 August 2017ReactomeTeamreactome version 61
86368view09:16, 11 July 2016ReactomeTeamNew pathway

External references

DataNodes

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NameTypeDatabase referenceComment
Asparagine N-linked glycosylationPathwayR-HSA-446203 (Reactome) N-linked glycosylation is the most important form of post-translational modification for proteins synthesized and folded in the Endoplasmic Reticulum (Stanley et al. 2009). An early study in 1999 revealed that about 50% of the proteins in the Swiss-Prot database at the time were N-glycosylated (Apweiler et al. 1999). It is now established that the majority of the proteins in the secretory pathway require glycosylation in order to achieve proper folding.
The addition of an N-glycan to a protein can have several roles (Shental-Bechor & Levy 2009). First, glycans enhance the solubility and stability of the proteins in the ER, the golgi and on the outside of the cell membrane, where the composition of the medium is strongly hydrophilic and where proteins, that are mostly hydrophobic, have difficulty folding properly. Second, N-glycans are used as signal molecules during the folding and transport process of the protein: they have the role of labels to determine when a protein must interact with a chaperon, be transported to the golgi, or targeted for degradation in case of major folding defects. Third, and most importantly, N-glycans on completely folded proteins are involved in a wide range of processes: they help determine the specificity of membrane receptors in innate immunity or in cell-to-cell interactions, they can change the properties of hormones and secreted proteins, or of the proteins in the vesicular system inside the cell.
All N-linked glycans are derived from a common 14-sugar oligosaccharide synthesized in the ER, which is attached co-translationally to a protein while this is being translated inside the reticulum. The process of the synthesis of this glycan, known as Synthesis of the N-glycan precursor or LLO, constitutes one of the most conserved pathways in eukaryotes, and has been also observed in some eubacteria. The attachment usually happens on an asparagine residue within the consensus sequence asparagine-X-threonine by an complex called oligosaccharyl transferase (OST).
After being attached to an unfolded protein, the glycan is used as a label molecule in the folding process (also known as Calnexin/Calreticulin cycle) (Lederkremer 2009). The majority of the glycoproteins in the ER require at least one glycosylated residue in order to achieve proper folding, even if it has been shown that a smaller portion of the proteins in the ER can be folded without this modification.
Once the glycoprotein has achieved proper folding, it is transported via the cis-Golgi through all the Golgi compartments, where the glycan is further modified according to the properties of the glycoprotein. This process involves relatively few enzymes but due to its combinatorial nature, can lead to several millions of different possible modifications. The exact topography of this network of reactions has not been established yet, representing one of the major challenges after the sequencing of the human genome (Hossler et al. 2006).
Since N-glycosylation is involved in an great number of different processes, from cell-cell interaction to folding control, mutations in one of the genes involved in glycan assembly and/or modification can lead to severe development problems (often affecting the central nervous system). All the diseases in genes involved in glycosylation are collectively known as Congenital Disorders of Glycosylation (CDG) (Sparks et al. 2003), and classified as CDG type I for the genes in the LLO synthesis pathway, and CDG type II for the others.
GlcNAc:ManMetaboliteCHEBI:71553 (ChEBI)
GlcNAcMetaboliteCHEBI:17411 (ChEBI)
H2OMetaboliteCHEBI:15377 (ChEBI)
MAN2B1 ProteinO00754 (Uniprot-TrEMBL)
MAN2B1:Zn2+ComplexR-HSA-8853668 (Reactome)
MAN2B2 ProteinQ9Y2E5 (Uniprot-TrEMBL)
MAN2B2:Zn2+ComplexR-HSA-6799591 (Reactome)
MAN2C1 ProteinQ9NTJ4 (Uniprot-TrEMBL)
MAN2C1:Zn2+ComplexR-HSA-6799546 (Reactome)
MANBAProteinO00462 (Uniprot-TrEMBL)
ManMetaboliteCHEBI:4208 (ChEBI)
Zn2+ MetaboliteCHEBI:29105 (ChEBI)
alpha-D-Man-(1->2)-alpha-D-Man-(1->2)-alpha-D-Man-(1->3)-MetaboliteCHEBI:91284 (ChEBI)
alpha-D-Man-(1->3)-MetaboliteCHEBI:91279 (ChEBI)
alpha-D-Man-(1->3)-MetaboliteCHEBI:91280 (ChEBI)

Annotated Interactions

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SourceTargetTypeDatabase referenceComment
GlcNAc:ManArrowR-HSA-6799581 (Reactome)
GlcNAc:ManR-HSA-8853710 (Reactome)
GlcNAcArrowR-HSA-8853710 (Reactome)
H2OR-HSA-6799545 (Reactome)
H2OR-HSA-6799581 (Reactome)
H2OR-HSA-8853686 (Reactome)
MAN2B1:Zn2+mim-catalysisR-HSA-8853686 (Reactome)
MAN2B2:Zn2+mim-catalysisR-HSA-6799581 (Reactome)
MAN2C1:Zn2+mim-catalysisR-HSA-6799545 (Reactome)
MANBAmim-catalysisR-HSA-8853710 (Reactome)
ManArrowR-HSA-6799545 (Reactome)
ManArrowR-HSA-6799581 (Reactome)
ManArrowR-HSA-8853686 (Reactome)
ManArrowR-HSA-8853710 (Reactome)
R-HSA-6799545 (Reactome) Endoplasmic reticulum-associated degradation of misfolded glycoproteins ensures only functional, correctly folded proteins exit from the endoplasmic reticulum and that misfolded ones are targeted for lysosomal degradation. Misfolded glycoproteins are deglycosylated in the cytosol and the resultant free oligosaccharide can be further hydrolysed by alpha-mannosidase 2C1 (MAN2C1). For example, the oligosaccharide structure GlcNAc (Man)9 is hydrolysed by MAN2C1 to GlcNAc (Man)5, an intermediate structure formed in the N-glycan precursor biosynthesis pathway (Suzuki et al. 2006).
R-HSA-6799581 (Reactome) Endoplasmic reticulum-associated degradation of misfolded glycoproteins ensures only functional, correctly folded proteins exit from the endoplasmic reticulum and that misfolded ones are targeted for lysosomal degradation. Once located to the lysosome, epididymis-specific alpha-mannosidase (MAN2B2) can hydrolyse nondegraded oligosaccharides such as GlcNAc (Man)3 to GlcNAc (Man)2 quickly and GlcNAc (Man)2 to GlcNAc:Man slowly (Park et al. 2005, Venkatesan et al. 2009). MAN2B2 is ubiquitously expressed and belongs to a broad alpha-mannosidase family called class 2 alpha-mannosidases (CAZy glycosylhydrolase family 38, GH38). GH38 members are found in the Golgi complex, lysosomes, and cytosol where they are involved in either glycoprotein biosynthesis or catabolism.
R-HSA-8853391 (Reactome) Free oligosaccharides with the structure GlcNAc (Man)5 are transported from the cytosol to the lysosomal lumen by an unknown mechanism (Saint-Pol et al. 1997).
R-HSA-8853686 (Reactome) Lysosomal alpha-mannosidase (MAN2B1) belongs to a broad alpha-mannosidase family called class 2 alpha-mannosidases (CAZy glycosylhydrolase family 38, GH38). GH38 members are found in the Golgi complex, lysosomes, and cytosol where they are involved in either glycoprotein biosynthesis or catabolism. MAN2B1 (Liao et al. 1996, Berg et al. 1999) is involved in the sequential hydrolysis of mannose oligosaccharides sent to lysosomes for degradation. From the non-reducing end, MAN2B1 catalyses the hydrolysis of alpha(1,2), alpha(1,3) and alpha(1,6) mannosidic linkages (Beccari et al. 1999). Defects in the MAN2B1 gene cause alpha-mannosidosis (MIM:248500), a rare lysosomal storage disease. The disorder is characterised by a range of clinical phenotypes, the major manifestations being mental impairment, hearing impairment, skeletal changes, and immunodeficiency (Borgwardt et al. 2014, Riise Stensland et al. 2015).
R-HSA-8853710 (Reactome) Beta-mannosidase (MANBA) is the final exoglycosidase in the degradation pathway for N-linked oligosaccharides of glycoproteins, cleaving the beta-mannoside linkage of the disaccharide Man-1,4-GlcNAc (Alkhayat et al. 1998, Percheron et al. 1992). Defects in MANBA causes beta-mannosidosis (MIM:248510), a lysosomal storage disease of glycoprotein catabolism. A wide range of symptoms are observed, dependent on age of onset. The disease is associated with various degrees of mental retardation in most of the cases, hearing loss and speech impairment, hypotonia, epilepsy and peripheral neuropathy (Alkhayat et al. 1998, Molho-Pessach et al. 2007).
alpha-D-Man-(1->2)-alpha-D-Man-(1->2)-alpha-D-Man-(1->3)-R-HSA-6799545 (Reactome)
alpha-D-Man-(1->3)-ArrowR-HSA-6799545 (Reactome)
alpha-D-Man-(1->3)-ArrowR-HSA-8853391 (Reactome)
alpha-D-Man-(1->3)-ArrowR-HSA-8853686 (Reactome)
alpha-D-Man-(1->3)-R-HSA-6799581 (Reactome)
alpha-D-Man-(1->3)-R-HSA-8853391 (Reactome)
alpha-D-Man-(1->3)-R-HSA-8853686 (Reactome)
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