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Literature summary for 2.4.1.109 extracted from

  • Nakamura, N.; Stalnaker, S.H.; Lyalin, D.; Lavrova, O.; Wells, L.; Panin, V.M.
    Drosophila Dystroglycan is a target of O-mannosyltransferase activity of two protein O-mannosyltransferases, Rotated Abdomen and Twisted (2010), Glycobiology, 20, 381-394.
    View publication on PubMedView publication on EuropePMC

Application

Application Comment Organism
medicine using Drosophila as model for investigating the mechanisms of human congenital muscular dystrophies (dystroglycanopathies) resulting from the abnormal glycosylation of alpha-dystroglycan Drosophila melanogaster

Protein Variants

Protein Variants Comment Organism
additional information mutants lacking or overexpressing one of the two or both O-mannosyltransferases Rotated Abdomen and Twisted show that both are required for in vivo production of high molecular mass dystroglycan, overexpression of only one isoform does not increase its amount over wild-type, lack of either one of the isoforms or both isoforms lacking double mutant do not produce the high molecular mass dystroglycan Drosophila melanogaster
yes mutants lacking or overexpressing one of the two or both O-mannosyltransferases Rotated Abdomen and Twisted show that both are required for in vivo production of high molecular mass dystroglycan, overexpression of only one isoform does not increase its amount over wild-type, lack of either one of the isoforms or both isoforms lacking double mutant do not produce the high molecular mass dystroglycan Drosophila melanogaster

Localization

Localization Comment Organism GeneOntology No. Textmining
microsome
-
Drosophila melanogaster
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-

Natural Substrates/ Products (Substrates)

Natural Substrates Organism Comment (Nat. Sub.) Natural Products Comment (Nat. Pro.) Rev. Reac.
dolichyl phosphate D-mannose + alpha-dystroglycan Drosophila melanogaster in vitro asssay with both enzyme isoforms (RT and TW), 20 mM Tris, pH 8.0, mercaptoethanol, EDTA, n-octyl-beta-D-thioglucoside, sugar donor is tritium-labeled, alpha-dystroglycan isoform C contains mucin-type domain that is target of O-mannose modifications in mammals, high grade of O-mannosylation, A isoform without noticeable O-mannosylation, rabbit alpha-dystroglycan as positive control shows lower O-mannosylation dolichyl phosphate + O-D-mannosyl-[alpha-dystroglycan]
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?

Organism

Organism UniProt Comment Textmining
Drosophila melanogaster
-
-
-

Source Tissue

Source Tissue Comment Organism Textmining
larva
-
Drosophila melanogaster
-

Substrates and Products (Substrate)

Substrates Comment Substrates Organism Products Comment (Products) Rev. Reac.
dolichyl phosphate D-mannose + alpha-dystroglycan in vitro asssay with both enzyme isoforms (RT and TW), 20 mM Tris, pH 8.0, mercaptoethanol, EDTA, n-octyl-beta-D-thioglucoside, sugar donor is tritium-labeled, alpha-dystroglycan isoform C contains mucin-type domain that is target of O-mannose modifications in mammals, high grade of O-mannosylation, A isoform without noticeable O-mannosylation, rabbit alpha-dystroglycan as positive control shows lower O-mannosylation Drosophila melanogaster dolichyl phosphate + O-D-mannosyl-[alpha-dystroglycan]
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?

Synonyms

Synonyms Comment Organism
Drosophila O-mannosyltransferase Rotated Abdomen
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Drosophila melanogaster
Drosophila O-mannosyltransferase Twisted
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Drosophila melanogaster
O-mannosyltransferase
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Drosophila melanogaster
RT
-
Drosophila melanogaster
TW
-
Drosophila melanogaster

General Information

General Information Comment Organism
malfunction defects in O-mannosylation are associated with muscular dystrophies (dystroglycanopathies) Drosophila melanogaster
physiological function O-mannosylation can modulate the ligand-binding activity of alpha-dystroglycan Drosophila melanogaster