N-glycan branching requirement in neuronal and postnatal viability

被引:65
作者
Ye, ZY [1 ]
Marth, JD [1 ]
机构
[1] Univ Calif San Diego, Glycobiol Res & Training Ctr, Howard Hughes Med Inst, Dept Cellular & Mol Med, La Jolla, CA 92093 USA
关键词
apoptosis; development; genetics; N-glycans; neurobiology;
D O I
10.1093/glycob/cwh069
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
The structural variations among extracellular N-glycans reflect the activity of glycosyltransferases and glycosidases that operate in the Golgi apparatus. More than other types of vertebrate glycans, N-glycans are highly branched oligosaccharides with multiple antennae linked to an underlying mannose core structure. The branching patterns of N-glycans consist of three types, termed high-mannose, hybrid, and complex. Though most extracellular mammalian N-glycans are of the complex type, some cells variably express hybrid and high-mannose forms. Nevertheless, a requirement for hybrid and complex N-glycan branching exists in embryonic development and postnatal function among mice and humans inheriting defective Mgat1 or Mgat2 alleles. The resulting defects in formation N-glycan branching patterns cause multiple abnormalities, including neurologic defects, and have inferred the presence of distinct functions for hybrid and complex N-glycan branches among different cell lineages. We have further explored N-glycan structure-function relationships in vivo by using Cre-loxP conditional mutagenesis to abolish hybrid and complex N-glycan branching specifically among neuronal cells. Our findings show that hybrid N-glycan branching is an essential posttranslational modification among neurons. Loss of Mgat1 resulted in a unique pattern of neuronal glycoprotein deficiency concurrent with caspase 3 activation and apoptosis. Such animals exhibited severe locomotor deficits, tremors, paralysis, and early postnatal death. Unexpectedly, neuronal Mgat2 deletion resulting in the loss of complex but not hybrid N-glycan branching was well tolerated without phenotypic markers of neuronal or locomotor dysfunction. Structural features associated with hybrid N-glycan branching comprise a requisite posttranslational modification to neuronal glycoproteins that permits normal cellular function and viability.
引用
收藏
页码:547 / 558
页数:12
相关论文
共 31 条
[1]  
BENDIAK B, 1987, J BIOL CHEM, V262, P5784
[2]   COMPLEX ASPARAGINE-LINKED OLIGOSACCHARIDES IN MGAT1-NULL EMBRYOS [J].
CAMPBELL, RM ;
METZLER, M ;
GRANOVSKY, M ;
DENNIS, JW ;
MARTH, JD .
GLYCOBIOLOGY, 1995, 5 (05) :535-543
[3]   Neutral N-glycans in adult rat brain tissue - Complete characterisation reveals fucosylated hybrid and complex structures [J].
Chen, YJ ;
Wing, DR ;
Guile, GR ;
Dwek, RA ;
Harvey, DJ ;
Zamze, S .
EUROPEAN JOURNAL OF BIOCHEMISTRY, 1998, 251 (03) :691-703
[4]   Alpha-mannosidase-II deficiency results in dyserythropoiesis and unveils an alternate pathway in oligosaccharide biosynthesis [J].
Chui, D ;
OhEda, M ;
Liao, YF ;
Panneerselvam, K ;
Lal, A ;
Marek, KW ;
Freeze, HH ;
Moremen, KW ;
Fukuda, MN ;
Marth, JD .
CELL, 1997, 90 (01) :157-167
[5]  
Clark RAC, 1998, J NEUROCHEM, V70, P2594
[6]   MOLECULAR-CLONING AND EXPRESSION OF CDNA-ENCODING THE RAT UDP-N-ACETYLGLUCOSAMINE-ALPHA-6-D-MANNOSIDE BETA-1,2-N-ACETYLGLUCOSAMINYLTRANSFERASE-II [J].
DAGOSTARO, GAF ;
ZINGONI, A ;
MORITZ, RL ;
SIMPSON, RJ ;
SCHACHTER, H ;
BENDIAK, B .
JOURNAL OF BIOLOGICAL CHEMISTRY, 1995, 270 (25) :15211-15221
[7]   Virus-assisted mapping of neural inputs to a feeding center in the hypothalamus [J].
DeFalco, J ;
Tomishima, M ;
Liu, HY ;
Zhao, C ;
Cai, XL ;
Marth, JD ;
Enquist, L ;
Friedman, JM .
SCIENCE, 2001, 291 (5513) :2608-2613
[8]  
HOESCHE C, 1993, J BIOL CHEM, V268, P26494
[9]   MICE LACKING N-ACETYLGLUCOSAMINYLTRANSFERASE-I ACTIVITY DIE AT MIDGESTATION, REVEALING AN ESSENTIAL ROLE FOR COMPLEX OR HYBRID N-LINKED CARBOHYDRATES [J].
IOFFE, E ;
STANLEY, P .
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA, 1994, 91 (02) :728-732
[10]   CARBOHYDRATE-DEFICIENT GLYCOPROTEIN-SYNDROME TYPE-II - A DEFICIENCY IN GOLGI LOCALIZED N-ACETYL-GLUCOSAMINYLTRANSFERASE-II [J].
JAEKEN, J ;
SCHACHTER, H ;
CARCHON, H ;
DECOCK, P ;
CODDEVILLE, B ;
SPIK, G .
ARCHIVES OF DISEASE IN CHILDHOOD, 1994, 71 (02) :123-127