Literature DB >> 2136385

HEMPAS disease: genetic defect of glycosylation.

M N Fukuda1.   

Abstract

Congenital dyserythropoietic anaemia Type II or HEMPAS (hereditary erythroblastic multinuclearity with positive acidified serum lysis test) is a rare genetic anaemia in humans, inherited in an autosomally recessive mode. Biochemical analyses of HEMPAS erythrocyte membranes suggested strongly that HEMPAS is caused by defective glycosylation of erythrocyte membrane glycoproteins. Most recently a HEMPAS case has been identified as being defective in the gene encoding Golgi alpha-mannosidase II by using cDNA probe of alpha-mannosidase II. At present, it is not clear whether HEMPAS is a genetically heterogenous collection of glycosylation deficiencies, as some HEMPAS cases showed a low level of N-acetylglucosaminyltransferase II. Abnormal glycosylation of serum glycoproteins and association of liver cirrhosis in HEMPAS patients indicate that HEMPAS disease is not restricted to erythroid cells. On the other hand, normal development of HEMPAS patients during embryonic stage strongly suggests the possibilities of fetal type isozyme in place of defective glycosylation enzyme.

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Year:  1990        PMID: 2136385     DOI: 10.1093/glycob/1.1.9

Source DB:  PubMed          Journal:  Glycobiology        ISSN: 0959-6658            Impact factor:   4.313


  14 in total

1.  Localization of the congenital dyserythropoietic anemia II locus to chromosome 20q11.2 by genomewide search.

Authors:  P Gasparini; E Miraglia del Giudice; J Delaunay; A Totaro; M Granatiero; S Melchionda; L Zelante; A Iolascon
Journal:  Am J Hum Genet       Date:  1997-11       Impact factor: 11.025

Review 2.  A retrospective and prospective view of glycopathology.

Authors:  A Kobata
Journal:  Glycoconj J       Date:  1998-04       Impact factor: 2.916

3.  Determination of glycan structures and molecular masses of the glycovariants of serum transferrin from a patient with carbohydrate deficient syndrome type II.

Authors:  B Coddeville; H Carchon; J Jaeken; G Briand; G Spik
Journal:  Glycoconj J       Date:  1998-03       Impact factor: 2.916

Review 4.  Golgi glycosylation and human inherited diseases.

Authors:  Hudson H Freeze; Bobby G Ng
Journal:  Cold Spring Harb Perspect Biol       Date:  2011-09-01       Impact factor: 10.005

Review 5.  The carbohydrate-deficient glycoprotein syndromes: an overview.

Authors:  J Jaeken; H Carchon
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

6.  Molecular cloning and expression of cDNAs encoding human alpha-mannosidase II and a previously unrecognized alpha-mannosidase IIx isozyme.

Authors:  M Misago; Y F Liao; S Kudo; S Eto; M G Mattei; K W Moremen; M N Fukuda
Journal:  Proc Natl Acad Sci U S A       Date:  1995-12-05       Impact factor: 11.205

7.  A novel disorder caused by defective biosynthesis of N-linked oligosaccharides due to glucosidase I deficiency.

Authors:  G J Gerwig; E Bause; L K Nuytinck; J F Vliegenthart; W Breuer; J P Kamerling; M F Espeel; J J Martin; N W Chan; G A Dacremont
Journal:  Am J Hum Genet       Date:  2000-04-28       Impact factor: 11.025

Review 8.  A journey to the world of glycobiology.

Authors:  A Kobata
Journal:  Glycoconj J       Date:  2000 Jul-Sep       Impact factor: 2.916

9.  Carbohydrate-deficient glycoprotein syndrome: not an N-linked oligosaccharide processing defect, but an abnormality in lipid-linked oligosaccharide biosynthesis?

Authors:  L D Powell; K Paneerselvam; R Vij; S Diaz; A Manzi; N Buist; H Freeze; A Varki
Journal:  J Clin Invest       Date:  1994-11       Impact factor: 14.808

10.  Isolation of a mutant Arabidopsis plant that lacks N-acetyl glucosaminyl transferase I and is unable to synthesize Golgi-modified complex N-linked glycans.

Authors:  A von Schaewen; A Sturm; J O'Neill; M J Chrispeels
Journal:  Plant Physiol       Date:  1993-08       Impact factor: 8.340

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