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PMID: 11567948 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

Successful treatment of carbohydrate deficient glycoprotein syndrome type 1b with oral mannose.

Archives of disease in childhood ·Vol. 85 ·No. 4 ·2001-10-00 ·Pages 339-40

Hendriksz CJ, McClean P, Henderson MJ, Keir DG, Worthington VC, Imtiaz F, Schollen E, Matthijs G, Winchester BG

Abstract

An Asian girl presented with failure to thrive, congenital hepatic fibrosis, protein losing enteropathy, and hypoglycaemia. Phosphomannose isomerase activity in skin fibroblasts was reduced. She is homozygous for a mutation, D131N, in the phosphomannose isomerase gene (PM1), consistent with the diagnosis of carbohydrate deficient glycoprotein syndrome type 1b. She responded to oral mannose treatment.

MeSH Terms
Administration, Oral Congenital Disorders of Glycosylation/drug therapy,enzymology,genetics Electrophoresis, Agar Gel Female Homozygote Humans Infant Mannose/administration & dosage Mannose-6-Phosphate Isomerase/deficiency,genetics Mutation Transferrin/chemistry
Chemicals
Transferrin Mannose-6-Phosphate Isomerase Mannose
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Hendriksz C J
Children's Liver and GI Unit, Department of Paediatrics, St James's University Hospital, Beckett Street, Leeds LS9 7TF, UK.
McClean P
Henderson M J
Keir D G
Worthington V C
Imtiaz F
Schollen E
Matthijs G
Winchester B G
Article Info
Journal
Archives of disease in childhood
Abbr.
Arch Dis Child
ISSN
1468-2044
Published
2001-10-00
Pages
339-40
Language
English
Region
England
NLM ID
0372434
PMCID
PMC1718944
Subset
IM
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