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PMID: 10972374 Published · ppublish English Letter Research Support, Non-U.S. Gov't

Recombinant human alpha-glucosidase from rabbit milk in Pompe patients.

Lancet (London, England) ·Vol. 356 ·No. 9227 ·2000-07-29 ·Pages 397-8

Van den Hout H, Reuser AJ, Vulto AG, Loonen MC, Cromme-Dijkhuis A, Van der Ploeg AT

Abstract

Pompe's disease is a fatal muscular disorder caused by lysosomal alpha-glucosidase deficiency. In an open-label study, four babies with characteristic cardiomyopathy were treated with recombinant human alpha-glucosidase (rhGAA) from rabbit milk at starting doses of 15 mg/kg or 20 mg/kg, and later 40 mg/kg. The enzyme was generally well tolerated. Activity of alpha-glucosidase normalised in muscle. Tissue morphology and motor and cardiac function improved. The left-ventricular-mass index decreased significantly. We recommend early treatment. Long-term effects are being studied.

MeSH Terms
Animals Animals, Genetically Modified Cardiomyopathy, Hypertrophic/complications,drug therapy Glycogen Storage Disease Type II/complications,drug therapy Humans Milk/chemistry Rabbits Recombinant Proteins/isolation & purification,therapeutic use alpha-Glucosidases/isolation & purification,therapeutic use
Chemicals
Recombinant Proteins alpha-Glucosidases
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Van den Hout H
Reuser A J
Vulto A G
Loonen M C
Cromme-Dijkhuis A
Van der Ploeg A T
Article Info
Journal
Lancet (London, England)
Abbr.
Lancet
ISSN
0140-6736
Published
2000-07-29
Pages
397-8
Language
English
Region
England
NLM ID
2985213R
Subset
IM
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