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PMID: 11405345 Published · ppublish English Clinical Trial Journal Article Research Support, Non-U.S. Gov't

Enzyme therapy for pompe disease with recombinant human alpha-glucosidase from rabbit milk.

Journal of inherited metabolic disease ·Vol. 24 ·No. 2 ·2001-04-00 ·Pages 266-74

Van den Hout JM, Reuser AJ, de Klerk JB, Arts WF, Smeitink JA, Van der Ploeg AT

Abstract

Pompe disease is a metabolic myopathy caused by deficiency of lysosomal acid alpha-glucosidase. In this report we review the first 36 weeks of a clinical study on the safety and efficacy of enzyme therapy aimed at correcting the deficiency. Four patients with infantile Pompe disease were enrolled. They received recombinant human alpha-glucosidase from transgenic rabbit milk. The product is generally well tolerated and reaches the primary target tissues. Normalization of alpha-glucosidase activity in skeletal muscle was obtained and degradation of PAS-positive material was seen in tissue sections. The clinical condition of all patients improved. The effect on heart was most significant, with an impressive reduction of the left ventricular mass index (LVMI). Motor function improved. The positive preliminary results stimulate continuation and extension of efforts towards the realization of enzyme therapy for Pompe disease.

MeSH Terms
Animals Animals, Genetically Modified Female Glycogen Storage Disease Type II/drug therapy,pathology Humans Infant Infant, Newborn Male Milk/enzymology Muscle, Skeletal/enzymology Rabbits Recombinant Proteins/adverse effects,therapeutic use alpha-Glucosidases/adverse effects,therapeutic use
Chemicals
Recombinant Proteins alpha-Glucosidases
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Van den Hout J M
Department of Pediatrics, Sophia Children's Hospital, University Hospital Rotterdam, The Netherlands. vanderploeg@alkg.azr.nl
Reuser A J
de Klerk J B
Arts W F
Smeitink J A
Van der Ploeg A T
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Article Info
Journal
Journal of inherited metabolic disease
Abbr.
J Inherit Metab Dis
ISSN
0141-8955
Published
2001-04-00
Pages
266-74
Language
English
Region
United States
NLM ID
7910918
Subset
IM
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