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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