Literature DB >> 11405345

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

J M Van den Hout1, A J Reuser, J B de Klerk, W F Arts, J A Smeitink, A T Van der Ploeg.   

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.

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Year:  2001        PMID: 11405345     DOI: 10.1023/a:1010383421286

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  12 in total

1.  AN ELECTRON MICROSCOPIC AND BIOCHEMICAL STUDY OF TYPE II GLYCOGENOSIS.

Authors:  P BAUDHUIN; H G HERS; H LOEB
Journal:  Lab Invest       Date:  1964-09       Impact factor: 5.662

2.  Glycogen-storage disease of the myocardium with obstruction to left ventricular outflow.

Authors:  K H EHLERS; J W HAGSTROM; D S LUKAS; S F REDO; M A ENGLE
Journal:  Circulation       Date:  1962-01       Impact factor: 29.690

Review 3.  Enzyme replacement in Pompe disease: an attempt with purified human acid alpha-glucosidase.

Authors:  T de Barsy; P Jacquemin; F Van Hoof; H G Hers
Journal:  Birth Defects Orig Artic Ser       Date:  1973-03

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

Authors:  H Van den Hout; A J Reuser; A G Vulto; M C Loonen; A Cromme-Dijkhuis; A T Van der Ploeg
Journal:  Lancet       Date:  2000-07-29       Impact factor: 79.321

5.  Biochemical, immunological, and cell genetic studies in glycogenosis type II.

Authors:  A J Reuser; J F Koster; A Hoogeveen; H Galjaard
Journal:  Am J Hum Genet       Date:  1978-03       Impact factor: 11.025

6.  alpha-Glucosidase deficiency in generalized glycogenstorage disease (Pompe's disease).

Authors:  H G HERS
Journal:  Biochem J       Date:  1963-01       Impact factor: 3.857

Review 7.  Left ventricular myocardial mass determined by cross-sectional echocardiography in normal newborns, infants, and children.

Authors:  M Vogel; W Staller; K Bühlmeyer
Journal:  Pediatr Cardiol       Date:  1991-07       Impact factor: 1.655

Review 8.  Glycogenosis type II (acid maltase deficiency).

Authors:  A J Reuser; M A Kroos; M M Hermans; A G Bijvoet; M P Verbeet; O P Van Diggelen; W J Kleijer; A T Van der Ploeg
Journal:  Muscle Nerve Suppl       Date:  1995

9.  Enzyme therapy in type 1 Gaucher disease: comparative efficacy of mannose-terminated glucocerebrosidase from natural and recombinant sources.

Authors:  G A Grabowski; N W Barton; G Pastores; J M Dambrosia; T K Banerjee; M A McKee; C Parker; R Schiffmann; S C Hill; R O Brady
Journal:  Ann Intern Med       Date:  1995-01-01       Impact factor: 25.391

10.  Lysosomes in type II glycogenosis. Changes during administration of extract from Aspergillus niger.

Authors:  G Hug; W K Schubert
Journal:  J Cell Biol       Date:  1967-10       Impact factor: 10.539

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  40 in total

Review 1.  Inhibition of substrate synthesis as a strategy for glycolipid lysosomal storage disease therapy.

Authors:  F M Platt; M Jeyakumar; U Andersson; D A Priestman; R A Dwek; T D Butters; T M Cox; R H Lachmann; C Hollak; J M Aerts; S Van Weely; M Hrebícek; C Moyses; I Gow; D Elstein; A Zimran
Journal:  J Inherit Metab Dis       Date:  2001-04       Impact factor: 4.982

Review 2.  Newborn screening for neuropathic lysosomal storage disorders.

Authors:  Wuh-Liang Hwu; Yin-Hsiu Chien; Ni-Chung Lee
Journal:  J Inherit Metab Dis       Date:  2010-06-08       Impact factor: 4.982

3.  A modified PAS stain combined with immunofluorescence for quantitative analyses of glycogen in muscle sections.

Authors:  Gert Schaart; Reinout P Hesselink; Hans A Keizer; Gerrit van Kranenburg; Maarten R Drost; Matthijs K C Hesselink
Journal:  Histochem Cell Biol       Date:  2004-08-03       Impact factor: 4.304

Review 4.  Treatment of lysosomal storage disorders : progress with enzyme replacement therapy.

Authors:  Marianne Rohrbach; Joe T R Clarke
Journal:  Drugs       Date:  2007       Impact factor: 9.546

5.  Will GM animals follow the GM plant fate?

Authors:  Núria Vàzquez-Salat; Louis-Marie Houdebine
Journal:  Transgenic Res       Date:  2012-09-18       Impact factor: 2.788

6.  Therapeutic neonatal hepatic gene therapy in mucopolysaccharidosis VII dogs.

Authors:  Katherine Parker Ponder; John R Melniczek; Lingfei Xu; Margaret A Weil; Thomas M O'Malley; Patricia A O'Donnell; Van W Knox; Gustavo D Aguirre; Hamutal Mazrier; N Matthew Ellinwood; Meg Sleeper; Albert M Maguire; Susan W Volk; Robert L Mango; Jean Zweigle; John H Wolfe; Mark E Haskins
Journal:  Proc Natl Acad Sci U S A       Date:  2002-09-13       Impact factor: 11.205

7.  Induction of tolerance to a recombinant human enzyme, acid alpha-glucosidase, in enzyme deficient knockout mice.

Authors:  Nina Raben; Kanneboyina Nagaraju; Alicia Lee; Nina Lu; Yesenia Rivera; Tejas Jatkar; John J Hopwood; Paul H Plotz
Journal:  Transgenic Res       Date:  2003-04       Impact factor: 2.788

8.  Adult onset glycogen storage disease type II (adult onset Pompe disease): report and magnetic resonance images of two cases.

Authors:  Andrew Del Gaizo; Sima Banerjee; Michael Terk
Journal:  Skeletal Radiol       Date:  2009-12       Impact factor: 2.199

9.  Restoration of muscle functionality by genetic suppression of glycogen synthesis in a murine model of Pompe disease.

Authors:  Gaelle Douillard-Guilloux; Nina Raben; Shoichi Takikita; Arnaud Ferry; Alban Vignaud; Isabelle Guillet-Deniau; Maryline Favier; Beth L Thurberg; Peter J Roach; Catherine Caillaud; Emmanuel Richard
Journal:  Hum Mol Genet       Date:  2009-12-03       Impact factor: 6.150

Review 10.  Enzyme replacement and enhancement therapies for lysosomal diseases.

Authors:  R J Desnick
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

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