Literature DB >> 21477922

24-months results in two adults with Pompe disease on enzyme replacement therapy.

Stefan Vielhaber1, Andrea Brejova, Grazyna Debska-Vielhaber, Joern Kaufmann, Helmut Feistner, Mircea A Schoenfeld, Friedemann Awiszus.   

Abstract

OBJECTIVE: Late-onset Pompe disease is a slowly progressive disorder resulting from deficiency of lysosomal acid alpha-glucosidase (GAA). Since 2006, an intravenous enzyme replacement therapy (ERT) with Myozyme™ (alglucosidase alfa) is available but long-term experience with ERT in late-onset Pompe disease is still limited.
METHODS: Two adult patients with impaired walking ability and disease duration of 10 and 13 years, respectively received ERT over a period of 24 months. Clinical and functional parameters including dynamometer-based assessment of proximal muscle strength were registered longitudinally.
RESULTS: In both patients some gain in function and physical endurance could be observed which was collaborated by stable dynamometer tests. No serious adverse events occurred and no patient required de novo ventilation.
CONCLUSION: The clinical results from our data base imply that long term enzyme replacement therapy seems to somewhat affect functionality and quality of life and can stabilize the otherwise progressive disease course in patients with late-onset Pompe disease.
Copyright © 2010 Elsevier B.V. All rights reserved.

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Year:  2011        PMID: 21477922     DOI: 10.1016/j.clineuro.2010.09.016

Source DB:  PubMed          Journal:  Clin Neurol Neurosurg        ISSN: 0303-8467            Impact factor:   1.876


  13 in total

1.  36 months observational clinical study of 38 adult Pompe disease patients under alglucosidase alfa enzyme replacement therapy.

Authors:  Caroline Regnery; Cornelia Kornblum; Frank Hanisch; Stefan Vielhaber; Nicola Strigl-Pill; Birgit Grunert; Wolfgang Müller-Felber; Franz Xaver Glocker; Matthias Spranger; Marcus Deschauer; Eugen Mengel; Benedikt Schoser
Journal:  J Inherit Metab Dis       Date:  2012-01-31       Impact factor: 4.982

2.  In vivo bone architecture in pompe disease using high-resolution peripheral computed tomography.

Authors:  Aneal Khan; Zachary Weinstein; David A Hanley; Robin Casey; Colleen McNeil; Barbara Ramage; Steven Boyd
Journal:  JIMD Rep       Date:  2012-06-06

3.  Ten years of the international Pompe survey: patient reported outcomes as a reliable tool for studying treated and untreated children and adults with non-classic Pompe disease.

Authors:  J C van der Meijden; D Güngör; M E Kruijshaar; A D J Muir; H A Broekgaarden; A T van der Ploeg
Journal:  J Inherit Metab Dis       Date:  2014-08-12       Impact factor: 4.982

Review 4.  Enzyme replacement therapy in late-onset Pompe disease: a systematic literature review.

Authors:  Antonio Toscano; Benedikt Schoser
Journal:  J Neurol       Date:  2012-08-28       Impact factor: 4.849

Review 5.  [Ultrasound of muscular diseases in children and adolescents].

Authors:  L von Rohden; Julian H W Jürgens
Journal:  Radiologe       Date:  2017-12       Impact factor: 0.635

Review 6.  The clinical relevance of outcomes used in late-onset Pompe disease: can we do better?

Authors:  Robin Lachmann; Benedikt Schoser
Journal:  Orphanet J Rare Dis       Date:  2013-10-12       Impact factor: 4.123

7.  Forced vital capacity and cross-domain late-onset Pompe disease outcomes: an individual patient-level data meta-analysis.

Authors:  Kenneth I Berger; Steve Kanters; Jeroen P Jansen; Andrew Stewart; Susan Sparks; Kristina An Haack; Anna Bolzani; Gaye Siliman; Alaa Hamed
Journal:  J Neurol       Date:  2019-06-11       Impact factor: 4.849

8.  Muscle imaging data in late-onset Pompe disease reveal a correlation between the pre-existing degree of lipomatous muscle alterations and the efficacy of long-term enzyme replacement therapy.

Authors:  Kai Michael Gruhn; Christoph Malte Heyer; Anne-Katrin Güttsches; Robert Rehmann; Volkmar Nicolas; Tobias Schmidt-Wilcke; Martin Tegenthoff; Matthias Vorgerd; Rudolf Andre Kley
Journal:  Mol Genet Metab Rep       Date:  2015-04-21

9.  Quality of life and participation in daily life of adults with Pompe disease receiving enzyme replacement therapy: 10 years of international follow-up.

Authors:  Deniz Güngör; Michelle E Kruijshaar; Iris Plug; Dimitris Rizopoulos; Tim A Kanters; Stephan C A Wens; Arnold J J Reuser; Pieter A van Doorn; Ans T van der Ploeg
Journal:  J Inherit Metab Dis       Date:  2015-11-03       Impact factor: 4.982

Review 10.  Patient and observer reported outcome measures to evaluate health-related quality of life in inherited metabolic diseases: a scoping review.

Authors:  Carlota Pascoal; Sandra Brasil; Rita Francisco; Dorinda Marques-da-Silva; Agnes Rafalko; Jaak Jaeken; Paula A Videira; Luísa Barros; Vanessa Dos Reis Ferreira
Journal:  Orphanet J Rare Dis       Date:  2018-11-28       Impact factor: 4.123

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