Literature DB >> 11996648

Enzyme replacement therapy for the mucopolysaccharide storage disorders.

Emil D Kakkis1.   

Abstract

The mucopolysaccharide storage disorders are a group of lysosomal storage disorders associated with deficiencies of lysosomal enzymes required for the normal sequential degradation of glycosaminoglycans, formerly known as mucopolysaccharides. The accumulation of glycosaminoglycans in a wide variety of tissues results in a complex and progressive disease leading to death in the first or second decade in most patients. Studies of enzyme replacement in animal models of mucopolysaccharide disorders have demonstrated the potential of parenterally administered enzyme to reduce glycosaminoglycan storage and microscopic pathology. Clinical studies of enzyme replacement therapy are currently underway for mucopolysaccharidosis I, mucopolysaccharidosis VI and mucopolysaccharidosis II. The complexity and heterogeneity of the mucopolysaccharide disorders provide significant challenges for clinical study design and evaluation. Innovative clinical development strategies may be needed to lower the development cost and time for complex rare disease therapies to assure that such patients receive therapies they deserve.

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Year:  2002        PMID: 11996648     DOI: 10.1517/13543784.11.5.675

Source DB:  PubMed          Journal:  Expert Opin Investig Drugs        ISSN: 1354-3784            Impact factor:   6.206


  13 in total

Review 1.  An overview of Korean patients with mucopolysaccharidosis and collaboration through the Asia Pacific MPS Network.

Authors:  Sung Yoon Cho; Young Bae Sohn; Dong-Kyu Jin
Journal:  Intractable Rare Dis Res       Date:  2014-08

2.  Enzyme replacement therapy and extended newborn screening for mucopolysaccharidoses: opinions of treating physicians.

Authors:  David J Coman; Ian M Hayes; Veronica Collins; Margaret Sahhar; J Ed Wraith; Martin B Delatycki
Journal:  JIMD Rep       Date:  2011-06-22

3.  Targeting of the CNS in MPS-IH using a nonviral transferrin-alpha-L-iduronidase fusion gene product.

Authors:  Mark J Osborn; Ron T McElmurry; Brandon Peacock; Jakub Tolar; Bruce R Blazar
Journal:  Mol Ther       Date:  2008-06-03       Impact factor: 11.454

4.  Sialic acid deposition impairs the utility of AAV9, but not peptide-modified AAVs for brain gene therapy in a mouse model of lysosomal storage disease.

Authors:  Yong Hong Chen; Kristin Claflin; James C Geoghegan; Beverly L Davidson
Journal:  Mol Ther       Date:  2012-05-15       Impact factor: 11.454

5.  Mucopolysaccharidosis I: Alpha-L-Iduronidase mutations in three Tunisian families.

Authors:  S Laradi; T Tukel; M Erazo; J Shabbeer; L Chkioua; S Khedhiri; S Ferchichi; M Chaabouni; A Miled; R J Desnick
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.750

6.  Enzyme replacement in a human model of mucopolysaccharidosis IVA in vitro and its biodistribution in the cartilage of wild type mice.

Authors:  Melita Dvorak-Ewell; Dan Wendt; Chuck Hague; Terri Christianson; Vish Koppaka; Danielle Crippen; Emil Kakkis; Michel Vellard
Journal:  PLoS One       Date:  2010-08-16       Impact factor: 3.240

7.  Mucopolysaccharidosis I, II, and VI: Brief review and guidelines for treatment.

Authors:  Roberto Giugliani; Andressa Federhen; Maria Verônica Muñoz Rojas; Taiane Vieira; Osvaldo Artigalás; Louise Lapagesse Pinto; Ana Cecília Azevedo; Angelina Acosta; Carmen Bonfim; Charles Marques Lourenço; Chong Ae Kim; Dafne Horovitz; Denize Bonfim; Denise Norato; Diane Marinho; Durval Palhares; Emerson Santana Santos; Erlane Ribeiro; Eugênia Valadares; Fábio Guarany; Gisele Rosone de Lucca; Helena Pimentel; Isabel Neves de Souza; Jordão Correa; José Carlos Fraga; José Eduardo Goes; José Maria Cabral; José Simionato; Juan Llerena; Laura Jardim; Liane Giuliani; Luiz Carlos Santana da Silva; Mara L Santos; Maria Angela Moreira; Marcelo Kerstenetzky; Márcia Ribeiro; Nicole Ruas; Patricia Barrios; Paulo Aranda; Rachel Honjo; Raquel Boy; Ronaldo Costa; Carolina Souza; Flavio F Alcantara; Silvio Gilberto A Avilla; Simone Fagondes; Ana Maria Martins
Journal:  Genet Mol Biol       Date:  2010-12-01       Impact factor: 1.771

8.  The potential investment impact of improved access to accelerated approval on the development of treatments for low prevalence rare diseases.

Authors:  Brigitta E Miyamoto; Emil D Kakkis
Journal:  Orphanet J Rare Dis       Date:  2011-07-06       Impact factor: 4.123

9.  CNS penetration of intrathecal-lumbar idursulfase in the monkey, dog and mouse: implications for neurological outcomes of lysosomal storage disorder.

Authors:  Pericles Calias; Mikhail Papisov; Jing Pan; Nancy Savioli; Vasily Belov; Yan Huang; Jason Lotterhand; Mary Alessandrini; Nan Liu; Alan J Fischman; Jan L Powell; Michael W Heartlein
Journal:  PLoS One       Date:  2012-01-18       Impact factor: 3.240

Review 10.  The cell biology of disease: lysosomal storage disorders: the cellular impact of lysosomal dysfunction.

Authors:  Frances M Platt; Barry Boland; Aarnoud C van der Spoel
Journal:  J Cell Biol       Date:  2012-11-26       Impact factor: 10.539

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