Document Detail


Laronidase treatment of mucopolysaccharidosis I.
MedLine Citation:
PMID:  15691212     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The lysosomal storage disorder (LSD) mucopolysaccharidosis type I (MPS I, McKusick 25280, Hurler syndrome, Hurler-Scheie syndrome, Scheie syndrome) is caused by a deficiency in the lysosomal enzyme, alpha-L-iduronidase (EC 3.2.1.76). MPS I patients can present within a diverse clinical spectrum, ranging from classical Hurler syndrome to attenuated Scheie syndrome. Laronidase (Aldurazyme) enzyme replacement therapy has been developed as a treatment strategy for MPS I patients and has been approved for clinical practice. Here we review the pre-clinical studies and clinical trials that have been used to demonstrate that intravenous laronidase therapy is well tolerated and effective for treating MPS I patients who do not have neuronal pathology. Current challenges for a viable treatment strategy for all MPS I patients include development of an early screening protocol that identifies patients before the onset of irreversible pathology, methods to predict disease severity, appropriate treatment for neuropathology, and an effective patient monitoring regimen.
Authors:
Ed J Wraith; John J Hopwood; Maria Fuller; Peter J Meikle; Doug A Brooks
Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Review    
Journal Detail:
Title:  BioDrugs : clinical immunotherapeutics, biopharmaceuticals and gene therapy     Volume:  19     ISSN:  1173-8804     ISO Abbreviation:  BioDrugs     Publication Date:  2005  
Date Detail:
Created Date:  2005-02-04     Completed Date:  2005-06-14     Revised Date:  2007-11-15    
Medline Journal Info:
Nlm Unique ID:  9705305     Medline TA:  BioDrugs     Country:  New Zealand    
Other Details:
Languages:  eng     Pagination:  1-7     Citation Subset:  IM    
Affiliation:
Willink Biochemical Genetics Unit, Royal Manchester Children's Hospital, Manchester, UK.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Animals
Clinical Trials as Topic
Half-Life
Humans
Iduronidase / adverse effects,  pharmacokinetics,  therapeutic use*
Infusions, Intravenous
Mucopolysaccharidosis I / drug therapy*,  genetics,  metabolism
Chemical
Reg. No./Substance:
EC 3.2.1.76/Iduronidase

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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