Document Detail


Enzyme replacement therapy for mucopolysaccharidoses: opinions of patients and families.
MedLine Citation:
PMID:  18410781     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
OBJECTIVES: To assess the opinions of individuals with mucopolysaccharidoses (MPS) and their parents regarding the use of enzyme replacement therapy (ERT). STUDY DESIGN: A validated questionnaire, including hypothetical clinical scenarios about ERT for MPS, was distributed to members of MPS support groups in the United States and Australia. RESULTS: The questionnaire was completed by 249 MPS support group members. Overall, 92% were in favor of ERT where MPS causes severe physical problems but does not affect intellect, and 69% were in favor of ERT where the physical limitations are mild and intellect is spared. Only 47% were in favor of ERT where severe physical and intellectual problems are well established; however, 77% were in favor of ERT in this situation if treatment begun early prolongs life and improves quality of life. CONCLUSION: Most respondents were in favor of ERT for MPS, even where it would not alter the intellectual deterioration. The medical community has a responsibility to advocate for their patients in situations where ERT is appropriate and recognize the economic burden and "family function burden" ERT can incur.
Authors:
David J Coman; Ian M Hayes; Veronica Collins; Margaret Sahhar; J Ed Wraith; Martin B Delatycki
Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't     Date:  2008-02-20
Journal Detail:
Title:  The Journal of pediatrics     Volume:  152     ISSN:  1097-6833     ISO Abbreviation:  J. Pediatr.     Publication Date:  2008 May 
Date Detail:
Created Date:  2008-04-15     Completed Date:  2008-05-06     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  0375410     Medline TA:  J Pediatr     Country:  United States    
Other Details:
Languages:  eng     Pagination:  723-7     Citation Subset:  AIM; IM    
Affiliation:
Bruce Lefroy Centre for Genetic Health Research, Murdoch Childrens Research Institute, Victoria, Australia.
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MeSH Terms
Descriptor/Qualifier:
Adult
Australia
Child
Family / psychology*
Female
Health Care Surveys
Humans
Male
Mucopolysaccharidoses / drug therapy*,  psychology
Patient Acceptance of Health Care*
Sulfatases / adverse effects,  therapeutic use*
Treatment Outcome
United States
Chemical
Reg. No./Substance:
EC 3.1.6.-/Sulfatases

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


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