Document Detail


The diagnosis of HPRT deficiency in the 21st century.
MedLine Citation:
PMID:  18600505     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We have studied 36 patients with HPRT deficiency, 25 with Lesch-Nyhan syndrome and 11 with partial HPRT deficiency (grades 1 to 3). Patients diagnosed with HPRT deficiency have increased 50% since 2000. The most relevant recent advances have been made in molecular diagnosis. Nevertheless, enzyme determinations are still essential for the diagnosis of HPRT deficiency. Therapy for the neurological manifestations of HPRT deficiency has not advanced. Allopurinol remains the drug of choice to diminish uric acid overproduction, but the optimal allopurinol dose must be established in each patient to prevent xanthine or uric acid urolithiasis, a process aided by sequential determination of urinary oxypurines and uric acid.
Authors:
R J Torres; J G Puig
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Nucleosides, nucleotides & nucleic acids     Volume:  27     ISSN:  1532-2335     ISO Abbreviation:  Nucleosides Nucleotides Nucleic Acids     Publication Date:  2008 Jun 
Date Detail:
Created Date:  2008-07-07     Completed Date:  2008-10-30     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  100892832     Medline TA:  Nucleosides Nucleotides Nucleic Acids     Country:  United States    
Other Details:
Languages:  eng     Pagination:  564-9     Citation Subset:  IM    
Affiliation:
Division of Clinical Biochemistry, La Paz University Hospital, Madrid, Spain. rtorres.hulp@salud.madrid.org
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Allopurinol / therapeutic use
Child, Preschool
Humans
Hypoxanthine Phosphoribosyltransferase / deficiency*,  genetics,  metabolism*
Lesch-Nyhan Syndrome / diagnosis*,  drug therapy,  enzymology,  metabolism
Mutation
Purines / metabolism
Chemical
Reg. No./Substance:
0/Purines; 120-73-0/purine; 315-30-0/Allopurinol; EC 2.4.2.8/Hypoxanthine Phosphoribosyltransferase

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


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