The diagnosis of HPRT deficiency in the 21st century

被引:7
作者
Torres, R. J.
Puig, J. G.
机构
[1] La Paz Univ Hosp, Div Clin Biochem, Madrid, Spain
[2] La Paz Univ Hosp, Div Internal Med, Madrid, Spain
关键词
Lesch-Nyhan syndrome; HPRT; molecular diagnosis; allopurinol; urolithiasis;
D O I
10.1080/15257770802135778
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
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.
引用
收藏
页码:564 / 569
页数:6
相关论文
共 8 条
[1]   Normal HPRT coding region in a male with gout due to HPRT deficiency [J].
Dawson, PA ;
Gordon, RB ;
Keough, DT ;
Emmerson, BT .
MOLECULAR GENETICS AND METABOLISM, 2005, 85 (01) :78-80
[2]   Delineation of the motor disorder of Lesch-Nyhan disease [J].
Jinnah, HA ;
Visser, JE ;
Harris, JC ;
Verdu, A ;
Larovere, L ;
Ceballos-Picot, I ;
Gonzalez-Alegre, P ;
Neychev, V ;
Torres, RJ ;
Dulac, O ;
Desguerre, I ;
Schretlen, DJ ;
Robey, KL ;
Barabas, G ;
Bloem, BR ;
Nyhan, W ;
De Kremer, R ;
Eddey, GE ;
Puig, JG ;
Reich, SG .
BRAIN, 2006, 129 :1201-1217
[3]   A SPECIFIC ENZYME DEFECT IN GOUT ASSOCIATED WITH OVERPRODUCTION OF URIC ACID [J].
KELLEY, WN ;
ROSENBLO.FM ;
HENDERSO.JF ;
SEEGMILL.JE .
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA, 1967, 57 (06) :1735-+
[4]   FAMILIAL DISORDER OF URIC ACID METABOLISM + CENTRAL NERVOUS SYSTEM FUNCTION [J].
LESCH, M ;
NYHAN, WL .
AMERICAN JOURNAL OF MEDICINE, 1964, 36 (04) :561-&
[5]   The spectrum of hypoxanthine-guanine phosphoribosyltransferase (HPRT) deficiency -: Clinical experience based on 22 patients front 18 Spanish families [J].
Puig, JG ;
Torres, RJ ;
Mateos, FA ;
Ramos, TH ;
Arcas, JM ;
Buño, AS ;
O'Neill, P .
MEDICINE, 2001, 80 (02) :102-112
[6]   ENZYME DEFECT ASSOCIATED WITH A SEX-LINKED HUMAN NEUROLOGICAL DISORDER AND EXCESSIVE PURINE SYNTHESIS [J].
SEEGMILLER, JE ;
ROSENBLOOM, FM ;
KELLEY, WN .
SCIENCE, 1967, 155 (3770) :1682-+
[7]  
Torres RJ, 2000, HUM MUTAT, V15, P383, DOI 10.1002/(SICI)1098-1004(200004)15:4<383::AID-HUMU17>3.0.CO
[8]  
2-2