Document Detail


A case of familial renal hypouricemia associated with increased secretion of para-aminohippurate and idiopathic edema.
MedLine Citation:
PMID:  7099326     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A 42-year-old housewife had hypouricemia (serum uric acid ranging from 0.5 to 1.5 mg/dl; 30-89 mumol/l), increased uric acid clearance (47.6-83.0 ml/min), increased maximum tubular secretory capacity for para-aminohippurate, and idiopathic edema. Urate excretion was only minimally suppressed by pyrazinamide, and paradoxically decreased by probenecid. Uric acid clearance did not show any appreciable change after long-term administration of ticrynafen. In response to an increment of extracellular volume by hypertonic saline infusion or long-term 9 alpha-fluorohydrocortisone administration, urate clearance did not show any substantial increase. These data may suggest that not only presecretory but possibly also postsecretory reabsorption of urate is impaired in this patient. No other renal tubular abnormalities were detected. Family study revealed that her renal hypouricemia is hereditary. She was unable to increase urinary excretion of sodium during hypertonic saline infusion and failed to change the response to the sodium-retaining action of 9 alpha-fluorohydrocortisone, presumably accounting for her edema.
Authors:
O Matsuda; T Shiigai; Y Ito; K Aonuma; J Takeuchi
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Nephron     Volume:  30     ISSN:  0028-2766     ISO Abbreviation:  Nephron     Publication Date:  1982  
Date Detail:
Created Date:  1982-09-10     Completed Date:  1982-09-10     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  0331777     Medline TA:  Nephron     Country:  SWITZERLAND    
Other Details:
Languages:  eng     Pagination:  178-86     Citation Subset:  IM    
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MeSH Terms
Descriptor/Qualifier:
Adult
Aged
Aminohippuric Acids / metabolism*
Edema / etiology
Female
Humans
Kidney Function Tests
Male
Pedigree
Probenecid / diagnostic use
Pyrazinamide / diagnostic use
Renal Tubular Transport, Inborn Errors / genetics*,  metabolism
Saline Solution, Hypertonic
Ticrynafen / diagnostic use
Uric Acid / blood*
p-Aminohippuric Acid / metabolism*
Chemical
Reg. No./Substance:
0/Aminohippuric Acids; 0/Saline Solution, Hypertonic; 40180-04-9/Ticrynafen; 57-66-9/Probenecid; 61-78-9/p-Aminohippuric Acid; 69-93-2/Uric Acid; 98-96-4/Pyrazinamide

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


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