A Case of Familial Renal Hypouricemia Associated with Increased Secretion of Para-Aminohippurate and Idiopathic Edema

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Abstract

A 43-year-old housewife had hypouricemia (serum uric acid ranging from 0.5 to 1.5 mg/dl; 30–89 µmol/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 9α-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 9oc-fluorohydrocortisone, presumably accounting for her edema.

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Author and article information

Journal

Journal ID (publisher-id): NEF

Journal ID (nlm-ta): Nephron

Journal ID (doi): 10.1159/issn.1660-8151

Title: Nephron

Publisher: S. Karger AG

ISSN (Print): 1660-8151

ISSN (Electronic): 2235-3186

Publication date Subscription-year: 1982

Publication date (Print): 1982

Publication date (Electronic): 03 December 2008

Volume: 30

Issue: 2

Pages: 178-186

Affiliations

^aSecond Department of Internal Medicine, Tokyo Medical and Dental University, Yushima, and ^bDepartment of Internal Medicine, Nakano General Hospital, Nakano, Tokyo, Japan

Article

Publisher ID: 182457 Other ID: Nephron 1982;30:178–186

DOI: 10.1159/000182457

PubMed ID: 7099326

SO-VID: c6ffed56-bc45-4984-a683-e363adc86a7b

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Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

History

Date accepted : 12 March 1981

Page count

Pages: 9

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