An atypical case of primary renal tubular hypokalaemic metabolic alkalosiswith chronic tophaceous gout

Citation
Y. Moriwaki et al., An atypical case of primary renal tubular hypokalaemic metabolic alkalosiswith chronic tophaceous gout, CLIN RHEUMA, 20(5), 2001, pp. 372-375
Citations number
23
Language
INGLESE
art.tipo
Article
Categorie Soggetti
Rheumatology
Journal title
CLINICAL RHEUMATOLOGY
ISSN journal
0770-3198 → ACNP
Volume
20
Issue
5
Year of publication
2001
Pages
372 - 375
Database
ISI
SICI code
0770-3198(2001)20:5<372:AACOPR>2.0.ZU;2-G
Abstract
A 55-year-old woman was referred to our ward for further evaluation of mark ed hyperuricaemia and suspected tophi. On physical examination, huge subcut aneous nodules were observed on the knee joints as well as a small nodule o n the lateral side of the left sole. Blood chemistry showed marked hyperuri caemia (0.85 mmol/l), hypokalaemia (2.7 mmol/l) and a mild degree of renal insufficiency. Arterial blood gas analysis showed signs of metabolic alkalo sis. Daily urinary uric acid excretion on a purine non-restricted diet was 8.9 mmol/day. Uric acid clearance and fractional uric acid clearance were 0 .8 ml/min and 2.6%, respectively. Plasma renin activity was 21.8 ng/ml/h, a nd plasma angiotensin II and aldosterone concentrations were 61 and 121 pg/ ml, respectively. However, pressor response to an intravenous administratio n of angiotensin II was normal. The urinary calcium to creatinine molar rat io was 0.069, and serum magnesium concentration was normal to supranormal. A biopsy of the subcutaneous nodule showed a typical appearance of tophus. Based on these findings, the patient was diagnosed with an atypical case of renal tubular hypokalaemic metabolic alkalosis, with marked hyperuricaemia and tophi as the initial manifestations. So far, only four cases of Bartte r's syndrome with gout and/or hyperuricaemia have been described in Japan. This rare case is presented and its mechanism of hyperuricaemia discussed.