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An uncommon presentation of Sjögren's syndrome and brucellosis

Authors :
Gülperi Çelik
Fatih Çölkesen
Sema Yilmaz
Levent Kebapcilar
Suleyman Hilmi Ipekci
Suleyman Baldane
Ercument Ozturk
Source :
Transfusion and apheresis science : official journal of the World Apheresis Association : official journal of the European Society for Haemapheresis. 51(1)
Publication Year :
2014

Abstract

We describe herein a case of hypokalemia due to proximal renal tubular acidosis (RTA) and Fanconi's syndrome (FS) and nephrogenic diabetes insipidus with DIC – a rare complication of Sjogren's syndrome (SS) and brucellosis. The interesting feature of this case was the presentation with severe hypokalemia, causing acute flaccid quadriparesis with cardiac arrest which is extremely rare. The patient was a 48-year-old woman who suffered cardiopulmonary arrest an hour after hospitalization. Analysis of a blood sample obtained before her cardiopulmonary arrest yielded surprising results: laboratory investigations showed profound hypokalemia (1.1 mEq/L) with renal K wasting, hyperchloremic metabolic acidosis with normal anion gap, hypophosphatemia with hypouricemia, glucosuria, and proteinuria. A diagnosis of RTA and FS were made. On the seventh day, she looked acutely ill, temperature 38.8 °C and pale, and her physical examination revealed purpuric skin lesions on both legs. The serum antibrucella titration agglutination test was found to be 1 of 160 positive with a nosocomial infection. The clinical and laboratory findings were consistent with disseminated intravascular coagulation (DIC). She was unable to concentrate her urine and so a diagnosis of nephrogenic diabetes insipidus (NDI) was reached. A thorough survey for the cause of FS, RTA and NDI revealed that she had xerophthalmia and xerostomia accompanied by high anti-Ro antibody, positive Schirmer test, confirming the diagnosis of SS.

Details

ISSN :
14730502
Volume :
51
Issue :
1
Database :
OpenAIRE
Journal :
Transfusion and apheresis science : official journal of the World Apheresis Association : official journal of the European Society for Haemapheresis
Accession number :
edsair.doi.dedup.....91c521f474f20b742659e05fa800ec3b