Literature DB >> 20414123

Hypokalemic paralysis as primary presentation of Fanconi syndrome associated with Sjögren syndrome.

Chih-Chiang Wang1, Jeng-Chuan Shiang, Wen-Te Huang, Shih-Hua Lin.   

Abstract

Hypokalemic paralysis is a rare presentation of Fanconi syndrome (FS) caused by Sjögren Syndrome (SS). We describe a 39-year-old man who manifested flaccid paralysis of 4 limbs. Laboratory investigations showed profound hypokalemia (1.6 mmol/L) with renal K wasting, hyperchloremic metabolic acidosis with positive urine anion gap, hypophosphatemia with hyperphosphaturia, hypouricemia with hyperuricosuria, normoglycemic glycosuria, and abnormal serum creatinine concentration 2.2 mg/dL. A thorough survey for the cause of FS revealed that he had xerophthalmia and xerostomia accompanied by high anti-Ro antibody, positive Schirmer test, and delayed saliva excretion on sialoscintigraphy, confirming the diagnosis of SS. Potassium citrate, active vitamin D3, and high phosphate diet for his FS coupled with mycophenolate mofetil for SS resolved clinical symptoms and ameliorated renal function. Early recognition of HP due to the underlying SS-related FS with prompt therapy not only could terminate potentially life-threatening hypokalemia, but also improve renal outcome.

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Year:  2010        PMID: 20414123     DOI: 10.1097/RHU.0b013e3181df903f

Source DB:  PubMed          Journal:  J Clin Rheumatol        ISSN: 1076-1608            Impact factor:   3.517


  6 in total

Review 1.  [Interstitial nephritis in rheumatic diseases].

Authors:  P Korsten; G A Müller
Journal:  Z Rheumatol       Date:  2015-05       Impact factor: 1.372

2.  Recommendations for evaluation and diagnosis of extra-glandular manifestations of primary sjogren syndrome: results of an epidemiologic systematic review/meta-analysis and a consensus guideline from the Brazilian Society of Rheumatology (articular, pulmonary and renal).

Authors:  Virginia Fernandes Moça Trevisani; Alisson Pugliesi; Sandra Gofinet Pasoto; Maria Lucia Lemos Lopes; Lissiane Karine Noronha Guedes; Samira Tatiyama Miyamoto; Marilena Leal Mesquita Silvestre Fernandes; Sonia Cristina de Magalhães Souza Fialho; Aysa César Pinheiro; Laura Caldas Dos Santos; Simone Appenzeller; Tania Fidelix; Sandra Lúcia Euzébio Ribeiro; Danielle Christinne Soares Egypto de Brito; Tatiana Nayara Libório-Kimura; Maria Carmen Lopes Ferreira Silva Santos; Diego Ustárroz Cantali; Juliana D'Agostino Gennari; Vinicius Tassoni Civile; Ana Carolina Pereira Nunes Pinto; César Ramos Rocha-Filho; Fabiola Reis Oliveira; Aline Pereira da Rocha; Valeria Valim
Journal:  Adv Rheumatol       Date:  2022-06-01

Review 3.  Sjögren's syndrome complicated with Fanconi syndrome and Hashimoto's thyroiditis: Case report and literature review.

Authors:  Mingmin Shi; Lei Chen
Journal:  J Int Med Res       Date:  2016-03-09       Impact factor: 1.671

4.  Ectopic germinal center and megalin defect in primary Sjogren syndrome with renal Fanconi syndrome.

Authors:  Jing Wang; Yubing Wen; Mengyu Zhou; Xiaoxiao Shi; Lanping Jiang; Mingxi Li; Yang Yu; Xuemei Li; Xuewang Li; Wen Zhang; Andrew L Lundquist; Limeng Chen
Journal:  Arthritis Res Ther       Date:  2017-06-02       Impact factor: 5.156

5.  Hypokalemic paralysis as a presenting manifestation of primary Sjögren's syndrome: A report of two cases.

Authors:  Deepak Khandelwal; Saptarshi Bhattacharya; Rajesh Khadgawat; Satbir Kaur; Nikhil Tandon; Ariachery C Ammini
Journal:  Indian J Endocrinol Metab       Date:  2012-09

6.  Sjögren's Syndrome Associated with Fanconi's Syndrome and Osteomalacia.

Authors:  Jorge O Gutierrez; Maria F Zurita; Luis A Zurita
Journal:  Am J Case Rep       Date:  2018-04-03
  6 in total

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