Hypokalaemic periodic paralysis revealing primary Sjogren's syndrome

Berrhoute, L.; Kissani, Najib; Essaadouni, L.; Zahlane, M.; Louhab, Nissrine

doi:10.1016/j.lpm.2019.09.040

Cited by 2 publications

(2 citation statements)

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“…Two additional case reports with this association were published in 2020, and another study published recently showed a prevalence of 14.81% of DRTA (16 patients) in a cohort of 108 patients with SLE [ 6 , 7 , 8 ]. Generally, the majority of cases with complete DRTA published in literature are diagnosed due to severe symptoms associated with hypokalemia, such as quadriparesis or even respiratory arrest, and therefore, the real prevalence of DRTA associated with autoimmune diseases is difficult to estimate [ 6 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 ]. Therefore, urinary acidification tests are essential in diagnosing those patients who did not develop any symptoms and would otherwise escape undiagnosed.…”

Section: Epidemiology Clinical Presentation and Diagnosismentioning

confidence: 99%

“…Aiming for a better understanding of the disease, we performed a search of the PubMed database using MeSH descriptors (Acidosis, Renal Tubular; Lupus Erythematosus, Systemic; Sjogren’s Syndrome, Systemic Vasculitis, Rheumatoid Vasculitis, Arthritis, Rheumatoid, Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis, Churg-Strauss Syndrome, IgA Vasculitis, Spondylitis, Ankylosing, Cryoglobulins, Hepatitis, Autoimmune, Liver Cirrhosis, Biliary), and we identified 37 individual case reports published since December 2016 with the association of distal renal tubular acidosis and the following autoimmune disorders: SLE, Sjögren’s syndrome, autoimmune hepatitis, primary biliary cirrhosis and rheumatoid arthritis (see Table 1 ) [ 6 , 10 , 11 , 13 , 15 , 16 , 17 , 18 , 20 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 ]. We also identified a case of hereditary autoimmune polyendocrine syndrome (DRTA, hypoparathyroidism, Addison disease), published in a series of cases, which was not included in our analysis, due to the lack of individual data availability [ 39 ].…”

Section: Epidemiology Clinical Presentation and Diagnosismentioning

confidence: 99%

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Distal Renal Tubular Acidosis in Patients with Autoimmune Diseases—An Update on Pathogenesis, Clinical Presentation and Therapeutic Strategies

Ungureanu

Ismail

2022

Biomedicines

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Distal renal tubular acidosis (DRTA) has been reported in association with autoimmune diseases, such as Sjögren’s syndrome, systemic lupus erythematosus (SLE), autoimmune hepatitis, primary biliary cirrhosis, rheumatoid arthritis and autoimmune thyroiditis. Whether we talk about the complete or incomplete form of DRTA associated with autoimmune diseases, the real incidence is unknown because asymptomatic patients usually are not identified, and most of the reported cases are diagnosed due to severe symptoms secondary to hypokalemia, a frequent finding in these cases. The mechanisms involved in DRTA in patients with autoimmune diseases are far from being fully elucidated and most of the data has come from patients with Sjögren’s syndrome. This review will present different hypotheses raised to explain this association. Also, aiming for a better understanding of the association between autoimmune diseases and DRTA, our review summarizes data from 37 case reports published in the last five years. We will emphasize data regarding clinical presentation, biological alterations, treatment and outcome. A very important question is whether immunosuppressive therapy is helpful in DRTA associated with autoimmune diseases. Because the pathology is rather rare, treatment is not standardized, and reported results are often contradictory. Corticosteroids are frequently used, but multiple other immunosuppressive drugs have been proposed and will be approached in this review.

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