Stéphanie Malbos1, Pablo Ureña-Torres2, Thomas Bardin3, Hang-Korng Ea4. 1. Service de rhumatologie, pôle appareil locomoteur, centre Viggo-Petersen, hôpital Lariboisière, AP-HP, 2, rue Ambroise-Paré, 75010 Paris, France. 2. Clinique du Landy, 93400 Saint-Ouen, France; Service des explorations fonctionnelles rénales, hôpital Necker, 75015 Paris, France. 3. Service de rhumatologie, pôle appareil locomoteur, centre Viggo-Petersen, hôpital Lariboisière, AP-HP, 2, rue Ambroise-Paré, 75010 Paris, France; UFR de médecine, université Paris Diderot, Sorbonne Paris Cité, 75205 Paris, France. 4. Service de rhumatologie, pôle appareil locomoteur, centre Viggo-Petersen, hôpital Lariboisière, AP-HP, 2, rue Ambroise-Paré, 75010 Paris, France; UFR de médecine, université Paris Diderot, Sorbonne Paris Cité, 75205 Paris, France; Inserm, UMR-1132, hôpital Lariboisière, 75010 Paris, France. Electronic address: hang-korng.ea@lrb.aphp.fr.
Abstract
BACKGROUND: Calcific uremic arteriolopathy (CUA) or calciphylaxis is a severe complication of advanced chronic kidney disease (CKD) and dialysis. Few effective treatments are available and the mortality rate is high. We report 4 cases in which sodium thiosulfate therapy was rapidly effective. CASES: Sodium thiosulfate therapy was given to 4 Caucasian patients (3 females and 1 male aged 49 to 91 years) with CUA. The causes of end-stage CKD were nephroangiosclerosis (n=2) and diabetic nephropathy (n=2). The lesions developed 1 to 6.5 years after the initiation of hemodialysis and involved the lower limbs in 2 patients, the fingers in 1 patient, and a breast in the remaining patient. They were responsible for pain and skin necrosis in all 4 patients. Local superinfection occurred in 3 patients. Intravenous sodium thiosulfate was given in a dosage of 12.5 to 25g after each hemodialysis session, for 12 to 24 weeks. The pain and trophic disorders resolved fully in all 4 patients. The side effects consisted of nausea and vomiting (n=2) and a moderate blood pressure decrease (n=1). No recurrences were noted during the follow-up of 5 to 17 months after treatment discontinuation. CONCLUSION: The findings from this small case-series suggest that sodium thiosulfate may hold promise for the treatment of CUA.
BACKGROUND:Calcific uremic arteriolopathy (CUA) or calciphylaxis is a severe complication of advanced chronic kidney disease (CKD) and dialysis. Few effective treatments are available and the mortality rate is high. We report 4 cases in which sodium thiosulfate therapy was rapidly effective. CASES: Sodium thiosulfate therapy was given to 4 Caucasian patients (3 females and 1 male aged 49 to 91 years) with CUA. The causes of end-stage CKD were nephroangiosclerosis (n=2) and diabetic nephropathy (n=2). The lesions developed 1 to 6.5 years after the initiation of hemodialysis and involved the lower limbs in 2 patients, the fingers in 1 patient, and a breast in the remaining patient. They were responsible for pain and skin necrosis in all 4 patients. Local superinfection occurred in 3 patients. Intravenous sodium thiosulfate was given in a dosage of 12.5 to 25g after each hemodialysis session, for 12 to 24 weeks. The pain and trophic disorders resolved fully in all 4 patients. The side effects consisted of nausea and vomiting (n=2) and a moderate blood pressure decrease (n=1). No recurrences were noted during the follow-up of 5 to 17 months after treatment discontinuation. CONCLUSION: The findings from this small case-series suggest that sodium thiosulfate may hold promise for the treatment of CUA.
Authors: Marie-Sophie L Y de Koning; Solmaz Assa; Carlijn G Maagdenberg; Dirk J van Veldhuisen; Andreas Pasch; Harry van Goor; Erik Lipsic; Pim van der Harst Journal: J Interv Cardiol Date: 2020-09-24 Impact factor: 2.279