Abstract |
Intravenous sodium thiosulfate (ivSTS) is a promising new therapeutic option for calciphylaxis related to end-stage renal disease. However, its effect on tumoral calcinosis (TC) complicating autoimmune connective-tissue diseases has been scarcely described. We report here 4 cases (3 adults and 1 child) of TC treated with ivSTS. TC was secondary to CREST syndrome, dermatomyositis (1 adult and 1 child) and systemic erythematous lupus and involved multiple sites in all cases. In all 4 patients, TC was responsible for joint pain, reduced mobility, inflammatory flares and skin fistulations. One patient experienced difficulty sitting due to the pain induced by calcified lesions on the buttock; another patient had major disability, moved only with wheelchair and was under opioid treatment for pain. For all patients, treatment with several medications before STS was unsuccessful. The 3 adults received at least 6 cycles of ivSTS (20g/d, 5 days/month) and the child received a daily infusion of 17g STS during 1 month then a 9-g/d infusion during 3 months. Two adults and the child showed clinical improvement with STS treatment and the third adult felt disappointed and stopped STS treatment after 6 months. The child also stopped STS after 6 months due to vomiting. In one patient, an intensive regimen of ivSTS (20g every 2 days) controlled recurrent flares and fistulations. Unfortunately, TC remained unchanged. Further studies are needed to decipher how STS modulates ectopic calcification, the optimal regimen and posology.
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Authors | Arthur Mageau, Vincent Guigonis, Voa Ratzimbasafy, Thomas Bardin, Pascal Richette, Pablo Urena, Hang-Korng Ea |
Journal | Joint bone spine
(Joint Bone Spine)
Vol. 84
Issue 3
Pg. 341-344
(May 2017)
ISSN: 1778-7254 [Electronic] France |
PMID | 27955821
(Publication Type: Case Reports, Journal Article)
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Copyright | Copyright © 2016 Société française de rhumatologie. Published by Elsevier SAS. All rights reserved. |
Chemical References |
- Chelating Agents
- Thiosulfates
- sodium thiosulfate
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Topics |
- Administration, Intravenous
- Calcinosis
(diagnostic imaging, drug therapy, etiology)
- Chelating Agents
(administration & dosage)
- Humans
- Thiosulfates
(administration & dosage)
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