کد مقاله | کد نشریه | سال انتشار | مقاله انگلیسی | نسخه تمام متن |
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5667755 | 1407867 | 2017 | 4 صفحه PDF | دانلود رایگان |
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 4Â cases (3Â adults and 1Â child) of TC treated with ivSTS. TC was secondary to CREST syndrome, dermatomyositis (1Â adult and 1Â child) and systemic erythematous lupus and involved multiple sites in all cases. In all 4Â 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 3Â adults received at least 6Â cycles of ivSTS (20Â g/d, 5Â days/month) and the child received a daily infusion of 17Â g STS during 1Â month then a 9-g/d infusion during 3Â months. Two adults and the child showed clinical improvement with STS treatment and the third adult felt disappointed and stopped STS treatment after 6Â months. The child also stopped STS after 6Â months due to vomiting. In one patient, an intensive regimen of ivSTS (20Â g every 2Â 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.
Journal: Joint Bone Spine - Volume 84, Issue 3, May 2017, Pages 341-344