Complete resolution of recurrent calciphylaxis with long‐term intravenous sodium thiosulfate

Abstract

A 35-year-old morbidly obese woman on home haemodialysis presented with painful indurated subcutaneous nodules histologically characteristic of calciphylaxis. After failing to respond to conventional treatment, she was commenced on an intravenous infusion of 25 g of sodium thiosulfate three times per week. Two weeks after commencing sodium thiosulfate, the pain resolved completely. By 12 weeks, the lesions had healed and the infusions were ceased. Two months later, skin lesions recurred, but resolved again within 3 months of recommencement of sodium thiosulfate treatment, which was continued for 8 months. The treatment was well tolerated. There has been no recurrence of lesions in the 18 months since the cessation of sodium thiosulfate. Clinical trials to determine the optimum dose and duration of therapy for sodium thiosulfate treatment of calciphylaxis should be given priority because of its high rate of success in treating what is otherwise a severe and mostly lethal condition.

Sodium Thiosulfate: Calciphylaxis

“…Conventional therapy consists of intensifying treatment with a low calcium dialysate for those receiving hemodialysis, noncalcium phosphate binders, cinacalcet, selective vitamin D analogues, analgesics, or aggressive wound care techniques. [1][2][3] Sodium thiosulfate traditionally has been used as an antidote in cyanide poisoning and as a nephroprotectant during cisplatin administration. It is thought to have both cation-chelating and antioxidant properties.…”

“…Based on the lack of recurrence with extended treatment, the authors suggested that the optimal duration of sodium thiosulfate therapy is several months. 2 In another case report, a woman 45 years of age with chronic allograft nephropathy following a renal transplant 10 years earlier developed resistant, painful ulcers on her left leg. Symptoms progressed to septic shock despite treatment with analgesics and antibiotics, necessitating amputation of the left leg.…”

Calciphylaxis: A systematic review of existing and emerging therapies

“…21 The most important potential adverse effect associated with STS is anion gap metabolic acidosis due to the resulting presence of thiosulfuric acid. 42 This gap acidosis may result in deleterious effects on protein and bone metabolism, the requirement of oral sodium bicarbonate to correct the acidosis (possibly presenting an unacceptable sodium load), or development of hypocalcemia, which may result in a prolonged QT interval. 42,61 Despite marked favorable attention recently, no prospective, randomized controlled trials of STS in calciphylaxis have yet been completed.…”

“…42 This gap acidosis may result in deleterious effects on protein and bone metabolism, the requirement of oral sodium bicarbonate to correct the acidosis (possibly presenting an unacceptable sodium load), or development of hypocalcemia, which may result in a prolonged QT interval. 42,61 Despite marked favorable attention recently, no prospective, randomized controlled trials of STS in calciphylaxis have yet been completed. In addition, many reported cases involve the use of multiple therapies along with STS; in this context, it is difficult to attribute successful therapy to STS alone.…”

A complex case of fatal calciphylaxis in a female patient with hyperparathyroidism secondary to end stage renal disease of graft and coexistence of haemolytic uremic syndrome

“…It is reported that intravenous administration of sodium thiosulfate 6,7 before surgical parathyroidectomy increases the solubility of calcium deposits, reduces disseminated precipitation of calcium crystals and counteracts occlusions in small and medium calibre arteries, improving clinical picture acting as antioxidant and calcium binding drug.…”