Successful treatment of calciphylaxis in a renal transplant recipient with combination of intralesional sodium thiosulphate, intravenous sodium thiosulphate and fish skin graft.

Abstract

Calciphylaxis is a rare small vessel vasculopathy with high mortality rate of 80%. Sepsis is the leading cause of the mortality. To date, there is still lack of consensus regarding the optimal approach to the treatment of calciphylaxis. We present a 42-year-old lady who developed calciphylaxis 12 years after kidney transplant with preserved graft function and normal mineral levels. Two months before her presentation, she was started on warfarin for upper limb deep vein thrombosis. Her warfarin was stopped. Intralesional (IL) and intravenous (IV) sodium thiosulphate (STS) were started concurrently. She initially showed improvement; however, the recovery was further complicated by wound infection (Figure 1). Culture-directed antibiotics was started, followed by surgical wound debridement. The infection was under controlled, and ulcer progression was arrested. Fish skin graft was transplanted with good uptake (Figure 2). She was able to ambulate again with walking aid at 90 days after surgery. Kidney transplant is a potential treatment for calciphylaxis, but there are reported cases of new-onset calciphylaxis after kidney transplant. This highlights the importance of high clinical suspicion for calciphylaxis in a kidney transplant recipient with preserved graft function. We summarised the case reports of calciphylaxis in kidney transplant recipients with preserved graft function (Table 1). Calciphylaxis that developed within 12 months after kidney transplant was associated with disrupted mineral balance. We identified 6 case reports of calciphylaxis which developed more than 12 months after the kidney transplant and found strong association between warfarin use and calciphylaxis development. Therefore, warfarin should be used judiciously in patients with ESRD. Timely intervention is crucial in the management of calciphylaxis. In our case, the concurrent administration of intravenous and intralesional STS, aggressive wound debridement, and the application of fish skin graft had yielded a good clinical outcome for calciphylaxis. STS enhances the solubility of calcium deposits and restores endothelial cell dysfunction through its antioxidant effect. Numerous studies have supported the use of either intravenous (IV) or intralesional (IL) STS in the treatment of calciphylaxis, but concurrent use of IV and IL STS is uncommon. The distribution of IV STS to the active ulcer is potentially affected by the calcified vessels. IL STS is a highly targeted delivery method to the ulcer bypassing the systemic distribution. We hypothesised IL STS could lead to rapid resolution of the calcium deposits and restoration of the endothelial function. This could potentially increase the subsequent absorption of the IV STS to the affected areas. Surgical debridement with split-skin transplantation has been shown to prevent deep infection and facilitate the ulcer healing. Fish skin graft contains high-concentration omega-3 polyunsaturated fatty acids, eicosapentaenoic acid and docosahexaenoic acid. These lipids are proven to reduce inflammation and promote wound healing in chronic ulcer. Although fish skin graft has been used in chronic wound such as diabetic foot ulcer and yielded good clinical outcome, this is the first case in the literature that described the use of fish skin graft in calciphylaxis wound.