Regression of vascular calcification following an acute episode of calciphylaxis: a case report

INTRODUCTION In clinical situations, vascular calcification tends to progress and is difficult to completely arrest or reverse. Calciphylaxis, a severe complication of end-stage renal disease, is a specific form of vascular calcification. Control studies have provided evidence that monotherapy with sodium thiosulfate or cinacalcet delays the progression of vascular calcification. Successful treatment of calciphylaxis with sodium thiosulfate or cinacalcet has also been reported. We report a case demonstrating the regression of vascular calcification following an acute episode of necrotic skin lesions suspected to be calciphylaxis. During the successful multimodal treatment, sodium thiosulfate and cinacalcet were sequentially administered in addition to surgical debridement and percutaneous transluminal angioplasty. CASE PRESENTATION We describe the case of a 71-year-old Asian woman on hemodialysis who presented with suspected calciphylaxis lesions in her lower left leg. Plain radiographs revealed diffuse calcified vessel changes in her lower extremities. During the initial wound treatment with a course of intravenous sodium thiosulfate, our patient's predialysis serum levels of total calcium markedly increased, yielding no calciphylaxis improvement. The necrotic wounds began healing only after surgical debridement. A percutaneous transluminal angioplasty was performed to dilate a 70% stenosis in her left posterior tibial artery. Our patient was then treated with cinacalcet, resulting in improved control of her calcium, phosphate and parathyroid hormone serum levels. The lesions completely healed after six months of multimodal treatment. Repeated plain radiographs in the following two years revealed gradual vascular calcification regression in her lower extremities. CONCLUSION In addition to the favorable outcome of our patient's wounds, radiology was used to document the regression of calcification in the large and small arteries of her lower limbs. However, it is difficult to determine the precise mechanism of the multimodal treatment that caused the vascular calcification regression and wound healing. The clinical course suggested that the surgical treatment and percutaneous transluminal angioplasty substantially contributed to healing her wounds. Cinacalcet and sodium thiosulfate may have played distinct roles in the regression of her vascular calcification. A well-controlled study or large case series are required to assess the additive effects of these agents when treating vascular calcification.