Calcific uremic arteriolopathy, commonly referred to as “calciphylaxis,” is a rare life-threatening condition observed in patients with chronic kidney disease and end-stage renal disease on dialysis. This results in necrotic, ischemic, tender dermal lesions anywhere in the body, but mainly on the abdominal wall and lower extremities, where subcutaneous tissue is abundant. Histologically, it is defined by calcification in dermal capillaries, arterioles, and subcutaneous adipose tissues. It can occur in all advanced stages of chronic kidney disease as well as end-stage renal disease patients on hemodialysis or peritoneal dialysis. Our case highlights a successful case of calciphylaxis in a young female patient who underwent parathyroidectomy and intensification of peritoneal dialysis regimen along with the infusion of sodium thiosulphate injection resulting in complete resolution of the lesion in 3 months. With limited evidence of treatment options and increased frequency of this condition in a dialysis patient, our case highlights the key aspects of calciphylaxis management in a young end-stage renal disease patient who didn’t need a change of dialysis modality. We also review the risk factors and current practiced management options of this condition in our article.