Calciphylaxis is a serious complication in chronic kidney disease (CKD) patients and requires a multidisciplinary approach. Dialysis patients are the most affected, especially those who are obese and diabetic, have liver diseases and autoimmune diseases, or are on systemic glucocorticoids. Parathyroidectomy (PTx) is curative for secondary hyperparathyroidism (SHPT) and calciphylaxis in most cases. Calciphylaxis after parathyroidectomy is a rare presentation. We present a case of a young patient who developed calciphylaxis after parathyroidectomy, an uncommon presentation.