Abstract Penile gangrene is a rare disease. It represents a poor prognostic sign in end stage renal disease (ESRD) patients and an indicator of metastatic vascular calcification. Pathogenesis of this life threatening condition is not clearly understood and treatment is also controversial. We describe the case of a 61-year-old man known to have diabetes mellitus, chronic renal failure on haemodialysis, who was complaining of worsening groin pain for 4 weeks. On examination dry gangrene of glans penis was noted. Cultures from the penis showed multiple organisms. Computed tomography (CT) showed diffuse calcification of external and internal iliac arteries. Later, he developed caciphylaxis of right anterior thigh .His overall condition did not improve in spite of adequate antibiotics and he was shifted to intensive care unit (ICU) where he required high doses of vasopressors. Clinically he kept deteriorating and passed away due to septic shock. Keywords: Calciphylaxis, Penile, Renal disease, Necrosis. Introduction Calciphylaxis is a rare but frequently reported disease in dialysis patients. Penile calciphylaxis is rare and has poor outcome unless treated aggressively. There are few case reports and case series reported from all over the world including developing countries. 1,2 In patients of chronic renal failure (CRF), it is an indicator of extensive vascular calcification due to secondary hyperparathyroidism also known as calcific uraemic arteriolopathy (CAU), which usually affects distal extremities. Penile gangrene secondary to various causes like, priapism, vasculitis, penile prosthesis, fournier's gangrene, and uraemia has been reported. Treatment is complicated and ranges from conservative measures to more aggressive approaches such as penectomy. We are presenting a dialysis depended diabetic patient who was diagnosed with penile gangrene due to possible calciphylaxis and in spite of appropriate medical management, his condition deteriorated and he expired. Case Report A 61-year-old African American male was seen at the Saint Luke's Hospital, in April 2013, with past medical history of diabetes mellitus, ischaemic cardiomyopathy (EF of 20- 25%, status post biventricular defibrillator), chronic atrial fibrillation, peripheral vascular disease, ESRD who had been on maintenance peritoneal dialysis for one year presented with worsening groin pain for one month. He was shifted on haemodialysis one month ago because of fluid overload and scrotal oedema from peritoneal dialysis. He was seen in dialysis unit one day before the admission when he had a temperature of 38.1°C during dialysis session. He was pancultured and given vancomycin and cefepime. He tolerated dialysis well but was somnolent and lethargic after that. On presentation, he was confused, having severe pain in his groin area with temperature of 37.6°C, blood pressure of 93/51mmHg and pulse rate of 84bpm. On examination, groin area was foul smelling, the glans and the skin involving the coronal sulcus was black, hard, and firm, suggestive of dry gangrene. There was no erythema or fluctuance. Initial data showed haemoglobin and haematocrit of 10.2g/dl and 31.3%, white blood cell (WBC) count 11.5 K/ul, platelet count 129 K/ul, sodium 141mmol/L, potassium 4.3mmol/L, chloride 97mmol/L, bicarbonate 26mmol/L, glucose 120mg/dl, BUN 22mg/dl, creatinine 3.6mg/dl, albumin 2.9g/dl, total protein 6.5g/dl, ALT 26 U/L, AST 43 U/L, total bilirubin 2.9g/dl, INR was 1.3. The patient was evaluated by infectious disease specialist and was started on intravenous (I/V) cefazolin and metronidazole along with I/V hydration. Staff urologist and nephrologist were consulted and the initial plan was to watch him closely and consider for partial penectomy but the patient refused any surgical interventions. Although his blood, peritoneal and urine cultures were negative, cultures from the penis showed heavy growth of corynebacterium, staphylococcus aureus and gamma haemolytic streptococci. Later he also developed discolouration in the anterior aspect of his right thigh, which was hard and very tender to touch, consistent with calciphylaxis. Para thyroid hormone (PTH) level was 260pg/ml (normal= 11-54pg/ml). A clinical diagnosis was made. Optimally, a biopsy should J Pak Med Assoc 711 CASE REPORT Calciphylaxis leading to penile necrosis Imran Haider, 1 Muhammad Siddiqui, 2 Wisam Naji, 3 Maria Sheikh, 4 Ayesha Waqar 5 1-4 St. Luke's Hospital, St. Louis, MO, USA, 5 Great River Hospital, Burlington, Iowa, USA. Correspondence: Imran Haider. Email: drimranhaider6@yahoo.com