Penile calciphylaxis in a patient on combined peritoneal dialysis and hemodialysis

Abstract

Calciphylaxis presents with painful purpura and intractable skin ulcers on the trunk and particularly the distal extremities, and it mainly occurs in patients on chronic dialysis. A 66-year-old man with renal failure due to diabetic nephropathy was on peritoneal dialysis alone for 1 year, followed by peritoneal dialysis combined with hemodialysis for 3 years. He developed calciphylaxis of the penis, which was diagnosed from the skin biopsy findings and clinical observation. To treat this condition, PD was stopped and HD was performed three times a week. In addition, warfarin therapy was discontinued and infusion of sodium thiosulfate was performed. The penile ulcers decreased in size and pain was markedly improved, so the patient was discharged from hospital. Following discharge, PD was resumed after changing the peritoneal dialysis fluid to bicarbonate-buffered dialysate. The penile ulcers eventually resolved completely. There have been very few reports about calciphylaxis in patients on combined dialysis modalities. In our patient, penile calciphylaxis progressed when lactate-buffered peritoneal dialysis fluid was used and resolved after switching to bicarbonate-buffered fluid together with cessation of warfarin therapy and infusion of sodium thiosulfate.

Efficacy and Safety of Sodium Thiosulfate in Japanese Patients with Calciphylaxis

“…On the other hand, relatively few case reports have described the effect of STS for calciphylaxis in Japanese patients [8][9][10][11][12][13] . STS was effective in all of the previously reported Japanese patients who were undergoing dialysis.…”

“…STS was effective in all of the previously reported Japanese patients who were undergoing dialysis. In two case reports, STS (10 g/ day) was administered 3 times/week during dialysis 10,11) . Conversely, Tokashiki et al and Ishikawa et al administered 20 and 5-25 g/day, respectively 9,13) .…”

“…To avoid side effects, we initiated intravenous STS at a low daily dosage (10 g/day) and gradually increased it. Case 1 (non-dialysis patient) was started with a daily dose of 10 g STS, considering the patient's older age and previous reports 10) . In cases 2 and 3, intravenous STS was started at a dose of 10 g after every dialysis and was gradually increased.…”

A rapid unfavorable penile calciphylaxis case followed by total penectomy

“…To date, less than 80 cases of penile calciphylaxis have been described in the literature but the prevalence of the pathology is probably underestimated. Penile calciphylaxis is an exclusion diagnosis that is based on the combination of clinical, biological, and histological elements 7 . Clinically, the patient presents with ulcered and painful lesions that are resistant to treatment and appear in the context of end‐stage kidney disease and often dialysis.…”

“…Histologically, deep biopsy can provide information that is useful for diagnosis. The von Kossa staining highlights calcium deposits, but their presence is not specific and could depend on the biopsy site and medical history of the patient 7 . However, a recent comparative study demonstrated that the presence of more or fewer interstitial vascular calcifications in hemodialyzed patients has good sensitivity and specificity for calciphylaxis and does not seem to be related to dialysis 8 .…”