Development of Proximal Calciphylaxis with Penile Involvement after Parathyroidectomy in a Patient on Hemodialysis

Abstract

Elevated parathyroid hormone (PTH) levels and hyperphosphatemia are thought to be associated with the development of calciphylaxis. We report a patient on hemodialysis who developed proximal calciphylaxis with consistently low PTH levels after parathyroidectomy. A 31-year-old man was admitted to our hospital because of abdominal skin ulcerations. Calciphylaxis spread to the penis, and simultaneous progressive lung calcification was evident on chest X-ray, suggestive of pulmonary calciphylaxis on 99m Tc-methylene disphosphonate scintigraphy. The patient died of respiratory failure despite intensive treatment including hyperbaric oxygen therapy. This is the first report of a patient on hemodialysis who developed calciphylaxis involving the penis after parathyroidectomy. (Internal Medicine 43: 63-68, 2004)

Calciphylaxis: A Report of Six Cases and Review of Literature

“…42 A patient on HD who developed calciphylaxis after parathyroidectomy has been reported as well. 26 These findings suggest other possible unidentified factors in the pathogenesis of calciphylaxis. Both hyperparathyroidism and adynamic bone disease in patients with relative or absolute hypoparathyroidism may cause elemental derangement in mineral metabolism predisposing for development of calciphylaxis.…”

“…However, skeletal and heart muscle, joint, lung, eye, penis, breast, pancreas, and intestinal tissue involvements have also been reported. [22][23][24][25][26][27][28][29][30][31][32] Lesions begin characteristically as a livedo reticularis pattern of mottling with violaceous, superficial, tender nodules with palpable subcutaneous deposits of Ca or thickened blood vessels. 5,12,33,34 Lesions progress to ulceration and become hemorrhagic with dry necrosis.…”

“…[63][64][65] Steroid treatment and administration of prostaglandin E1 and vasodilators demonstrated no beneficial effect. 26 Improvement of calciphylaxis after intravenous pamidronate therapy has been recently reported as a new alternative therapy. 12,66 Adynamic bone disease should be excluded before starting bisphosphonate therapy.…”

Calciphylaxis: A systematic review of existing and emerging therapies

“…59,97,98 To date, 46 cases of calciphylaxis treated with HBO have been published, with 34 noting success. 15,[99][100][101][102][103][104][105][106][107][108] These patients were treated by 10 different groups of authors, with the largest case series including 12 patients. Notably, adjunctive therapies for calciphylaxis were also used in every patient receiving HBO.…”

Ever-Changing Concepts of Calciphylaxis

“…In this issue of Internal Medicine, Oikawa and associates report a case of penile calciphylaxis (8).…”