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      Calciphylaxis in a nondialysis patient treated with sodium thiosulfate and high dose of oxygen

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          Background: Calciphylaxis in a nondialysis patient is a rare condition and is characterized by calcific deposition in tissue. We present a case of calciphylaxis in a nondialysis patient who was diagnosed by clinical presentation and skin biopsy and was treated with sodium thiosulfate with improvement of skin lesions. Case: A 43-year-old female with type 2 diabetes and atrial fibrillation taking oral anticoagulation medication presented with reddish drainage from the right buttock. On physical examination, a large perirectal abscess overlying necrosis was found. She also developed acute kidney injury with creatinine of 3.7 mg/dL at peak from 0.8 mg/dL at baseline. She received antibiotics intravenously and wound debridement. During hospitalization, she developed areas of numerous painful erythematous lesions with central dusky necrosis on bilateral lower extremities. Punch biopsy was done, which initially revealed small-vessel vasculitis. However, those lesions did not respond to steroid therapy. A second biopsy was done showing extensive fat necrosis and medial calcification of vessel walls consistent with calciphylaxis. She was treated with high-flow oxygen and sodium thiosulfate intralesionally and intravenously for 6 months. The lesions remarkably reduced in size and were less painful on follow-up. Conclusion: High-dose oxygen and sodium thiosulfate could potentially be effective treatments for calciphylaxis in nondialysis patients.

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          Most cited references 6

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          Calciphylaxis: risk factors, diagnosis, and treatment.

          Calciphylaxis is a rare but devastating condition that has continued to challenge the medical community since its early descriptions in the scientific literature many decades ago. It is predominantly seen in patients with chronic kidney failure treated with dialysis (uremic calciphylaxis) but is also described in patients with earlier stages of chronic kidney disease and with normal kidney function. In this review, we discuss the available medical literature regarding risk factors, diagnosis, and treatment of both uremic and nonuremic calciphylaxis. High-quality evidence for the evaluation and management of calciphylaxis is lacking at this time due to its rare incidence and poorly understood pathogenesis and the relative paucity of collaborative research efforts. We hereby provide a summary of recommendations developed by a multidisciplinary team for patients with calciphylaxis.
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            Calciphylaxis in patients on hemodialysis: a prevalence study.

            Calciphylaxis is characterized by painful, violaceous, mottled skin lesions (livedo reticularis) that may progress to tissue necrosis, nonhealing ulcers, gangrene, and potentially amputation, sepsis, or death. The prevalence and characteristics of patients who have calciphylaxis need further identification to predict which patients on dialysis may benefit from close monitoring or early surgical intervention. All 242 patients undergoing hemodialysis in an outpatient unit were reviewed retrospectively during a 15-month cross-sectional study of the prevalence and characteristics of calciphylaxis. Ten patients (prevalence, 4.1%) had calciphylaxis. Patients with calciphylaxis were significantly younger (49 versus 60 years; p = 0.01), had undergone hemodialysis longer (80 versus 20 months; p < 0.0001), and had higher median serum calcium (9.7 versus 9.2 mg/dl; p = 0.03), phosphate (8.2 versus 5.7 mg/dl; p = 0.001), calcium phosphate product (81.5 versus 52.9; p = 0.0004), parathyroid hormone (1496 versus 138 pg/ml; p < 0.0001), and alkaline phosphatase levels (188 versus 89 IU/L; p = 0.0001). Bone surveys were positive in all 10 patients with calciphylaxis compared with 49 (21%) of the 232 patients without calciphylaxis (p < 0.0001). All patients who underwent parathyroidectomy for calciphylaxis had dramatic healing of the ulcers. The presence of calciphylaxis is higher among younger patients who had undergone longer periods of hemodialysis. Therefore this group of patients should be monitored aggressively and treated expeditiously for complications of secondary hyperparathyroidism.
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              Cutaneous calciphylaxis: a retrospective histopathologic evaluation.

              Calciphylaxis is a rare and life-threatening disease characterized by cutaneous necrosis and vascular calcification. Often, skin biopsy specimens are not diagnostic because of the limited depth of the specimen, biopsy site, and clinical stage. To better understand the utility of various histologic features in rendering the diagnosis of calciphylaxis and to compare von Kossa versus Alizarin red stains in the detection of calcium deposits, we retrospectively analyzed the histologic features and histochemical stain findings of 56 skin biopsies from 27 consecutive patients seen at Massachusetts General Hospital from October 2002 to April 2012, with confirmed diagnosis of calciphylaxis and compared with that of 19 skin biopsies from 17 patients with other disease processes. All forms of vascular calcification and vascular thrombosis were significantly associated with cutaneous calciphylaxis. Perieccrine calcium deposition, highly specific to calciphylaxis, was the only form of calcium deposition noted in 4 (7%) skin biopsies from patients with calciphylaxis. Although the staining appears to be comparable, the deposits seen on Alizarin red appeared larger and were birefringent. Although subtle, perieccrine calcification may aid in the diagnosis of calciphylaxis in settings where typical vascular and extravascular calcification are not identified. Performing both von Kossa and Alizarin red stains might increase the detection of calcium deposit.

                Author and article information

                Clin Nephrol Case Stud
                Clinical Nephrology. Case Studies
                Dustri-Verlag Dr. Karl Feistle
                6 July 2017
                : 5
                : 38-41
                [1 ]Department of Internal Medicine, Bassett Medical Center and Columbia University College of Physicians and Surgeons, Cooperstown, NY, USA, and
                [2 ]Department of Preventive and Social Medicine, Faculty of Medicine Siriraj Hospital, Mahidol University, Thailand
                Author notes
                Sikarin Upala, MD Department of Internal Medicine, Bassett Medical Center, 1 Atwell Road, Cooperstown, NY 13326, USA sikarin.upala@
                © Dustri-Verlag Dr. K. Feistle

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                Case Report

                sodium thiosulfate , nondialysis, calciphylaxis


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