Regression of vascular calcification following an acute episode of calciphylaxis: a case report

Journal of Medical Case Reports

Yao-Min Hung¹  Chien-Ming Chen²  Ing-Jer Huang³  Hui-Tsung Yeh³ 

[1] Department of Emergency Medicine, Kaohsiung Veterans General Hospital, 386 Dazhong 1st Road, Zuoying District, Kaohsiung City 81362, Taiwan;Department of Plastic and Reconstructive Surgery, Chiayi Branch, Chang Gung Memorial Hospital, 6 West Section, Jiapu Road, Puzi City, Chiayi County 61363, Taiwan;Hong Yi Clinic of Nephrology, 309 MinZu Road, Chiayi City 600, Taiwan

关键词: Vascular calcification;    Sodium thiosulfate;    Cinacalcet;    Calciphylaxis;   

Others  :  822748
DOI  :  10.1186/1752-1947-8-52

 received in 2013-05-31, accepted in 2013-12-16,  发布年份 2014

【 摘 要 】

Introduction

In clinical situations, vascular calcification tends to progress and is difficult to completely arrest or reverse. Calciphylaxis, a severe complication of end-stage renal disease, is a specific form of vascular calcification. Control studies have provided evidence that monotherapy with sodium thiosulfate or cinacalcet delays the progression of vascular calcification. Successful treatment of calciphylaxis with sodium thiosulfate or cinacalcet has also been reported. We report a case demonstrating the regression of vascular calcification following an acute episode of necrotic skin lesions suspected to be calciphylaxis. During the successful multimodal treatment, sodium thiosulfate and cinacalcet were sequentially administered in addition to surgical debridement and percutaneous transluminal angioplasty.

Case presentation

We describe the case of a 71-year-old Asian woman on hemodialysis who presented with suspected calciphylaxis lesions in her lower left leg. Plain radiographs revealed diffuse calcified vessel changes in her lower extremities. During the initial wound treatment with a course of intravenous sodium thiosulfate, our patient’s predialysis serum levels of total calcium markedly increased, yielding no calciphylaxis improvement. The necrotic wounds began healing only after surgical debridement. A percutaneous transluminal angioplasty was performed to dilate a 70% stenosis in her left posterior tibial artery. Our patient was then treated with cinacalcet, resulting in improved control of her calcium, phosphate and parathyroid hormone serum levels. The lesions completely healed after six months of multimodal treatment. Repeated plain radiographs in the following two years revealed gradual vascular calcification regression in her lower extremities.

Conclusion

In addition to the favorable outcome of our patient’s wounds, radiology was used to document the regression of calcification in the large and small arteries of her lower limbs. However, it is difficult to determine the precise mechanism of the multimodal treatment that caused the vascular calcification regression and wound healing. The clinical course suggested that the surgical treatment and percutaneous transluminal angioplasty substantially contributed to healing her wounds. Cinacalcet and sodium thiosulfate may have played distinct roles in the regression of her vascular calcification. A well-controlled study or large case series are required to assess the additive effects of these agents when treating vascular calcification.

【 授权许可】

   
2014 Yeh et al.; licensee BioMed Central Ltd.

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【 参考文献 】

• [1]Fine A, Zacharias J: Calciphylaxis is usually non-ulcerating: risk factors, outcome and therapy. Kidney Int 2002, 61:2210-2217.
• [2]Mawad HW, Sawaya BP, Sarin R, Malluche HH: Calcific uremic arteriolopathy in association with low turnover uremic bone disease. Clin Nephrol 1999, 52:160-166.
• [3]Shmidt E, Murthy NS, Knudsen JM, Weenig RH, Jacobs MA, Starnes AM, Davis MD: Net-like pattern of calcification on plain soft-tissue radiographs in patients with calciphylaxis. J Am Acad Dermatol 2012, 67:1296-1301.
• [4]Raymond CB, Wazny LD: Sodium thiosulfate, bisphosphonates, and cinacalcet for treatment of calciphylaxis. Am J Health Syst Pharm 2008, 65:1419-1429.
• [5]Bleibel W, Hazar B, Herman R: A case report comparing various radiological tests in the diagnosis of calcific uremic arteriolopathy. Am J Kidney Dis 2006, 48:659-661.
• [6]Bellasi A, Ferramosca E, Muntner P, Ratti C, Wildman RP, Block GA, Raggi P: Correlation of simple imaging tests and coronary artery calcium measured by computed tomography in hemodialysis patients. Kidney Int 2006, 70:1623-1628.
• [7]Sood AR, Wazny LD, Raymond CB, Leung K, Komenda P, Reslerova M, Verrelli M, Rigatto C, Sood MM: Sodium thiosulfate-based treatment in calcific uremic arteriolopathy: a consecutive case series. Clin Nephrol 2011, 75:8-15.
• [8]Mathews SJ, de Las FL, Podaralla P, Cabellon A, Zheng S, Bierhals A, Spence K, Slatopolsky E, Davila-Roman VG, Delmez JA: Effects of sodium thiosulfate on vascular calcification in end-stage renal disease: a pilot study of feasibility, safety and efficacy. Am J Nephrol 2011, 33:131-138.
• [9]Adirekkiat S, Sumethkul V, Ingsathit A, Domrongkitchaiporn S, Phakdeekitcharoen B, Kantachuvesiri S, Kitiyakara C, Klyprayong P, Disthabanchong S: Sodium thiosulfate delays the progression of coronary artery calcification in haemodialysis patients. Nephrol Dial Transplant 2010, 25:1923-1929.
• [10]Mataic D, Bastani B: Intraperitoneal sodium thiosulfate for the treatment of calciphylaxis. Ren Fail 2006, 28:361-363.
• [11]Pasch A, Schaffner T, Huynh-Do U, Frey BM, Frey FJ, Farese S: Sodium thiosulfate prevents vascular calcifications in uremic rats. Kidney Int 2008, 74:1444-1453.
• [12]Aladrén Regidor MJ: Cinacalcet reduces vascular and soft tissue calcification in secondary hyperparathyroidism (SHPT) in hemodialysis patients. Clin Nephrol 2009, 71:207-213.
• [13]De Schutter TM, Behets GJ, Jung S, Neven E, D’Haese PC, Querfeld U: Restoration of bone mineralization by cinacalcet is associated with a significant reduction in calcitriol-induced vascular calcification in uremic rats. Calcif Tissue Int 2012, 91:307-315.
• [14]Tsuruta Y, Ohbayashi T, Fujii M, Myochin H, Mizutani R, Narita M, Maeda K: Change in coronary artery calcification score due to cinacalcet hydrochloride administration. Ther Apher Dial 2008, 12:S34-S37.
• [15]Raggi P, Chertow GM, Torres PU, Csiky B, Naso A, Nossuli K, Moustafa M, Goodman WG, Lopez N, Downey G, Dehmel B, Floege J, ADVANCE Study Group: The ADVANCE study: a randomized study to evaluate the effects of cinacalcet plus low-dose vitamin D on vascular calcification in patients on hemodialysis. Nephrol Dial Transplant 2011, 26:1327-1339.