Successful Management of Calciphylaxis in a Kidney Transplant Patient: Case Report

Introduction Calciphylaxis is a rare and often fatal condition mostly associated with end-stage renal disease. The pathophysiology remains elusive and treatment options are scarce. We present a rare case of severe calciphylaxis after kidney transplantation in a patient with persistent hyperparathyroidism. Case description A 78-year-old man with a history of end-stage renal disease developed edema and ulcerations on both lower limbs 14 months after kidney transplantation while receiving an mammalian target of rapamycin inhibitor to manage polyoma virus-associated nephropathy. Skin biopsies taken from the ulcerations confirmed calciphylaxis. A multimodal treatment regimen combining medical (calcium-free phosphate binders, cinacalcet, paricalcitol, sodium thiosulfate, antibiotic treatment) and surgical treatments (debridement and autologous skin transplantation) ultimately resulted in successful wound healing. Discussion We describe a case of severe calciphylaxis in a nonuremic patient after kidney transplantation. Rapid diagnosis by skin biopsy and an aggressive multimodal therapy regimen followed by long-term oral sodium thiosulfate treatment were crucial factors for a favorable outcome.

[1]  A. Dominguez,et al.  Calciphylaxis: Controversies in Pathogenesis, Diagnosis and Treatment , 2016, The American journal of the medical sciences.

[2]  S. Somach Calciphylaxis (Calcific Uremic Arteriolopathy) , 2016 .

[3]  G. Frawley,et al.  Hyperbaric oxygen in the treatment of calciphylaxis: A case series and literature review , 2015, Nephrology.

[4]  M. Kamdar,et al.  Calciphylaxis: risk factors, diagnosis, and treatment. , 2015, American journal of kidney diseases : the official journal of the National Kidney Foundation.

[5]  David Steele,et al.  Intralesional sodium thiosulfate for the treatment of calciphylaxis. , 2013, JAMA dermatology.

[6]  S. Brunelli,et al.  Sodium thiosulfate therapy for calcific uremic arteriolopathy. , 2013, Clinical journal of the American Society of Nephrology : CJASN.

[7]  S. Soroka,et al.  Oral Sodium Thiosulfate as Maintenance Therapy for Calcific Uremic Arteriolopathy: A Case Series , 2013, American Journal of Nephrology.

[8]  A. Weaver,et al.  Retrospective analysis of tissue plasminogen activator as an adjuvant treatment for calciphylaxis. , 2013, JAMA dermatology.

[9]  J. Campistol,et al.  Tratamiento eficaz de la arteriolopatía urémica calcificante con bifosfonatos , 2012 .

[10]  J. Campistol,et al.  Successful treatment of calcific uraemic arteriolopathy with bisphosphonates. , 2012, Nefrologia : publicacion oficial de la Sociedad Espanola Nefrologia.

[11]  A. Levin,et al.  Multi-intervention management of calciphylaxis: a report of 7 cases. , 2011, American journal of kidney diseases : the official journal of the National Kidney Foundation.

[12]  E. Thodis,et al.  Calcific Uremic Arteriolopathy Treated with Cinacalcet, Paricalcitol, and Autologous Growth Factors , 2011, Journal of Cutaneous Medicine and Surgery.

[13]  N. Rogers,et al.  Calcific Uremic Arteriolopathy – The Argument for Hyperbaric Oxygen and Sodium Thiosulfate , 2010, Seminars in dialysis.

[14]  V. Liakopoulos,et al.  Pivotal role of paricalcitol in the treatment of calcific uremic arteriolopathy in the presence of a parathyroid adenoma. , 2010, American journal of kidney diseases : the official journal of the National Kidney Foundation.

[15]  P. Cockwell,et al.  Calciphylaxis following kidney transplantation: a case report , 2009, Journal of medical case reports.

[16]  J. Tomažič,et al.  Review of calciphylaxis and treatment of a severe case after kidney transplantation with iloprost in combination with hyperbaric oxygen and cultured autologous fibrin‐based skin substitutes , 2009, Clinical transplantation.

[17]  C. Raymond,et al.  Sodium thiosulfate, bisphosphonates, and cinacalcet for calciphylaxis. , 2009, CANNT journal = Journal ACITN.

[18]  J. Floege,et al.  Sodium thiosulfate in the treatment of calcific uremic arteriolopathy , 2009, Nature Reviews Nephrology.

[19]  P. Kostenuik,et al.  Inhibition of receptor activator of NF-kappaB ligand by denosumab attenuates vascular calcium deposition in mice. , 2009, The American journal of pathology.

[20]  K. Dujardin,et al.  Calciphylaxis in a cardiac patient without renal disease , 2009, Acta cardiologica.

[21]  C. Musso,et al.  Oral sodium thiosulfate solution as a secondary preventive treatment for calciphylaxis in dialysis patients. , 2008, Saudi journal of kidney diseases and transplantation : an official publication of the Saudi Center for Organ Transplantation, Saudi Arabia.

[22]  M. Wolf,et al.  Calciphylaxis from nonuremic causes: a systematic review. , 2008, Clinical journal of the American Society of Nephrology : CJASN.

[23]  Y. Nishimura,et al.  Intractable wounds caused by calcific uremic arteriolopathy treated with bisphosphonates. , 2007, Journal of the American Academy of Dermatology.

[24]  M. Pittelkow,et al.  Calciphylaxis: natural history, risk factor analysis, and outcome. , 2007, Journal of the American Academy of Dermatology.

[25]  M. Drezner,et al.  Calciphylaxis in the absence of end-stage renal disease. , 2006, Endocrine practice : official journal of the American College of Endocrinology and the American Association of Clinical Endocrinologists.

[26]  N. Lameire,et al.  A dramatic case of calciphylaxis 20 years after kidney transplantation. , 2004, Nephrology, Dialysis and Transplantation.

[27]  D. Spector,et al.  Successful treatment of calciphylaxis with intravenous sodium thiosulfate. , 2004, American journal of kidney diseases : the official journal of the National Kidney Foundation.

[28]  P. Price,et al.  Bisphosphonates Alendronate and Ibandronate Inhibit Artery Calcification at Doses Comparable to Those That Inhibit Bone Resorption , 2001, Arteriosclerosis, thrombosis, and vascular biology.

[29]  S. Myers,et al.  Calciphylaxis in patients on hemodialysis: a prevalence study. , 1997, Surgery.

[30]  Z. Twardowski,et al.  Hyperbaric oxygen therapy in calciphylaxis-induced skin necrosis in a peritoneal dialysis patient. , 1994, American journal of kidney diseases : the official journal of the National Kidney Foundation.