Atypical calciphylaxis in a patient receiving warfarin then resolving with cessation of warfarin and application of hyperbaric oxygen therapy

Clin Appl Thromb Hemost. 2010 Jun;16(3):345-50. doi: 10.1177/1076029609355588. Epub 2009 Dec 16.

Abstract

Calciphylaxis is a rare, usually fatal vasculopathic disorder characterized by cutaneous ischemia and necrosis due to calcification of arterioles. Although calciphylaxis is most frequently associated with end-stage renal disease (ESRD) and secondary hyperparathyroidism, it has been reported infrequently among patients on warfarin. No standard treatment has been established for atypical calciphylaxis; however, a potentially beneficial treatment is hyperbaric oxygen therapy (HBOT). A high degree of clinical suspicion, early diagnosis, and understanding the pathophysiology of this disease promotes the optimal management of this extremely morbid and often fatal condition.

Case report: We present a 63-year-old Polynesian woman with biopsy-proven calciphylaxis in the absence of ESRD or elevated serum calcium levels while taking warfarin. Therapeutic dose enoxaparin was substituted for warfarin and she received 40 sessions of HBOT during which lower extremity ulcers resolved.

Discussion: Warfarin has been implicated when calciphylaxis presents in an atypical fashion. No guidelines exist for treatment of atypical calciphylaxis in the setting of concomitant warfarin therapy. Up to 80% of calciphylaxis patients die within 1 year of diagnosis. Our patient was changed to low-molecular-weight heparin and received HBOT.

Conclusion: We present what we believe is the first case of atypical calciphylaxis thought to be attributable to warfarin treated with a therapeutic substitution of anticoagulant and HBOT leading to resolution of cutaneous lesions.

Publication types

  • Case Reports

MeSH terms

  • Anti-Bacterial Agents / therapeutic use
  • Anticoagulants / adverse effects*
  • Anticoagulants / therapeutic use
  • Calciphylaxis / chemically induced*
  • Calciphylaxis / diagnosis
  • Calciphylaxis / surgery
  • Calciphylaxis / therapy
  • Calcium-Binding Proteins / antagonists & inhibitors
  • Cellulitis / diagnosis
  • Cellulitis / drug therapy
  • Combined Modality Therapy
  • Debridement
  • Diabetes Mellitus, Type 2 / complications
  • Extracellular Matrix Proteins / antagonists & inhibitors
  • Female
  • Humans
  • Hyperbaric Oxygenation*
  • Leg Ulcer / etiology*
  • Leg Ulcer / surgery
  • Leg Ulcer / therapy
  • Matrix Gla Protein
  • Middle Aged
  • Obesity, Morbid / complications
  • Risk Factors
  • Thrombophilia / drug therapy
  • Warfarin / adverse effects*
  • Warfarin / therapeutic use

Substances

  • Anti-Bacterial Agents
  • Anticoagulants
  • Calcium-Binding Proteins
  • Extracellular Matrix Proteins
  • Warfarin