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PubMed1998

Systemic calciphylaxis associated with massive gastrointestinal hemorrhage.

A 38-year-old woman with insulin-dependent diabetes mellitus, hypertension, and end-stage renal disease developed digital ischemia, widespread cutaneous necrosis and eschar formation of both lower extremities, and extensive ulceration of the large intestine and cecum resulting in gastrointestinal hemorrhage. A mesenteric arteriogram revealed multiple stenotic areas and filling defects of the superior mesenteric artery and its tributaries, suggestive of vasculitis. A diagnosis of calciphylaxis was suspected, on antemortem skin biopsy, and was later confirmed by postmortem examination. This case further documents the relationship between calciphylaxis and significant visceral injury, and it represents, to our knowledge, the first case of calciphylaxis associated with massive gastrointestinal hemorrhage.

Adult↗
PubMed1998

Benign nodular calcification and calciphylaxis in a haemodialysed patient.

Several types of soft tissue calcification can be detected from radiographic evaluation of patients with end-stage renal failure. The factors that predispose to such calcification include an increase in CaxP product in serum, the degree of secondary hyperparathyroidism, the level of blood magnesium, the degree of alkalosis, and the presence of local tissue injury. Three major varieties include calcification of medium-sized arteries, periarticular or tumoral calcification and visceral calcification. Calciphylaxis is a phenomenon consisting of acute ischemic necrosis in presence of calcification of dermohypodermic arterioles. It occurs mostly in chronic renal failure patients with secondary or tertiary hyperparathyroidism with a persistently elevated calcium-phosphorus product. There are few options in treating calciphylaxis and the outcome is generally poor. The authors report the case of a haemodialised patient with benign nodular calcification and calciphylaxis. The coexistence of both entities in the same patient has never been described.

Calcinosis↗
PubMed1998

Uremic small artery disease: calciphylaxis with penis involvement.

Two male patients with chronic renal failure maintained on hemodialysis developed progressive clinical features of ischemic necrosis (so called calciphylaxis) of their extremities and penis. Both patients died. In one patient, penectomy provided tissue for histopathologic examination and there were changes of small artery calcification. A role of iron overload on the production of calciphylaxis is reviewed. This report is perhaps the first in nephrology literature on the occurrence of calciphylaxis involving penis and prepuce.

Aged↗
PubMed2001

Calciphylaxis causing necrotizing mastitis: a case report.

Calciphylaxis is a rare condition most commonly related to ESRD and hyperparathyroidism. We report a case of necrotizing mastitis caused by calciphylaxis following routine breast biopsy for microcalcifications. Early recognition of the potential for this condition should help in clinical management of patients with calciphylaxis.

Journal Article↗
PubMed2000

Rhabdomyolysis as the presenting manifestation of calciphylaxis.

A 43-year-old woman was admitted with progressive leg pains and weakness and was found to have rhabdomyolysis. Prior to this admission the patient had hypercalcemia, but this returned to normal following treatment with calcitonin. During the hospitalization, she developed the syndrome of calciphylaxis consisting of necrotic skin and muscle associated with vascular calcification. This is the first case report of rhabdomyolysis caused by calciphylaxis in a patient without chronic renal failure.

Adult↗
PubMed2001

Calciphylaxis, a poorly understood clinical syndrome: three case reports and a review of the literature.

Systemic calcification syndromes are a recognized complication occurring in some patients with end-stage renal disease (ESRD) and secondary hyperparathyroidism. These patients develop severe livedo reticularis and subcutaneous tissue lesions progressing to frank necrosis and ultimately large areas of eschar. Clinically this syndrome is known as calciphylaxis; these lesions are usually resistant to aggressive debridement, systemic antibiotics, and revascularization procedures. We report three patients with somewhat different clinical presentations but all sharing a common link of exquisitely painful leg ulcers initially being treated as ischemic lesions or venous stasis-type ulcerations. These three patients were diagnosed with calciphylaxis on the basis of clinical, biochemical, and histopathological criteria. Two patients underwent parathyroidectomy late in the progression of their disease, with some resolution of their ulcerative lesions.

Aged↗
PubMed2002

Calciphylaxis in a patient with rheumatoid arthritis without renal failure and hyperparathyroidism: the possible role of long-term steroid use and protein S deficiency.

Calciphylaxis is a rare and life-threatening condition of progressive cutaneous necrosis secondary to small and medium-sized vessel calcification that is seen almost exclusively in patients with end-stage renal disease and hyperparathyroidism. We report a case of calciphylaxis that may very well be due to a long-term steroid use in a rheumatoid arthritis patient who had neither end-stage renal disease nor hyperparathyroidism. We also discuss the possible role of protein S deficiency as a contributing factor.

Adult↗
PubMed1992

Widespread cutaneous and systemic calcification (calciphylaxis) in patients with the acquired immunodeficiency syndrome and renal disease.

BACKGROUND: Calciphylaxis is a form of widespread calcification that may occur in patients with renal disease and hyperparathyroidism. The skin is often affected secondary to vascular compromise and ischemia. Patients infected with the human immunodeficiency virus (HIV) are predisposed to renal failure and thus may develop this serious complication. OBJECTIVE: The purpose of this article is to describe two cases of fatal calciphylaxis in patients with the acquired immunodeficiency syndrome and renal disease. METHODS: Two patients were examined clinically and through the performance of serologic studies and skin biopsies. RESULTS: Both patients had a widespread livedo reticularis-like eruption, which rapidly progressed to cutaneous gangrene. Patient 1 had elevated serum calcium and phosphorus levels whereas patient 2 had normal values but had markedly elevated serum parathyroid hormone levels. Biopsy specimens in both cases showed vascular thrombosis and soft tissue calcification. CONCLUSION: Widespread calcification may develop in patients with HIV infection and renal failure and, in some cases, calcium and phosphorus levels may be normal.

Acquired Immunodeficiency Syndrome↗
PubMed2003

Therapy for calciphylaxis: an outcome analysis.

BACKGROUND: Violaceous skin lesions that progress to nonhealing ulcerations and gangrene characterize calciphylaxis. These lesions, which are associated with secondary hyperparathyroidism, are resistant to medical therapy and may lead to amputation, uncontrollable sepsis, and death. METHODS: In this retrospective analysis, patient, disease, and treatment variables were obtained from the patients' medical records. Paired t-tests, chi-square analysis, and two-tailed Fisher's exact tests compared variables. Survival curves were generated using the Kaplan-Meier method; the log-rank test determined survival differences. RESULTS: From 1993 to 2001, 35 patients were treated; 26 (74%) were female and 20 (57%) were African-American. The cause of the renal failure was hypertension in 37% and diabetes mellitus in 26%. Twenty-three patients (66%) underwent a parathyroid resection involving 4 glands and autotransplantation in 12, 3(1/2) glands in 7, and <3(1/2) glands in 4. Median follow-up was 26 months (range 2 to 90 months). Surgical patients showed improvement in serum calcium, phosphate, and parathyroid hormone values (P<.05) and had a longer median overall survival (80 months) than nonsurgical patients (35 months) (P<.001). CONCLUSIONS: Parathyroid resection for calciphylaxis reduces serum calcium, phosphate, and parathyroid hormone values and improves clinical response and overall survival.

Adult↗
PubMed1994

Hyperbaric oxygen therapy in calciphylaxis-induced skin necrosis in a peritoneal dialysis patient.

A 58-year-old white woman on continuous ambulatory peritoneal dialysis for 2 years developed calciphylaxis-induced necrotic skin lesions over both lower extremities. Despite subtotal parathyroidectomy and other conventional measures, skin lesions continued to worsen. Mapping of transcutaneous oxygen pressure showed markedly low values in involved areas. Skin ulcers completely healed after 38 sessions of hyperbaric oxygen therapy. The results in our case indicate that hyperbaric oxygen therapy may be useful in the treatment of skin ulcers secondary to calciphylaxis.

Calciphylaxis↗
PubMed1996

Proximal calciphylaxis in four insulin-requiring diabetic hemodialysis patients.

We present four cases of proximal calciphylaxis in end-stage renal disease patients treated with hemodialysis. All patients were diabetic and developed lesions in areas that had previously served as sites of insulin injection. We review the presentation, pathogenesis, pathology, prognosis, and treatment of this devastating condition. Finally, we hypothesize that subcutaneous injection of insulin may play a pathogenic role in the development of proximal calciphylaxis.

Abdomen↗
PubMed2000

Calciphylaxis in a patient without renal failure or elevated parathyroid hormone: possible aetiological role of chemotherapy.

Calciphylaxis is a rare, often fatal disease characterized clinically by progressive cutaneous necrosis and ulceration, and histologically by vascular calcification and thrombosis. It has been described in association with end-stage renal disease, after initiation of dialysis, following renal transplantation, and in patients with hyperparathyroidism. We present the first case of calciphylaxis occurring in a patient with both normal renal function and parathyroid hormone level and discuss the possible aetiological role of chemotherapy-induced functional protein C and protein S deficiency.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed2003

Calciphylaxis in a patient with alcoholic liver disease in the absence of renal failure.

Calciphylaxis is a rare, potentially life-threatening syndrome characterized by progressive microvascular and superficial soft tissue calcification, usually seen in patients with chronic renal failure. We describe an unusual case of calciphylaxis in a patient with alcoholic liver disease and normal renal function who responded well to conservative wound care.

Calciphylaxis↗
PubMed2001

Calciphylaxis: a poor prognostic indicator for limb salvage.

INTRODUCTION: Calciphylaxis (calcific uremic arteriolopathy) is a rare complication of end-stage renal disease in patients with secondary hyperparathyroidism. Subcutaneous skin nodules and extensive tissue necrosis with vascular calcification characterize this process. DESIGN AND SETTING: The design of the study included case series and literature review. The study was set in a tertiary care hospital. PATIENTS: Four patients presented over an 8-month period with extensive tissue loss and the subsequent diagnosis of calciphylaxis. Two of these patients were revascularized. One underwent primary amputation, and the final patient died before revascularization. The mortality rate for this group was 75%. All patients had significant vascular complications outside the area of initial presentation. CONCLUSIONS: These individuals represent a subset of patients who may not benefit from revascularization or may require extensive regulation of divalent metabolism before consideration for revascularization.

Aged↗
PubMedDate not supplied

Intensive tandem cryofiltration apheresis and hemodialysis to treat a patient with severe calciphylaxis, cryoglobulinemia, and end-stage renal disease.

This is the first report on tandem cryofiltration apheresis (CFA) and hemodialysis (HD). A 44 year old white man with Type II mixed cryoglobulinemia, hepatitis C virus (HCV), severe skin lesions, and end-stage renal disease (ESRD) on maintenance hemodialysis was air-transferred for CFA, which is only available at our medical center. The patient failed to respond to high dose steroids, immunosuppression, intravenous immunoglobulin (IVIG), and plasma exchange for the treatment of his cryoglobulinemia, and he failed alpha-interferon therapy for his HCV. On arrival, he was also found to have severe calciphylaxis secondary to ESRD with generalized, painful skin ulceration, necrosis, and penile gangrene. To treat both conditions, intensive, tandem CFA/HD was initiated. He received extensive wound care and surgical debridement. To prevent pressure ulcers and worsening of skin lesions, he was placed on the FluidAir (Kinetic Concepts Inc., San Antonio, TX) controlled air bed. The patient received 18 tandem CFA/HD treatments, and four extra HD treatments in one month. Sodium citrate was used as an anticoagulant for the CFA procedure. His plasma cryoglobulin (CG) level dropped from 6,157 to 420 microg/ml, and his calciphylaxis also improved. The CFA effectively removed 93% of CG, without significant removal of IgG, IgM, IgA, albumin, and fibrinogen. No albumin or fresh frozen plasma (FFP) was required as replacement fluid for CFA. No citrate toxicity or evidence of complement activation with the cryofilter was observed. The entire CFA procedure time (3(1/2) hours) was considered. Intensive, tandem CFA/HD was performed in a critically ill patient with no apparent adverse consequences.

Adult↗
PubMed2001

Control of serum phosphorus: implications for coronary artery calcification and calcific uremic arteriolopathy (calciphylaxis).

There is mounting evidence that elevated serum phosphorus is an important cardiovascular risk factor in patients with end stage renal disease. Recent work has shown that vascular smooth muscle cells have the ability to undergo osteoblastic differentiation and produce an environment conducive to mineralization. Serum phosphorus is an important stimulator of this process and the adverse cardiovascular effects of hyperphosphatemia are most likely mediated via its ability to enhance the development of vascular calcification. Arterial calcification, whether it is intimal or medial in location, is a strong independent risk factor for cardiovascular morbidity and mortality. Both coronary artery calcification and calciphylaxis are prototypical examples of arterial calcification that have been associated with poor phosphate control. Furthermore, several investigators have recently suggested that the prescription of large doses of calcium to achieve phosphate control may augment, rather than diminish, the risk of vascular calcification. This is more likely to be true in the presence of low turnover bone disease, a diagnosis difficult to make with routine laboratory testing. A brief review of the molecular biology of vascular calcification supports the concept that warfarin administration may exacerbate the calcific process, particularly in the setting of hyperphosphatemia, as has been reported in patients with calciphylaxis. Recognizing the consequences of poor phosphate control, it is time to adopt strict target levels that aim to normalize serum phosphorus levels. The available evidence supports that this control should not be achieved through the use of supraphysiologic doses of supplemental calcium.

Arterioles↗
PubMed2003

Early recognition and treatment of calciphylaxis.

Calciphylaxis is a condition with a high mortality rate that is often found in patients with renal failure. It is characterized by soft tissue calcification and painful skin ulceration. A serum calcium-phosphorus product of more than 60 mg2/dl2 indicates great risk for calciphylaxis. The diagnosis is made on the basis of an incisional biopsy showing calcification of the small, subcutaneous arteries.

Aged↗
PubMed2001

Fulminant and relentless cutaneous necrosis with excruciating pain caused by calciphylaxis developing in a patient undergoing peritoneal dialysis.

A 50-year-old Japanese female with chronic renal failure who had been on continuous ambulatory peritoneal dialysis developed fulminant systemic cutaneous necrosis that began as painful livedo reticularis-like skin lesions on her thighs. Because of disseminated vascular calcification within the muscular layer of her lower limbs, we eventually diagnosed her with calciphylaxis. The skin necrosis progressed rapidly, and she died of sepsis and pneumonia on the 53rd hospital day. In addition to her long-lasting severe hyperparathyroidism and extremely elevated serum phosphorus and calcium levels, mechanical, frictional stimulation inflicted on the local skin and administration of corticosteroids were suspected to have precipitated the calciphylaxis. Our lack of awareness of this disease in its early stages resulted in our missing the chance to do a parathyroidectomy that might have changed the course. It is important to know the clinical features of this rare disease in order to make a diagnosis as early as possible.

Calciphylaxis↗