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PubMed2001

Calciphylaxis is associated with hyperphosphatemia and increased osteopontin expression by vascular smooth muscle cells.

Calciphylaxis or calcific uremic arteriolopathy (CUA) is a fatal disease in dialysis patients due to calcification of cutaneous blood vessels. The pathogenesis has been attributed to elevated parathyroid hormone (PTH). However, recent studies evaluating vascular calcification in nondialysis patients have found that the smooth muscle cells play an active role, including production of the bone matrix protein osteopontin. To examine the involvement of various clinical parameters and smooth muscle cells of CUA, we performed a case-control analysis comparing 10 CUA patients with our current dialysis patients. Available histologic sections were immunostained for osteopontin, markers of smooth muscle cells, endothelial cells, and macrophages. Compared with our current dialysis population, patients with CUA were more likely to be obese, white, and female (P < 0.02). Comparison of laboratory values found CUA patients with lower serum albumin, greater serum phosphorus, and greater calcium X phosphorus product (P < 0.01). In contrast, there was no difference in the concentration of PTH or calcium between the 2 groups. Immunostaining of calcified blood vessels showed that all calcified vessels stained positive for osteopontin, whereas all the noncalcifed vessels showed no osteopontin localization. Staining for smooth muscle alpha-actin decreased in the medial layer with calcification, with cells appearing to be sloughed off, leading to near occlusion of the vessel lumen. Our case-control study demonstrates that hyperphosphatemia and an elevated calcium X phosphorus product is associated with CUA. Histologic examination suggests that the calcification is associated with increased expression of osteopontin by smooth muscle cells.

Adult↗
PubMed2000

Calciphylaxis: a rare limb and life threatening cause of ischaemic skin necrosis and ulceration.

Calciphylaxis (uraemic gangrene syndrome) is a rare complication of chronic renal failure and secondary hyperparathyroidism. Patients present with painful purple skin lesions which undergo necrosis and ulceration. The histology is specific. There is medial calcification with intimal hyperplasia and thrombosis of the lumen of small sized arteries in the underlying subcutaneous tissue. Death frequently arises from overwhelming sepsis. Early recognition of this condition and prompt parathyroidectomy can lead to rapid relief of symptoms and ulcer healing and may be life saving.

Biopsy↗
PubMed1988

A case of calciphylaxis. Case report.

A young woman on hemodialysis with hyperparathyroidism suddenly developed areas of skin necrosis caused by arterial calcification. Parathyroidectomy apparently arrested this process. Seven months after cadaver kidney transplantation, following reparation of a graft artery stenosis, necrosis spread anew from the wound gradually extending to large areas. The patient died and autopsy revealed calcification in many internal organs. The case fits the description of systemic calciphylaxis a syndrome now described in humans but originally experimentally induced in rats.

Adult↗
PubMed1994

Systemic calciphylaxis.

Systemic calciphylaxis is a rare, poorly understood syndrome of progressive ischemic necrosis, usually associated with hyperparathyroidism. The combination of hyperparathyroidism, usually secondary or tertiary, and chronic renal failure seems to produce a particular biochemical environment conducive to the development of an unusual progressive form of ischemia. Early recognition and diagnosis, appropriate therapy including wound care, and expeditious parathyroidectomy often will halt the progression of the disease, minimize limb loss, and prevent subsequent sepsis and death.

Calciphylaxis↗
PubMed1988

Epidermal and follicular calciphylaxis.

A patient with secondary hyperparathyroidism due to chronic renal failure developed epidermal and follicular calcification without concomitant dermal calcification in the lesions of toxic epidermal necrolysis. We propose that the combination of keratinocyte necrosis and an elevated serum calcium-phosphorus product resulted in epidermal calciphylaxis.

Adult↗
PubMed1961

Cutaneous molt induced by calciphylaxis in the rat.

A molt, conducive to the loss and subsequent replacement of all cutaneous layers, can be induced by topical "calciphylaxis" in the rat. This is accomplished by sensitization with dihydrotachysterol followed by challenge with egg white or ferric dextran.

Animals↗
PubMed1969

Calciphylaxis in man.

A patient with renal failure developed widespread calcification in the thighs after the injection of iron-dextran (Imferon). This is considered as an example of calciphylaxis, a process in which calcium is laid down in parts of the body following the administration of a "challenger." Iron-dextran is a known challenger, and should be used with caution in uraemic subjects, who may be sensitized by high serum parathyroid hormone levels. Contributing factors may have been a high calcium/phosphate product, steroids, and the patient's immobility.

Adult↗
PubMed2001

Calciphylaxis.

The phenomenon of calciphylaxis is rare, but potentially fatal. It has been recognised for a long time in patients with chronic renal failure with secondary hyperparathyroidism. Disturbed calcium and phosphate metabolism can result in painful necrosis of skin, subcutaneous tissue and acral gangrene. Appearance of the lesions is distinctive but the pathogenesis remains uncertain. The beneficial effects of parathyroidectomy are controversial. However, correction of hyperphosphataemia or occasionally hypercalcaemia is imperative. Fulminant sepsis as a consequence of secondary infection of necrotic and gangrenous tissue is a frequent cause of patient morbidity and mortality.

Calciphylaxis↗
PubMed2000

Typical features of calciphylaxis in a patient with end-stage renal failure, diabetes mellitus and oral anticoagulation.

We report a multimorbid patient with end-stage renal failure showing a large necrosis and livedo racemosa on the right thigh. Histology revealed medial calcification of the small arteries typical of calciphylaxis. We found the typical features of the disease with different risk factors like elevated calcium-phosphate product, diabetes mellitus and oral anticoagulation. On account of the location of the skin lesions, a bad prognosis was expected. In spite of therapeutical measures with lowering of the calcium and phosphate levels, the patient died 1 month after the diagnosis had been made.

Administration, Oral↗
PubMed1981

Systemic calciphylaxis revisited.

A syndrome characterized by rapidly progressive ischemic necrosis involving large areas of the skin and muscle, and by peripheral gangrene associated with extensive vascular calcifications was observed in a patient with end-stage renal failure on chronic hemodialysis. In an effort to control the disease, parathyroidectomy was performed which resulted in rapid improvement of tissue perfusion. However, the patient eventually died from sepsis within 2 months after admission. This case presents the typical features of the syndrome of systemic calciphylaxis. The literature is reviewed searching for similar cases of this poorly recognized, but life-threatening, clinical syndrome. The pathogenesis, clinical manifestations, and therapy of this unusual and rapidly progressive, but potentially reversible, condition are reviewed with emphasis on its prompt recognition and appropriate management.

Adult↗
PubMed1998

Calciphylaxis: a favorable outcome with hyperbaric oxygen.

A 66-year-old female with diabetes mellitus and end-stage renal disease presented with painful bilateral lower extremity livedo reticularis and necrotic ulcerations. Her distal lower extremity pulses were intact and plethysmographic studies confirmed relatively normal large vessel arterial perfusion. Extensive laboratory analysis was remarkable for an elevated calcium x phosphorous product and parathyroid hormone level. An ulcer biopsy revealed small vessel medial calcinosis, and calciphylaxis was subsequently diagnosed. Despite aggressive wound debridements, antibiotics and subtotal parathyroidectomy, her ulcers failed to improve significantly prompting a trial of hyperbaric oxygen therapy. After 7 weeks of hyperbaric treatments, her ulcers had essentially healed.

Aged↗
PubMed2000

Cutaneous calciphylaxis. An underrecognized clinicopathologic entity.

Calciphylaxis (CPX), an uncommon syndrome characterized, in part, by progressive cutaneous vascular calcification, is seen principally in the setting of renal failure-associated hyperparathyroidism and is difficult to distinguish histologically from other microvasculopathies. We assessed histologic specimens from 13 cases of clinicopathologically classic CPX of the skin and reviewed documented histologic findings in the literature. Our series included 7 "early" and 6 "late" lesions (absence or presence of tissue necrosis, respectively). Histologically, early lesions were subtle and almost inapparent microscopically. Late lesions were easier to recognize because of obvious epidermal ulceration, dermal necrosis, and easily seen mural vascular calcification. The most common finding in both groups was acute and chronic calcifying septal panniculitis. Endovascular fibroblastic proliferation was more common in advanced lesions. Necrosis of dermal collagen was identified in only a few early lesions. Frank luminal vascular thrombosis was infrequent in both groups. The cited histologic findings largely were mirrored by those in the literature. Although they are relatively nonspecific when considered in isolation, the cited histopathologic features of cutaneous CPX allow for the diagnosis of this potentially lethal disorder when they are seen in combination with one another, particularly if detailed clinical data also are available.

Adult↗
PubMedDate not supplied

Calciphylaxis: a complication of end-stage renal disease improved by parathyroidectomy.

A 43-year old Caucasian male with end-stage renal disease presented with painful skin lesions and high calcium phosphate product that did not respond to medical treatment. Skin biopsy confirmed the diagnosis of calciphylaxis. Urgent parathyroidectomy was performed and resulted in decrease in the calcium phosphate product and improvement of his symptoms and signs.

Adult↗
PubMed2001

Calciphylaxis in the patient with chronic renal failure.

Calciphylaxis is a rare, painful, necrotizing skin condition that occurs most frequently in patients with chronic renal failure who are receiving dialysis. These patients commonly have secondary hyperparathyroidism. Treatment involves a multidisciplinary approach. Surgical wound debridement, local wound care, pain control, and nutritional support are the primary care issues that must be addressed. The outcome is often poor, with the fatal outcome often resulting from sepsis.

Calciphylaxis↗
PubMed2000

Calciphylaxis and its relation to end-stage renal disease: a literature review and case presentation.

Calciphylaxis is an uncommon phenomenon usually observed in patients with chronic renal failure who are on dialysis and who have secondary hyperparathyroidism. Treatment is multifocal and may include wound debridement, wound excision, and various methods of achieving closure. Removal of the parathyroid glands often is advocated, but this option remains controversial. Despite treatment, there is a fatal outcome in a majority of cases due to septicemia. Prompt recognition and treatment are imperative to help prevent septicemia. A review of the literature and two case studies are presented.

Aged↗
PubMedDate not supplied

Calciphylaxis in a patient with POEMS syndrome without renal failure and/or hyperparathyroidism. A case report.

POEMS (Crow-Fukase) syndrome is a rare plasma cell lymphoproliferative disorder associated with polyneuropathy (P), organomegaly (O), endocrinopathy (E), monoclonal (M) gammopathy and skin (S) abnormalities. The latter are usually not specific and include hyperpigmentation, hypertrichosis, cutaneous angioma and skin-thickening. A 45-year-old Italian woman was admitted to hospital because of muscle weakness, marked fatigue and paresthesia of the upper and lower extremities. Two and a half years earlier, a POEMS syndrome had been diagnosed on the basis of a history of organomegaly and mild lymphadenopathy, IgA-lambda monoclonal gammopathy, hypothyroidism, severe lower and upper limb sensory-motor peripheral neuropathy and a single osteosclerotic lesion in the left humerus. Eight weeks later, she developed skin lesions bioptically shown to be due to calciphylaxis-induced cutaneous vasculitis. To our knowledge, this is the first case of POEMS syndrome with this peculiar type of vasculitis. The absence of predisposing conditions, namely renal failure, hyperparathyroidism or clotting disorders renders the pathogenetic mechanism(s) of this severe type of vasculitis more intriguing.

Blood Coagulation↗
PubMed1990

Calciphylaxis and systemic calcinosis. Collective review.

The phenomenon of calciphylaxis as defined by Selye is a condition of hypersensitivity that results in acute local calcification of various organs, with a whole host of morbid processes. Nephrocalcinosis and cutaneous calcifications have long been recognized in patients with chronic renal failure, but they have not often been reported in acute hyperparathyroidism or other causes of calcium-phosphate metabolism aberrations. The pathogenesis is not clear, and both the sensitizer and challenging agent in the hypersensitivity theory are often elusive, though hypercalcemia is the most consistent factor. The clinical features vary according to the organs affected and often mimic a gamut of more common conditions. Treatment is primarily supportive, with specific measures only possible when a sensitizer or challenger is identified. Correction of the hypercalcemia is, however, imperative. It is important to recognize this rare condition to avoid the more serious end results often reported.

Calcinosis↗