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PubMed1995

Calciphylaxis in a chronic hemodialysis patient with protein S deficiency.

Vascular calcifications are common in uremic patients whereas calciphylaxis is rare. We report the case of a 45-year-old woman on chronic hemodialysis since 1977. She had a subtotal parathyroidectomy in 1985, aortic and mitral valve replacement in 1986, and has been treated since then with nicoumalone. In June 1991, she presented with repeated, painful cutaneous necrosis suggesting panniculitis. A skin biopsy showed lobular panniculitis and evidence of calciphylaxis. There was an obvious biological hyperparathyroidism. Protein C functional level was in the normal range whereas protein S functional level was low, i.e. 42%. The patient underwent cervical surgery to remove two parathyroid glands, and daily hemodialysis sessions. Despite this treatment, cutaneous necrosis progressed with superinfection. A few weeks later, the patient died from a septic shock after a myocardic infarction. Necropsy was not performed.

Calciphylaxis↗
PubMed2003

Calciphylaxis in chronic, non-dialysis-dependent renal disease.

BACKGROUND: Calciphylaxis cutis is characterized by media calcification of arteries and, most prominently, of cutaneous and subcutaneous arterioles occurring in renal insufficiency patients. CASE REPORT: A 53-year-old woman with chronic cardiac and renal failure complained of painful crural, non-varicosis ulcers. She was hospitalized in an immobilized condition due to both the crural ulcerations and the existing heart-failure state (NYHA III-IV) having pleural and pericardial effusions, atrial fibrillation and weight loss of 30 kg over the past year. Despite normalization of calcium-phosphorus balance and improvement of renal function, the clinical course of crural ulcerations deteriorated during the following 3 months. After failure of surgical debridements, multiple courses of sterile-maggot therapy were introduced at a late stage to stabilize the wounds. The patient died of recurrent wound infections and sepsis paralleled by exacerbations of renal malfunction. CONCLUSIONS: The role of renal disease in vascular complications is discussed. Sterile-maggot debridement may constitute a therapy for the ulcerated calciphylaxis at an earlier stage, i.e. when first ulcerations appear.

Anemia↗
PubMed2005

Vascular ossification-calcification in metabolic syndrome, type 2 diabetes mellitus, chronic kidney disease, and calciphylaxis-calcific uremic arteriolopathy: the emerging role of sodium thiosulfate.

BACKGROUND: Vascular calcification is associated with metabolic syndrome, diabetes, hypertension, atherosclerosis, chronic kidney disease, and end stage renal disease. Each of the above contributes to an accelerated and premature demise primarily due to cardiovascular disease. The above conditions are associated with multiple metabolic toxicities resulting in an increase in reactive oxygen species to the arterial vessel wall, which results in a response to injury wound healing (remodeling). The endothelium seems to be at the very center of these disease processes, acting as the first line of defense against these multiple metabolic toxicities and the first to encounter their damaging effects to the arterial vessel wall. RESULTS: The pathobiomolecular mechanisms of vascular calcification are presented in order to provide the clinician-researcher a database of knowledge to assist in the clinical management of these high-risk patients and examine newer therapies. Calciphylaxis is associated with medial arteriolar vascular calcification and results in ischemic subcutaneous necrosis with vulnerable skin ulcerations and high mortality. Recently, this clinical syndrome (once thought to be rare) is presenting with increasing frequency. Consequently, newer therapeutic modalities need to be explored. Intravenous sodium thiosulfate is currently used as an antidote for the treatment of cyanide poisoning and prevention of toxicities of cisplatin cancer therapies. It is used as a food and medicinal preservative and topically used as an antifungal medication. CONCLUSION: A discussion of sodium thiosulfate's dual role as a potent antioxidant and chelator of calcium is presented in order to better understand its role as an emerging novel therapy for the clinical syndrome of calciphylaxis and its complications.

Animals↗
PubMed1998

Calcific cerebral embolism in systemic calciphylaxis.

Calciphylaxis represents a rare complication of end-stage renal disease with hyperparathyroidism. We report the case of a 26-year-old woman with systemic calciphylaxis secondary to chronic renal failure who developed mitral annular calcification and a right middle cerebral artery stroke. The high-density lesion seen on CT scan of the brain probably represents a calcified cerebral embolus originating from the mitral valve.

Adult↗
PubMedDate not supplied

Physical and chemical analyses of the mineral substance during the development of two experimental cutaneous calcifications in rats: topical calciphylaxis and topical calcergy.

In subcutaneous calcinoses induced in rats by topical calciphylaxis and topical calcergy, the ultrastructural aspects related to the evolution of calcified deposits were previously described. In the present study by means of X-ray diffraction, infrared spectrometry and electron spin resonance, different biophysical analyses are performed on the mineral substance deposited during development of calciphylaxis and calcergy. A rapid evolution of the calcium phosphate deposits into hydroxyapatite was noticed along the first 20 days of the calcinoses; from then, there is no important modification even at the later stages, and the characteristics of the mineral substance are mostly similar to that of bone tissue. The concentration of trace elements such as Mg2+ and Fe2+, is found higher in cutaneous calcinoses than in bone tissue.

Animals↗
PubMed1995

A dialysis patient with systemic calciphylaxis exhibiting rapidly progressive visceral ischemia and acral gangrene.

Systemic calciphylaxis is a rare, poorly understood syndrome of progressive peripheral ischemic necrosis and medial arterial calcification in patients with end-stage renal disease. We report a patient with this syndrome which developed following corticosteroid administration and who ultimately required amputation of the four extremities. Furthermore, cerebral, myocardial, splenic, and intestinal infarctions also developed in parallel with the increment of visceral arterial calcification. No evidence of noticeable hyperparathyroidism or elevation of serum calcium-phosphate product was observed. We speculated that, in addition to diabetes mellitus and chronic renal failure while undergoing dialysis therapy, the administration of corticosteroids might act synergistically to cause calciphylaxis.

Calciphylaxis↗
PubMed2001

An unusual presentation of calciphylaxis due to primary hyperparathyroidism.

We present the case of a 69-year-old woman with calciphylaxis due to primary hyperparathyroidism. A 0.5-g parathyroid adenoma was surgically removed, which resulted in complete recovery of the patient. Review of the literature revealed 7 other cases of calciphylaxis due to primary hyperparathyroidism and showed that prompt surgical removal of the autonomous parathyroid gland lesion results in clinical recovery of calciphylactic skin lesions.

Adenoma↗
PubMed1998

Calciphylaxis: one hospital's experience.

The lives of the ESRD population have been enhanced and prolonged by improved conservative management, efficient dialysis and successful transplantation. However, practitioners are noting some significant morbidity in these long-term surviving nephrology patients. Clinical signs of renal osteodystrophy can be very subtle and insidious. Bone pain, muscle weakness, and pruritus can be vague symptoms. Calciphylaxis, which is classified as a skin disorder is an extremely rare, painful, and progressive complication. Typically, the patient presents with vascular calcifications, cutaneous ulcerations and tissue necrosis. The treatment regimen for bone disease in the ESRD population will be reviewed. The symptoms of calciphylaxis, diagnostic tests and prognosis will be discussed. Four case studies describe our recent experience. Emphasis will be on the intense multidisciplinary care required for this devastating and often fatal syndrome.

Adult↗
PubMed2000

Severe necrotizing mastopathy caused by calciphylaxis alleviated by total parathyroidectomy.

Calciphylaxis is a complication caused by secondary hyperparathyroidism in patients with chronic renal failure. These patients may present with clinical findings of ischemic necrosis involving the skin and muscle resulting in subsequent gangrene and vascular calcifications. We report a rare case of necrotizing mastopathy caused by calciphylaxis in a 70-year-old female with end-stage renal disease whose symptoms resolved with a total parathyroidectomy.

Aged↗
PubMedDate not supplied

[Report of a case with skin lesions and calciphylaxis].

Calciphylaxis is an unusual complication of chronic renal failure associated with increased morbidity and mortality. This presentation is a clinical case describing a patient parathyreidectomized in 1997, followed by the condition was clinically suspected and then confirmed by skin biopsy. PTHi levels above 400 pg/ml together with scintigraphic images suggesting parathyroid nodules resulted in the decision to try a new surgical procedure, which yielded two nodules of thyroideal tissue. After the surgery and in spite of this pathology result, the patient recovered completely from her symptoms. This corroborates the theory that the etiology of calciphylaxis is multifactorial, acquiring special relevance the use of calcium-containing phosphathe binders (representing a high risk of hypercalcemia) in the treatment of Chronic Renal Disease.

Calciphylaxis↗
PubMed2001

[Calciphylaxis: an uncertain pathogenesis and controversial treatment].

Calciphylaxis is an uncommon but serious disease process that affects mainly patients with advanced renal failure. Calciphylaxis is characterized by dermal arteriolar calcification that leads to skin ulceration, necrosis, ischemia and secondary infection. The pathogenesis is poorly understood, although the calcium phosphorus product has been proposed as a major cause. Given the high morbidity and mortality rate, emphasis should be placed on prevention and early diagnosis of vascular calcification, as well as in prophylaxis of secondary infection. We present changing concepts in four patients receiving dialysis.

Aged↗
PubMed2002

Leg revascularization in patients with calciphylaxis.

Calciphylaxis is a rare complication in patients undergoing hemodialysis. The pathogenesis and risk factors for this disease are poorly understood, although an association with secondary hyperparathyroidism has been suggested. Only two cases of lower-extremity revascularization of patients with calciphylaxis appear in the literature; this report adds two more.

Aged↗
PubMed2004

Systemic lupus erythematosus and calciphylaxis.

Calciphylaxis, a rare condition seen in association with endstage renal disease, is characterized by the appearance of painful, indurated plaques, ecchymosis, ulceration, and eschar formation. We describe a patient with systemic lupus erythematosus, endstage renal disease, and skin lesions first diagnosed as lupus profundus with vasculitis. Further investigation confirmed the diagnosis of extensive calciphylaxis.

Adult↗
PubMedDate not supplied

Post-infectious acute renal failure due to calciphylaxis--when processes go the wrong way round.

Calciphylaxis is a rare life threatening disorder of small- and medium-sized vessel calcification that leads to cutaneous necrosis. While its pathogenesis is uncertain, nearly all cases have been described in patients with end-stage renal disease (ESRD) on dialysis or following renal transplantation which is why the lesion has also been referred to as calcific uremic arteriolopathy. We describe a patient with alcoholic cirrhosis and normal renal function who developed calciphylaxis. Due to infected cutaneous lesions, he developed an acute post-infectious glomerulonephritis with extra capillary proliferation.

Acute Kidney Injury↗
PubMedDate not supplied

Experimental skin calciphylaxis induced by iron citrate sorbitol in young dogs.

An experimental model of skin calciphylaxis using iron citrate sorbitol is presented. There were used for the experiment 12 young dogs sensitized with D3 vitamin and then injected with 0.2 ml Jectofer on the internal face of the shank. Macroscopic lesions become evident after 2-3 days last drug was administered. Nodular calcifications occurred after 7-8 days as white, hard and irregular lesions, when sectioned, presenting dissociable crystals. From the microscopic point of view fatty cysts, dermic granuloma and an amorphous irregular material are described. In the early period Perls stain is positive and becomes negative after 3 or 4 days. Von Kossa reaction is positive after 4 or 5 days and alizarin S after 7. Degenerative lesions of elastic fascicles are noticed and discussed in relationship with localization of calcium salts. On the basis of these data the possible succesion of skin calciphylaxis steps is discussed, but many things remain unknown.

Animals↗
PubMed1995

Calciphylaxis in three patients with end-stage renal disease.

BACKGROUND: Calciphylaxis is a rare and life-threatening condition of progressive cutaneous necrosis secondary to small- and medium-sized vessel calcification seen almost exclusively in patients with end-stage renal disease and hyperparathyroidism. Two patients had bullous lesions preceding their ulcerative lesions, an unusual presentation of this entity. One patient also had penile involvement that, to our knowledge, has not been described previously. OBSERVATIONS: Three patients, all of whom were being maintained on hemodialysis, developed painful, progressive leg ulcerations. Two patients had elevated parathormone levels, and the third patient did not. All patients had only very modest increases in their calcium x phosphate product. CONCLUSIONS: Calciphylaxis should be included in the differential diagnosis of panniculitis and vasculitis. It is important to diagnose promptly, as early treatment may prevent progression.

Adult↗
PubMed1997

Penile calciphylaxis.

Calciphylaxis is a condition of cutaneous necrosis secondary to small- and medium-sized vessel calcification that may progress rapidly and is often fatal. Patients with end-stage renal disease and hyperparathyroidism are almost exclusively at risk. Only 1 case of penile involvement has been previously described. At our institution, a 56-year-old man with end-stage renal disease presented with penile calciphylaxis. The patient received a series of treatments including circumcision, partial penectomy, amputation of necrotic phalanges, and a subtotal parathyroidectomy after which the patient's parathyroid hormone level normalized and the disease progression abated.

Calcinosis↗
PubMed1987

[Calcifying panniculitis associated with renal insufficiency: a tissue calciphylaxis syndrome].

Among the cutaneous manifestations of hyperparathyroidism, cases of panniculitis with calcification of the adipose tissue and necrosis of the skin have recently been reported, the mechanism incriminated being calciphylaxis, as defined by Selye on the basis of experiments. Experimental calciphylaxis consists of local or systemic calcium deposits followed by inflammatory necrosis or sclerosis. The deposits are induced by "provoking" or precipitating factors (metal salts, albumin, traumas) after a phase of sensitization (to parathyroid hormone, vitamins D2 or D3, dihydrotachysterol), provided a critical period is allowed between these two phases; the duration of that period depends on the experimental conditions. The case reported here concerns a 64-year old obese and diabetic woman who had presented with hard and tender nodosities and plaques in her abdominal and crural panniculi, ending in extensive and hyperalgesic necrosis (fig. 1 and 2). The panniculitis had occurred in a peculiar context: at the end of an episode of renal failure complicated with secondary hyperparathyroidism (serum PTH 12.9 mIU/ml; N = 1.5-4.4 mIU/ml) with moderate increase to 5,000 of the P x Ca product. Histological examination of a nodule of the thigh disclosed multiple foci of microcalcification (fig. 3, 4, 5) within the adipose lobules, in the interadipocyte spaces, in connective tissue septa and in the adventitia of small vessels (positive Von Kossa reaction). Electron microscopy showed dense calcium deposits between adipocytes, in subcutaneous septa (fig. 6, 8) and in more or less damaged vascular walls (fig. 9). Within the microfibrillar and granular fundamental substance, microcrystals looking like hydroxyapatite crystals (fig. 7) conglomerated into pincushion-like formations becoming increasingly denser and more compact.(ABSTRACT TRUNCATED AT 250 WORDS)

Abdominal Muscles↗