Diphosphonate Treatment of Calcinosis Universalis

Cram, Richard L.; Barmada, Riad; Geho, W. Blair; Ray, Robert D.

doi:10.1056/nejm197110282851808

Cited by 74 publications

(19 citation statements)

References 12 publications

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“…Wound toilet, combined with excision of calcinotic nodules was required in six children and proved to be an effective treatment. While many reports of regression of calcinosis in children on various putatively anticalcinotic therapies have been published [2,3,6,10,11], spontaneous resolution has been documented only infrequently [15]. Our experience, based on study of serial films of 11 children (8 of whom showed evidence of spontaneous resolution), leads us to conclude that this is much more common than the literature suggests and that circumspection is required in assessment of the benefits which have been claimed for specific therapies.…”

Section: Discussionmentioning

confidence: 90%

“…Therapeutic attempts to promote dissolution of abnormal calcium deposits have included low calcium/high phosphate diets, prolonged administration of aluminium hydroxide [10], diphosphonates [2], probenecid [3], and intravenous disodium EDTA [6]. Most of these studies have concerned single [2,10] or few cases [6,11], and no controlled trials have been reported.…”

mentioning

confidence: 99%

“…Most of these studies have concerned single [2,10] or few cases [6,11], and no controlled trials have been reported. Evaluation of the efficacy of anticalcinotic therapies requires detailed knowledge of the natural history of calcinosis, but this is still incomplete in the case of juvenile dermatomyositis.…”

mentioning

confidence: 99%

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Calcinosis in juvenile dermatomyositis

1978

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Abstract.A retrospective study of the radiographs and clinical records of 39 children with dermatomyositis, followed up at a referral centre for a mean period of eight years, revealed calcinosis in 29 children (18 girls, 11 boys). Clinical presentation of calcinosis was unpredictable and variable, ranging from four months to 12 years after onset of disease. Subcutaneous calcification, often associated with subsequent ulceration of overlying skin, was observed more frequently than calcinosis in intermuscular fascial planes (29: 7). Subcutaneous calcinosis was demonstrated most commonly about the knees and elbows and in the fingers and toes. Intermuscular calcinosis affected the fascial planes around the quadriceps, deltoid, biceps, and the gastrocnemius/soleus muscles.Calcinosis, predominantly of the subcutaneous lesions, regressed spontaneously in eight of 11 children where this could be assessed over a one to four year period. This high rate of spontaneous resolution must complicate evaluation of the efficacy of proposed anti-calcinotic treatments in juvenile dermatomyositis.While no apparent relationship was established between subcutaneous calcinosis, which was present in all 29 of the affected patients, and disease activity or severity, all seven children with the additional, extensive, and 'classical' intermuscular fascial plane calcification developed this complication early and had severe disease.These findings are discussed in relation to previous studies.

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Section: Discussionmentioning

confidence: 90%

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confidence: 99%

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Calcinosis in juvenile dermatomyositis

1978

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“…It has been used effectively to prevent renal and aortic calcification induced by vitamin D [47], in the treatment of calcinosis associated with connective tissue diseases [48] and in the reduction of heterotopic ossification associated with hip replacement and spinal cord injury [49], In the clinical report by Thomson and Johnson [31 ], a family with acute vitamin D intoxication was treated with disodium etidronate for 24 days in addi tion to prednisone and experienced reversal of band kera topathy. Two of the family members who had elevated blood urea nitrogen and creatinine after hydration also normalized these indices of renal function within the first month of therapy.…”

Section: Treatment O F Vitamin D Intoxicationmentioning

confidence: 99%

Calcinosis and Metastatic Calcification due to Vitamin D Intoxication

Allen¹,

Shah²

1992

Horm Res

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Vitamin D, a fat-soluble vitamin, can be associated with significant morbidity when prescribed in large doses. We describe a hypoparathyroid patient with vitamin D intoxication who developed painful periarticular calcinosis, nephrocalcinosis with hypertension and chronic renal failure in addition to band keratopathy and hearing loss. He was treated with combination therapy including prednisone, phosphate-binding antacid, phenytoin and disodium etidronate. After 20 months of follow-up there was a significant reduction of periarticular calcinosis, but no improvement in renal function, band keratopathy or hearing loss and possible calcification of the ossicles. The clinicopathologic features of metastatic calcification and the various treatment modalities are reviewed.

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“…EHDP was originally introduced for the treatment of Paget's disease (ALT-MANN et al 1973), myositis ossificans (BASSET et al 1969) and generalized calcinosis (CRAM et al 1971). In 1975 BAUMANN and his colleagues published their experience in the treatment of 9 patients.…”

Section: (Iii) Ethanehydroxydiphosphonate (Ehdp)mentioning

confidence: 99%