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Tumoral Calcinosis: Controversies in the Etiology and Alternatives in the Treatment

Tumoral Calcinosis: Controversies in the Etiology and Alternatives in the Treatment Abstract Objective: To examine our experience and review the literature concerning the diagnosis, origin, and treatment of tumoral calcinosis (TC). Design/Setting: Case series based on patients with TC treated in University of California—San Francisco hospitals from 1981 to 1992 and the review of the patients described in the English-language literature. Patients: The study included a total of 17 patients: 10 women and seven men. Main Outcome Measures: Sex, age, origin, symptoms, localization, treatment, and morbidity. Results: Seven men and six women, from 32 to 62 years of age, had known disorders of calcium metabolism, and four women, from 37 to 84 years of age, did not. The main causes of the calcium metabolic disorder were secondary hyperparathyroidism in 11 patients (85%) and primary hyperparathyroidism in two patients. In three patients there was a history of trauma at the involved site and in one patient the origin was unknown. Swelling and pain are the most common presenting complaints. Generalized pruritus was observed in 54% of the patients with metabolic disorders (P<.001) but not in patients without metabolic disorders. Among our patients with metabolic disorders, TC occurred most frequently at the shoulder (46%) and elbow (31%). Eleven patients with secondary hyperparathyroidism had received calcium carbonate to bind phosphate, a high level of calcium in the dialysate, and calcitriol (1,25-vitamin D) either orally, intravenously, or both, and three received epoetin alfa (Epogen). Following parathyroidectomy, the patients with hyperparathyroidism improved symptomatically, although calcifications did not change in size. One patient had the calcifications resected and did well, whereas another was treated by subtotal resection and had a recurrence 3 years later. All four of our patients without a metabolic disorder had complete resection of TC with no recurrence. Conclusion: We believe TC is becoming more common in uremic patients with secondary hyperparathyroidism because of recent changes in the medical treatment of these patients. The increased use of calcium carbonate to bind phosphate as well as calcitriol and calcium to suppress parathyroid function and possibly epoetin alfa are causing more patients to develop TC.(Arch Surg. 1993;128:737-745) References 1. Duret MH. Tumors multiples et singularies des burses sereuses (endotheliomes, peut etre d'origine parasitaire) . Bull Soc Anat . 1899;74:725-731. 2. Inclan A. Tumoral calcinosis . JAMA . 1943;121:490-495.Crossref 3. McKee PH, Liomba NG, Hutt MSR. Tumoral calcinosis: a pathological study of sixty-six cases . Br J Dermatol . 1982;107:669-674.Crossref 4. Berg D. Tumoral calcinosis . Br J Surg . 1972;59:570-571.Crossref 5. Palmer PES. Tumoral calcinosis . Br J Radiol . 1966;39:518-525.Crossref 6. Knowles S, Declerck G. Anthony P. Tumoral calcinosis . Br J Surg . 1983;70: 105-107.Crossref 7. Crock AD, Silver RM. Tumoral calcinosis presenting as adhesive capsulitis: case report and literature review . Arthritis Rheum . 1987;30:455-459.Crossref 8. Teutschlaender O. Uber progressive Lipogranulomatose der Muskulatur Zugleich ein Beitrag zur Pathogenese der Myopathica Osteoplastica Progressiva . Klin Wochenschr . 1935;14:451-453.Crossref 9. Veress B, Malik MOA, El Hassan AM. Tumoral calcinosis: a clinicopathological study of 20 cases . J Pathol . 1976;119:113-118.Crossref 10. Ghormley RK, McCrary WE. Multiple calcified bursae and calcified soft tissues . Trans West Surg Assoc . 1942;51:292-309. 11. Thompson JG. Calcifying collagenolysis (tumoral calcinosis) . Br J Radiol . 1966; 39:526-532.Crossref 12. Harkess IW, Peters HJ. Tumoral calcinosis: a report of six cases . J Bone Joint Surg Am . 1967;49:721-731. 13. Prince MJ, Schaeffer PC, Goldsmith RS, Chausmer AB. Hyperphosphatemic tumoral calcinosis: association with elevations of serum 1,25-dihydroxycholecalciferol concentrations . Ann Intern Med . 1982;96:586-591.Crossref 14. Clarke E, Swischuk LE, Hayden CK Jr. Tumoral calcinosis, diaphysis and hyperphosphatemia . Radiology . 1984;151:643-646.Crossref 15. Lyles KW, Halsey DL, Friedman NE, Lobaugh B. Correlations of serum concentrations of 1,25-dihydroxyvitamin D, phosphorus and parathyroid hormone in tumoral calcinosis . J Clin Endocrinol Metab . 1988:67:88-92.Crossref 16. Lyles KW, Burkes El, Ellis GJ, Lucas KS, Dolan EA, Drezner MK. Genetic transmission of tumoral calcinosis: autosomal dominant with variable clinical expressivity . J Clin Endocrinol Metab . 1985;60:1093-1096.Crossref 17. Ressnik CS. Tumoral calcinosis . Arthritis Rheum . 1989;32:1484-1486.Crossref 18. Baldursson H, Evans EB, Dodge WF, Jackson WT. Tumoral calcinosis with hyperphosphatemia: a report of a family with incidence in 4 siblings . J Bone Joint Surg Am . 1969;51:913-925. 19. Lafferty FW, Reynolds ES, Pearson OH. Tumoral calcinosis: a metabolic disease of unknown etiology . Am J Med . 1965;38:105-118.Crossref 20. McClatchie S, Bremner AD. Tumoral calcinosis: an unrecognized disease . Br J Med . 1969;1:153-155.Crossref 21. Hug I, Guncaga J. Tumoral calcinosis with sedimentation sign . Br J Radiol . 1974;47:734-736.Crossref 22. Pearson J, Clark OH. Heterotopic calcification in abdominal wounds . Surg Gynecol Obstet . 1978;146:371-374. 23. Duh Q-Y, Lim RC, Clark OH. Calciphylaxis in secondary hyperparathyroidism: diagnosis and parathyroidectomy . Arch Surg . 1991;126:1213-1219.Crossref 24. Gregosiewicz A, Warda E. Tumoral calcinosis: successful medical treatment: a case report . J Bone Joint Surg Am . 1989;71:1244-1249. 25. Andress DL, Norris KC, Coburn JW, Slatopolsky EA, Sherrard DJ. Intravenous calcitriol in the treatment of refractory osteitis fibrosa of chronic renal failure . N Engl J Med . 1989;321:274-279.Crossref 26. Mitnick PP, Goldfarb S, Slatopolsky E, Lemann J, Gray RW, Zalman AS. Calcium and phosphate metabolism in tumoral calcinosis . Ann Intern Med . 1980; 92:482-487.Crossref 27. Ibels LS. The pathogenesis of metastastic calcification in uremia . Prog Biochem Pharmacol . 1980;17:242-250. 28. Khafif RA, DeLima C, Silverberg A, Frankul R. Calciphylaxis and systemic calcinosis: collective review . Arch Intern Med . 1990;150:956-959.Crossref 29. Suzuki K, Takahashi S, Ito K, Tanaka Y, Sezai Y. Tumoral calcinosis in a patient undergoing hemodialysis . Acta Orthop Scand . 1979;50:27-31.Crossref 30. Demeure MJ, McGee DC, Wilkes W, Duh Q-Y, Clark OH. Results of surgical treatment for hyperparathyroidism associated with renal disease . Am J Surg . 1990;160:337-340.Crossref 31. Walker GS, Davison AM, Peacock M, McLahlan MSF. Tumoral calcinosis: a manifestation of extreme metastatic calcification occurring with 1 alpha-hydroxycholecalciferol therapy . Postgrad Med J . 1977;53:570-573.Crossref 32. Godfrey RS, Faddes DM, Daroca PJ Jr, Sutherland CM. Tumoral calcinosis and hyperparathyroidism . J La State Med Soc . 1987;139:26-32. 33. Luflin EG, Wilson DM, Smith LH, et al. Phosphorus excretion in tumoral calcinosis: response to parathyroid hormone and azetalomide . J Clin Endocrinol Metab . 1980;50:648-653.Crossref 34. Barton DL, Reeves RJ. Tumoral calcinosis: report of 3 cases and review of literature . Ann Intern Med . 1980;92:482-487.Crossref 35. Prasad VLN, Naresh KN, Govind Krishna SR, Ananthalerishman N, Veliath AL. Tumoral calcinosis . World J Surg . 1989;13:803-808.Crossref 36. Eisenberg B, Tzamaloukas AH, Hartstone MF, Listrom MB, Arrigton ER, Sherrad DJ. Periarticular tumoral calcinosis and hypercalcemia in a hemodialysis patient without hyperparathyroidism: a case report . J Nucl Med . 1990:31:1099-1103. 37. Hacihanefioglu U. Tumoral calcinosis: a clinical and pathological study of eleven unreported cases in Turkey . J Bone Joint Surg Am . 1978;60:1131-1135. 38. Mozaffarian G, Lafferty FW, Bearson OH. Treatment of tumoral calcinosis with phosphorus deprivation . Ann Intern Med . 1972;77:741-745.Crossref 39. Kirk TS, Simon MA. Tumoral calcinosis: report of a case with successful medical management . J Bone Joint Surg Am . 1981;63:1176-1179. 40. Davies M, Clements MR, Mawer EB, Fremont AJ. Tumoral calcinosis: clinical and metabolic response to phosphorus deprivation . Q J Med . 1987;63:493-498. 41. Walentynowicz JE, Mahoney MD, Saldane MJ. Tumoral calcinosis: case report with treatment failure . Orthop Rev . 1989;18:687-690. 42. Kallmeyer JC, Seimon LP, MacSerraigh ET. The effect of thyrocalcitonin therapy and phosphate deprivation on tumoral calcinosis . S Afr Med J . 1978;54: 963-966. 43. Sonin A, Nance EP Jr. Juxtaarticular calcification in secondary hyperparathyroidism: resorption after parathyroidectomy . South Med J . 1991;84:641-643. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Surgery American Medical Association

Tumoral Calcinosis: Controversies in the Etiology and Alternatives in the Treatment

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Publisher
American Medical Association
Copyright
Copyright © 1993 American Medical Association. All Rights Reserved.
ISSN
0004-0010
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1538-3644
DOI
10.1001/archsurg.1993.01420190027004
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Abstract

Abstract Objective: To examine our experience and review the literature concerning the diagnosis, origin, and treatment of tumoral calcinosis (TC). Design/Setting: Case series based on patients with TC treated in University of California—San Francisco hospitals from 1981 to 1992 and the review of the patients described in the English-language literature. Patients: The study included a total of 17 patients: 10 women and seven men. Main Outcome Measures: Sex, age, origin, symptoms, localization, treatment, and morbidity. Results: Seven men and six women, from 32 to 62 years of age, had known disorders of calcium metabolism, and four women, from 37 to 84 years of age, did not. The main causes of the calcium metabolic disorder were secondary hyperparathyroidism in 11 patients (85%) and primary hyperparathyroidism in two patients. In three patients there was a history of trauma at the involved site and in one patient the origin was unknown. Swelling and pain are the most common presenting complaints. Generalized pruritus was observed in 54% of the patients with metabolic disorders (P<.001) but not in patients without metabolic disorders. Among our patients with metabolic disorders, TC occurred most frequently at the shoulder (46%) and elbow (31%). Eleven patients with secondary hyperparathyroidism had received calcium carbonate to bind phosphate, a high level of calcium in the dialysate, and calcitriol (1,25-vitamin D) either orally, intravenously, or both, and three received epoetin alfa (Epogen). Following parathyroidectomy, the patients with hyperparathyroidism improved symptomatically, although calcifications did not change in size. One patient had the calcifications resected and did well, whereas another was treated by subtotal resection and had a recurrence 3 years later. All four of our patients without a metabolic disorder had complete resection of TC with no recurrence. Conclusion: We believe TC is becoming more common in uremic patients with secondary hyperparathyroidism because of recent changes in the medical treatment of these patients. The increased use of calcium carbonate to bind phosphate as well as calcitriol and calcium to suppress parathyroid function and possibly epoetin alfa are causing more patients to develop TC.(Arch Surg. 1993;128:737-745) References 1. Duret MH. Tumors multiples et singularies des burses sereuses (endotheliomes, peut etre d'origine parasitaire) . Bull Soc Anat . 1899;74:725-731. 2. Inclan A. Tumoral calcinosis . JAMA . 1943;121:490-495.Crossref 3. McKee PH, Liomba NG, Hutt MSR. Tumoral calcinosis: a pathological study of sixty-six cases . Br J Dermatol . 1982;107:669-674.Crossref 4. Berg D. Tumoral calcinosis . Br J Surg . 1972;59:570-571.Crossref 5. Palmer PES. Tumoral calcinosis . Br J Radiol . 1966;39:518-525.Crossref 6. Knowles S, Declerck G. Anthony P. Tumoral calcinosis . Br J Surg . 1983;70: 105-107.Crossref 7. Crock AD, Silver RM. Tumoral calcinosis presenting as adhesive capsulitis: case report and literature review . Arthritis Rheum . 1987;30:455-459.Crossref 8. Teutschlaender O. Uber progressive Lipogranulomatose der Muskulatur Zugleich ein Beitrag zur Pathogenese der Myopathica Osteoplastica Progressiva . Klin Wochenschr . 1935;14:451-453.Crossref 9. Veress B, Malik MOA, El Hassan AM. Tumoral calcinosis: a clinicopathological study of 20 cases . J Pathol . 1976;119:113-118.Crossref 10. Ghormley RK, McCrary WE. Multiple calcified bursae and calcified soft tissues . Trans West Surg Assoc . 1942;51:292-309. 11. Thompson JG. Calcifying collagenolysis (tumoral calcinosis) . Br J Radiol . 1966; 39:526-532.Crossref 12. Harkess IW, Peters HJ. Tumoral calcinosis: a report of six cases . J Bone Joint Surg Am . 1967;49:721-731. 13. Prince MJ, Schaeffer PC, Goldsmith RS, Chausmer AB. Hyperphosphatemic tumoral calcinosis: association with elevations of serum 1,25-dihydroxycholecalciferol concentrations . Ann Intern Med . 1982;96:586-591.Crossref 14. Clarke E, Swischuk LE, Hayden CK Jr. Tumoral calcinosis, diaphysis and hyperphosphatemia . Radiology . 1984;151:643-646.Crossref 15. Lyles KW, Halsey DL, Friedman NE, Lobaugh B. Correlations of serum concentrations of 1,25-dihydroxyvitamin D, phosphorus and parathyroid hormone in tumoral calcinosis . J Clin Endocrinol Metab . 1988:67:88-92.Crossref 16. Lyles KW, Burkes El, Ellis GJ, Lucas KS, Dolan EA, Drezner MK. Genetic transmission of tumoral calcinosis: autosomal dominant with variable clinical expressivity . J Clin Endocrinol Metab . 1985;60:1093-1096.Crossref 17. Ressnik CS. Tumoral calcinosis . Arthritis Rheum . 1989;32:1484-1486.Crossref 18. Baldursson H, Evans EB, Dodge WF, Jackson WT. Tumoral calcinosis with hyperphosphatemia: a report of a family with incidence in 4 siblings . J Bone Joint Surg Am . 1969;51:913-925. 19. Lafferty FW, Reynolds ES, Pearson OH. Tumoral calcinosis: a metabolic disease of unknown etiology . Am J Med . 1965;38:105-118.Crossref 20. McClatchie S, Bremner AD. Tumoral calcinosis: an unrecognized disease . Br J Med . 1969;1:153-155.Crossref 21. Hug I, Guncaga J. Tumoral calcinosis with sedimentation sign . Br J Radiol . 1974;47:734-736.Crossref 22. Pearson J, Clark OH. Heterotopic calcification in abdominal wounds . Surg Gynecol Obstet . 1978;146:371-374. 23. Duh Q-Y, Lim RC, Clark OH. Calciphylaxis in secondary hyperparathyroidism: diagnosis and parathyroidectomy . Arch Surg . 1991;126:1213-1219.Crossref 24. Gregosiewicz A, Warda E. Tumoral calcinosis: successful medical treatment: a case report . J Bone Joint Surg Am . 1989;71:1244-1249. 25. Andress DL, Norris KC, Coburn JW, Slatopolsky EA, Sherrard DJ. Intravenous calcitriol in the treatment of refractory osteitis fibrosa of chronic renal failure . N Engl J Med . 1989;321:274-279.Crossref 26. Mitnick PP, Goldfarb S, Slatopolsky E, Lemann J, Gray RW, Zalman AS. Calcium and phosphate metabolism in tumoral calcinosis . Ann Intern Med . 1980; 92:482-487.Crossref 27. Ibels LS. The pathogenesis of metastastic calcification in uremia . Prog Biochem Pharmacol . 1980;17:242-250. 28. Khafif RA, DeLima C, Silverberg A, Frankul R. Calciphylaxis and systemic calcinosis: collective review . Arch Intern Med . 1990;150:956-959.Crossref 29. Suzuki K, Takahashi S, Ito K, Tanaka Y, Sezai Y. Tumoral calcinosis in a patient undergoing hemodialysis . Acta Orthop Scand . 1979;50:27-31.Crossref 30. Demeure MJ, McGee DC, Wilkes W, Duh Q-Y, Clark OH. Results of surgical treatment for hyperparathyroidism associated with renal disease . Am J Surg . 1990;160:337-340.Crossref 31. Walker GS, Davison AM, Peacock M, McLahlan MSF. Tumoral calcinosis: a manifestation of extreme metastatic calcification occurring with 1 alpha-hydroxycholecalciferol therapy . Postgrad Med J . 1977;53:570-573.Crossref 32. Godfrey RS, Faddes DM, Daroca PJ Jr, Sutherland CM. Tumoral calcinosis and hyperparathyroidism . J La State Med Soc . 1987;139:26-32. 33. Luflin EG, Wilson DM, Smith LH, et al. Phosphorus excretion in tumoral calcinosis: response to parathyroid hormone and azetalomide . J Clin Endocrinol Metab . 1980;50:648-653.Crossref 34. Barton DL, Reeves RJ. Tumoral calcinosis: report of 3 cases and review of literature . Ann Intern Med . 1980;92:482-487.Crossref 35. Prasad VLN, Naresh KN, Govind Krishna SR, Ananthalerishman N, Veliath AL. Tumoral calcinosis . World J Surg . 1989;13:803-808.Crossref 36. Eisenberg B, Tzamaloukas AH, Hartstone MF, Listrom MB, Arrigton ER, Sherrad DJ. Periarticular tumoral calcinosis and hypercalcemia in a hemodialysis patient without hyperparathyroidism: a case report . J Nucl Med . 1990:31:1099-1103. 37. Hacihanefioglu U. Tumoral calcinosis: a clinical and pathological study of eleven unreported cases in Turkey . J Bone Joint Surg Am . 1978;60:1131-1135. 38. Mozaffarian G, Lafferty FW, Bearson OH. Treatment of tumoral calcinosis with phosphorus deprivation . Ann Intern Med . 1972;77:741-745.Crossref 39. Kirk TS, Simon MA. Tumoral calcinosis: report of a case with successful medical management . J Bone Joint Surg Am . 1981;63:1176-1179. 40. Davies M, Clements MR, Mawer EB, Fremont AJ. Tumoral calcinosis: clinical and metabolic response to phosphorus deprivation . Q J Med . 1987;63:493-498. 41. Walentynowicz JE, Mahoney MD, Saldane MJ. Tumoral calcinosis: case report with treatment failure . Orthop Rev . 1989;18:687-690. 42. Kallmeyer JC, Seimon LP, MacSerraigh ET. The effect of thyrocalcitonin therapy and phosphate deprivation on tumoral calcinosis . S Afr Med J . 1978;54: 963-966. 43. Sonin A, Nance EP Jr. Juxtaarticular calcification in secondary hyperparathyroidism: resorption after parathyroidectomy . South Med J . 1991;84:641-643.

Journal

Archives of SurgeryAmerican Medical Association

Published: Jul 1, 1993

References