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Plasmapheresis in Idiopathic Inflammatory Myopathy: Experience With 35 Patients

Plasmapheresis in Idiopathic Inflammatory Myopathy: Experience With 35 Patients Abstract • Thirty-five patients with idiopathic inflammatory myopathy (IM) were treated with plasmapheresis. They had had inadequate clinical responses to prednisone or to prednisone and cytotoxic immunosuppressive drug therapy. All patients received cyclophosphamide or chlorambucil in addition to plasmapheresis; 33 also received prednisone. Strength increased in 32 patients during combined therapy; patients with the most active disease experienced substantial improvement that approached clinical remission in some cases. Clinical improvement was documented by muscle testing, forced vital capacity, electromyography, muscle biopsy, and serum creatine phosphokinase activity. The major side effect of treatment was herpes zoster. Primary biliary cirrhosis improved in one patient. Although the effects of plasmapheresis cannot be dissociated from those of the concomitantly administered immunosuppressive drugs, the improvement seen in patients with active disease suggests that plasmapheresis with immunosuppressive drug therapy may find a place in their treatment and implicates circulating factors in the pathogenesis of IM. References 1. Eaton LM: The perspective of neurology in regard to polymyositis . Neurology 1954;4:245-263.Crossref 2. Barnes BE: Dermatomyositis and malignancy: A review of the literature . Ann Intern Med 1976;84:68-76.Crossref 3. Medsger TA, Rodnan GP, Moossy J, et al: Skeletal muscle involvement in progressive systemic sclerosis (scleroderma) . Arthritis Rheum 1968;11:554-568.Crossref 4. Brooke MH, Kaplan H: Muscle pathology in rheumatoid arthritis, polymyalgia rheumatica and polymyositis . Arch Pathol 1972;94:101-118. 5. Erbslöh F, Baedeker WD: Lupusmyopathie . Dtsch Med Wochenschr 1962;87:2464-2470.Crossref 6. Sharp GC, Irvin WS, Tan EM, et al: Mixed connective tissue disease: An apparently distinct rheumatic disease syndrome associated with a specific antibody to an extractable nuclear antigen (ENA) . Am J Med 1972;52:148-159.Crossref 7. Oxenhandler R, Hart M, Corman L, et al: Pathology of skeletal muscle in mixed connective tissue disease . Arthritis Rheum 1977;20:985-988.Crossref 8. Adams RD: Diseases of Muscle: A Study in Pathology , ed 3. Hagerstown, Md, Harper & Row Publishers Inc, 1975. 9. DeVere R, Bradley WG: Polymyositis: Its presentation, morbidity and mortality . Brain 1975;98:637-666.Crossref 10. Bohan A, Peter JB, Bowman RL, et al: A computer-assisted analysis of 153 patients with polymyositis and dermatomyositis . Medicine 1977;56:255-286.Crossref 11. Baumli HP, Mumenthaler M: The perifascicular atrophy factor: An aid in the histological diagnosis of polymyositis . J Neurol 1977;214:129-136.Crossref 12. Pickens P: Myositis and collagen disease: A muscle biopsy study . Mt Sinai J Med 1978;45:433-442. 13. Vilppula A: Muscular disorders in some collagen diseases . Acta Med Scand Suppl 1972;540:1-47. 14. Boylan RC, Sokoloff L: Vascular lesions in dermatomyositis . Arthritis Rheum 1960;3:379-386.Crossref 15. Banker BW, Victor M: Dermatomyositis (systemic angiopathy) of childhood . Medicine 1966;45:261-289.Crossref 16. Banker BQ: Dermatomyositis of childhood: Ultrastructural alterations of muscle and intramuscular blood vessels . J Neuropathol Exp Neurol 1975;34:46-75.Crossref 17. Carpenter S, Karpati G, Rothman S, et al: The childhood type of dermatomyositis . Neurology 1976;26:952-962.Crossref 18. Gonzalez-Angulo A, Fraga A, Mintz G, et al: Submicroscopic alterations in capillaries of skeletal muscles in polymyositis . Am J Med 1968;45:873-879.Crossref 19. Norton WL, Hurd ER, Lewis DC, et al: Evidence of microvascular injury in scleroderma and systemic lupus erythematosus: Quantitative study of the microvascular bed . J Lab Clin Med 1968;71:919-933. 20. Ringel SP, Thorne EG, Phanuphak P, et al: Immune complex vasculitis, polymyositis and hyperglobulinemic purpura . Neurology 1979;29:p-689. 21. Penn AS: Myasthenia gravis, dermatomyositis, and polymyositis: Immunopathological diseases , in Griggs RC, Moxley RT (eds): Advances in Neurology . New York, Raven Press, 1977, vol 17, pp 41-61. 22. Rossen RD, Hersh EM, Sharp JT, et al: Effect of plasma exchange on circulating immune complexes and antibody formation in patients treated with cyclophosphamide and prednisone . Am J Med 1977;63:674-682.Crossref 23. Birdsall HH, Brewer EJ, Rossen RD, et al: Clinical improvement associated with hypocomplementemia following plasmapheresis , in Dau PC (ed): Plasmapheresis and the Immunobiology of Myasthenia Gravis . Boston, Houghton Mifflin Co, 1979, pp 191-198. 24. Dau PC, Lindstrom JM, Cassel CK, et al: Plasmapheresis in myasthenia gravis and polymyositis , in Dau PC (ed): Plasmapheresis and the Immunobiology of Myasthenia Gravis . Boston, Houghton Mifflin Co, 1979, pp 229-247. 25. Brewer EJ, Giannini EH, Rossen RD, et al: Plasma exchange therapy of a childhood onset dermatomyositis patient . Arthritis Rheum 1980;23:509-513.Crossref 26. Dau PC, Lindstrom JM, Denys EH: Plasmapheresis in neurologic disorders , in Taswell HF, Pineda AA (eds): Therapeutic Plasma and Cytapheresis . Division of Blood Diseases and Resources, National Heart, Lung, and Blood Institute, to be published. 27. Dau PC, Bennington JL: Plasmapheresis in childhood dermatomyositis . J Pediatr 1981;98:237-240.Crossref 28. Menke AM, Dau PC: Technical notes on plasmapheresis , in Dau PC (ed): Plasmapheresis and the Immunobiology of Myasthenia Gravis . Boston, Houghton Mifflin Co, 1979, pp 351-358. 29. Hardin JA, Walker LC, Steere PH, et al: Circulating immune complexes in Lyme arthritis . J Clin Invest 1979;63:468-477.Crossref 30. Zubler RH, Lange G, Lambert PH, et al: Detection of immune complexes in unheated sera by a modified 125 I-Clq binding assay . J Immumil 1976;116:232-235. 31. Williams V, Gershon A, Brunell PA: Serological response to varicella-zoster membrane antigens measured by indirect immunofluorescence . J Infect Dis 1974;130:669-672.Crossref 32. Dau PC, Kahaleh MD, Sagebiel RW: Plasmapheresis and immunosuppressive drug therapy in scleroderma . Arthritis Rheum , to be published. 33. Robinson DR: Polymyositis , in Rubenstein E, Federman DD (eds): Scientific American Medicine . New York, Scientific American Inc, 1979, chap 15, sec VI, pp 1-7. 34. Bunch TW, Worthington JW, Combs JJ, et al: Azathioprine with prednisone for polymyositis: A controlled, clinical trial . Ann Intern Med 1980;92:365-369.Crossref 35. Niakan E, Pitner SE, Whitaker JN, et al: Immunosuppressive agents in corticorefractory childhood dermatomyositis . Neurology 1980;30:286-291.Crossref 36. Fries JF, Sharp GC, McDevitt HO, et al: Cyclophosphamide therapy in systemic lupus erythematosus and polymyositis . Arthritis Rheum 1973;16:154-162.Crossref 37. Sturgill BC, Worzniak MJ: Stimulation of proliferation of 19S antibody-forming cells in the spleens of immunized guinea pigs after exchange transfusion . Nature 1970;228:1304-1305.Crossref 38. Terman DS, Garcia-Rinaldi B, Dannerman D, et al: Specific suppression of antibody rebound after extracorporeal immunoabsorption: I. Comparison of single vs combination chemotherapeutic agents . Clin Exp Immunol 1978;34:32-41. 39. Brystryn J-C, Schenkein I, Uhr JW: A model for the regulation of antibody synthesis by serum antibody , in Amos B (ed): Progress in Immunology I . New York, Academic Press Inc, 1971, pp 628-637. 40. Dau PC: Plasmapheresis therapy in myasthenia gravis: A review . Muscle Nerve 1980;3:468-482.Crossref 41. Whitaker JN, Engel WK: Vascular deposits of immunoglobulin and complement in idiopathic inflammatory myopathy . N Engl J Med 1972;286:333-338.Crossref 42. Heffner RR, Barron SA, Jenis EH, et al: Skeletal muscle in polymyositis: Immunohistochemical study . Arch Pathol Lab Med 1979;103:310-313. 43. Behan WM, Behan PO: Complement abnormalities in polymyositis . J Neurol Sci 1977;34:241-246.Crossref 44. Von Kiraly K, Horvath A, Daroczy J, et al: Immunologische Aspekte bei der Dermatomyositis . Allerg Immunol 1972;18:141-160. 45. Oxenhandler R, Adelstein EH, Hart MN: Immunopathy of skeletal muscle: The value of direct immunofluorescence in the diagnosis of connective tissue disease . Hum Pathol 1977;8:321-328.Crossref 46. Klug H, Barthelmes H, Thormann T: Immunomorphologische and ultrastrukturelle Befunde an der Muskulatur bei progressiver Sklerodermie . Z Gesamte Inn Med 1977;32:357-363. 47. Judge DM, McGlynn TJ, Abt AB, et al: Immunologic myopathy: Linear IgG deposition and fulminant terminal episode . Arch Pathol Lab Med 1977;101:362-365. 48. Sokal JE, First D: Varicella-zoster infection in Hodgskin's disease . Am J Med 1965;39:452-463.Crossref 49. Goffinet DR, Glatstein EJ, Merigan TC: Herpes zoster-varicella infections and lymphoma . Ann Intern Med 1972;76:235-240.Crossref 50. Gallagher JG, Merigan TC: Prolonged herpes-zoster infection associated with immuno-suppressive therapy . Ann Intern Med 1979;91:842-846.Crossref 51. Schimpff S, Serpick A, Stoler B, et al: Varicella-zoster infection in patients with cancer . Ann Intern Med 1972;76:241-254.Crossref 52. Thomas HC, Potter BJ, Sherlock S: Is primary biliary cirrhosis an immune complex disease? Lancet 1977;2:1261-1263.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Neurology American Medical Association

Plasmapheresis in Idiopathic Inflammatory Myopathy: Experience With 35 Patients

Archives of Neurology , Volume 38 (9) – Sep 1, 1981

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American Medical Association
Copyright
Copyright © 1981 American Medical Association. All Rights Reserved.
ISSN
0003-9942
eISSN
1538-3687
DOI
10.1001/archneur.1981.00510090038003
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Abstract

Abstract • Thirty-five patients with idiopathic inflammatory myopathy (IM) were treated with plasmapheresis. They had had inadequate clinical responses to prednisone or to prednisone and cytotoxic immunosuppressive drug therapy. All patients received cyclophosphamide or chlorambucil in addition to plasmapheresis; 33 also received prednisone. Strength increased in 32 patients during combined therapy; patients with the most active disease experienced substantial improvement that approached clinical remission in some cases. Clinical improvement was documented by muscle testing, forced vital capacity, electromyography, muscle biopsy, and serum creatine phosphokinase activity. The major side effect of treatment was herpes zoster. Primary biliary cirrhosis improved in one patient. Although the effects of plasmapheresis cannot be dissociated from those of the concomitantly administered immunosuppressive drugs, the improvement seen in patients with active disease suggests that plasmapheresis with immunosuppressive drug therapy may find a place in their treatment and implicates circulating factors in the pathogenesis of IM. References 1. Eaton LM: The perspective of neurology in regard to polymyositis . Neurology 1954;4:245-263.Crossref 2. Barnes BE: Dermatomyositis and malignancy: A review of the literature . Ann Intern Med 1976;84:68-76.Crossref 3. Medsger TA, Rodnan GP, Moossy J, et al: Skeletal muscle involvement in progressive systemic sclerosis (scleroderma) . Arthritis Rheum 1968;11:554-568.Crossref 4. Brooke MH, Kaplan H: Muscle pathology in rheumatoid arthritis, polymyalgia rheumatica and polymyositis . Arch Pathol 1972;94:101-118. 5. Erbslöh F, Baedeker WD: Lupusmyopathie . Dtsch Med Wochenschr 1962;87:2464-2470.Crossref 6. Sharp GC, Irvin WS, Tan EM, et al: Mixed connective tissue disease: An apparently distinct rheumatic disease syndrome associated with a specific antibody to an extractable nuclear antigen (ENA) . Am J Med 1972;52:148-159.Crossref 7. Oxenhandler R, Hart M, Corman L, et al: Pathology of skeletal muscle in mixed connective tissue disease . Arthritis Rheum 1977;20:985-988.Crossref 8. Adams RD: Diseases of Muscle: A Study in Pathology , ed 3. Hagerstown, Md, Harper & Row Publishers Inc, 1975. 9. DeVere R, Bradley WG: Polymyositis: Its presentation, morbidity and mortality . Brain 1975;98:637-666.Crossref 10. Bohan A, Peter JB, Bowman RL, et al: A computer-assisted analysis of 153 patients with polymyositis and dermatomyositis . Medicine 1977;56:255-286.Crossref 11. Baumli HP, Mumenthaler M: The perifascicular atrophy factor: An aid in the histological diagnosis of polymyositis . J Neurol 1977;214:129-136.Crossref 12. Pickens P: Myositis and collagen disease: A muscle biopsy study . Mt Sinai J Med 1978;45:433-442. 13. Vilppula A: Muscular disorders in some collagen diseases . Acta Med Scand Suppl 1972;540:1-47. 14. Boylan RC, Sokoloff L: Vascular lesions in dermatomyositis . Arthritis Rheum 1960;3:379-386.Crossref 15. Banker BW, Victor M: Dermatomyositis (systemic angiopathy) of childhood . Medicine 1966;45:261-289.Crossref 16. Banker BQ: Dermatomyositis of childhood: Ultrastructural alterations of muscle and intramuscular blood vessels . J Neuropathol Exp Neurol 1975;34:46-75.Crossref 17. Carpenter S, Karpati G, Rothman S, et al: The childhood type of dermatomyositis . Neurology 1976;26:952-962.Crossref 18. Gonzalez-Angulo A, Fraga A, Mintz G, et al: Submicroscopic alterations in capillaries of skeletal muscles in polymyositis . Am J Med 1968;45:873-879.Crossref 19. Norton WL, Hurd ER, Lewis DC, et al: Evidence of microvascular injury in scleroderma and systemic lupus erythematosus: Quantitative study of the microvascular bed . J Lab Clin Med 1968;71:919-933. 20. Ringel SP, Thorne EG, Phanuphak P, et al: Immune complex vasculitis, polymyositis and hyperglobulinemic purpura . Neurology 1979;29:p-689. 21. Penn AS: Myasthenia gravis, dermatomyositis, and polymyositis: Immunopathological diseases , in Griggs RC, Moxley RT (eds): Advances in Neurology . New York, Raven Press, 1977, vol 17, pp 41-61. 22. Rossen RD, Hersh EM, Sharp JT, et al: Effect of plasma exchange on circulating immune complexes and antibody formation in patients treated with cyclophosphamide and prednisone . Am J Med 1977;63:674-682.Crossref 23. Birdsall HH, Brewer EJ, Rossen RD, et al: Clinical improvement associated with hypocomplementemia following plasmapheresis , in Dau PC (ed): Plasmapheresis and the Immunobiology of Myasthenia Gravis . Boston, Houghton Mifflin Co, 1979, pp 191-198. 24. Dau PC, Lindstrom JM, Cassel CK, et al: Plasmapheresis in myasthenia gravis and polymyositis , in Dau PC (ed): Plasmapheresis and the Immunobiology of Myasthenia Gravis . Boston, Houghton Mifflin Co, 1979, pp 229-247. 25. Brewer EJ, Giannini EH, Rossen RD, et al: Plasma exchange therapy of a childhood onset dermatomyositis patient . Arthritis Rheum 1980;23:509-513.Crossref 26. Dau PC, Lindstrom JM, Denys EH: Plasmapheresis in neurologic disorders , in Taswell HF, Pineda AA (eds): Therapeutic Plasma and Cytapheresis . Division of Blood Diseases and Resources, National Heart, Lung, and Blood Institute, to be published. 27. Dau PC, Bennington JL: Plasmapheresis in childhood dermatomyositis . J Pediatr 1981;98:237-240.Crossref 28. Menke AM, Dau PC: Technical notes on plasmapheresis , in Dau PC (ed): Plasmapheresis and the Immunobiology of Myasthenia Gravis . Boston, Houghton Mifflin Co, 1979, pp 351-358. 29. Hardin JA, Walker LC, Steere PH, et al: Circulating immune complexes in Lyme arthritis . J Clin Invest 1979;63:468-477.Crossref 30. Zubler RH, Lange G, Lambert PH, et al: Detection of immune complexes in unheated sera by a modified 125 I-Clq binding assay . J Immumil 1976;116:232-235. 31. Williams V, Gershon A, Brunell PA: Serological response to varicella-zoster membrane antigens measured by indirect immunofluorescence . J Infect Dis 1974;130:669-672.Crossref 32. Dau PC, Kahaleh MD, Sagebiel RW: Plasmapheresis and immunosuppressive drug therapy in scleroderma . Arthritis Rheum , to be published. 33. Robinson DR: Polymyositis , in Rubenstein E, Federman DD (eds): Scientific American Medicine . New York, Scientific American Inc, 1979, chap 15, sec VI, pp 1-7. 34. Bunch TW, Worthington JW, Combs JJ, et al: Azathioprine with prednisone for polymyositis: A controlled, clinical trial . Ann Intern Med 1980;92:365-369.Crossref 35. Niakan E, Pitner SE, Whitaker JN, et al: Immunosuppressive agents in corticorefractory childhood dermatomyositis . Neurology 1980;30:286-291.Crossref 36. Fries JF, Sharp GC, McDevitt HO, et al: Cyclophosphamide therapy in systemic lupus erythematosus and polymyositis . Arthritis Rheum 1973;16:154-162.Crossref 37. Sturgill BC, Worzniak MJ: Stimulation of proliferation of 19S antibody-forming cells in the spleens of immunized guinea pigs after exchange transfusion . Nature 1970;228:1304-1305.Crossref 38. Terman DS, Garcia-Rinaldi B, Dannerman D, et al: Specific suppression of antibody rebound after extracorporeal immunoabsorption: I. Comparison of single vs combination chemotherapeutic agents . Clin Exp Immunol 1978;34:32-41. 39. Brystryn J-C, Schenkein I, Uhr JW: A model for the regulation of antibody synthesis by serum antibody , in Amos B (ed): Progress in Immunology I . New York, Academic Press Inc, 1971, pp 628-637. 40. Dau PC: Plasmapheresis therapy in myasthenia gravis: A review . Muscle Nerve 1980;3:468-482.Crossref 41. Whitaker JN, Engel WK: Vascular deposits of immunoglobulin and complement in idiopathic inflammatory myopathy . N Engl J Med 1972;286:333-338.Crossref 42. Heffner RR, Barron SA, Jenis EH, et al: Skeletal muscle in polymyositis: Immunohistochemical study . Arch Pathol Lab Med 1979;103:310-313. 43. Behan WM, Behan PO: Complement abnormalities in polymyositis . J Neurol Sci 1977;34:241-246.Crossref 44. Von Kiraly K, Horvath A, Daroczy J, et al: Immunologische Aspekte bei der Dermatomyositis . Allerg Immunol 1972;18:141-160. 45. Oxenhandler R, Adelstein EH, Hart MN: Immunopathy of skeletal muscle: The value of direct immunofluorescence in the diagnosis of connective tissue disease . Hum Pathol 1977;8:321-328.Crossref 46. Klug H, Barthelmes H, Thormann T: Immunomorphologische and ultrastrukturelle Befunde an der Muskulatur bei progressiver Sklerodermie . Z Gesamte Inn Med 1977;32:357-363. 47. Judge DM, McGlynn TJ, Abt AB, et al: Immunologic myopathy: Linear IgG deposition and fulminant terminal episode . Arch Pathol Lab Med 1977;101:362-365. 48. Sokal JE, First D: Varicella-zoster infection in Hodgskin's disease . Am J Med 1965;39:452-463.Crossref 49. Goffinet DR, Glatstein EJ, Merigan TC: Herpes zoster-varicella infections and lymphoma . Ann Intern Med 1972;76:235-240.Crossref 50. Gallagher JG, Merigan TC: Prolonged herpes-zoster infection associated with immuno-suppressive therapy . Ann Intern Med 1979;91:842-846.Crossref 51. Schimpff S, Serpick A, Stoler B, et al: Varicella-zoster infection in patients with cancer . Ann Intern Med 1972;76:241-254.Crossref 52. Thomas HC, Potter BJ, Sherlock S: Is primary biliary cirrhosis an immune complex disease? Lancet 1977;2:1261-1263.Crossref

Journal

Archives of NeurologyAmerican Medical Association

Published: Sep 1, 1981

References