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Subepidermal Bullous Disease With a Circulating Anticoagulant

Subepidermal Bullous Disease With a Circulating Anticoagulant Abstract A case of bullous dermatitis associated with a circulating anticoagulant is presented. The course of the disease was prolonged (three years) and characterized by frequent exacerbations separated by short, partly incomplete remissions. The morphologic pattern of the cutaneous eruption was not characteristic of any of the bullous dermatoses, though it bore some resemblance to dermatitis herpetiformis. The histopathologic findings did not allow differentiation beyond the diagnosis of subepidermal bullous dermatosis. A therapeutic trial with sulfones was inconclusive while the response to large doses of systemic steroids was fair. Studies of coagulation revealed that the anticoagulant was an inhibitor of formed thromboplastin. The anticoagulant was first demonstrated before the patient received any blood transfusion. Results of subsequent coagulation studies were within normal limits. References 1. Margolius, A., Jr.; Jackson, D.P.; and Ratnoff, O.D.: Circulating Anticoagulants , Medicine 40:145-202 ( (May) ) 1961.Crossref 2. Quick, A.J., and Stefanini, M.: Activation of Plasma Thromboplastinogen and Evidence of an Inhibitor , Proc Soc Exp Biol Med 67:111-112, 1948.Crossref 3. Dieter, D.G.; Spooner, M.; and Pohle, F.J.: Studies on an Undetermined Circulating Anticoagulant: Case Report and Laboratory Findings , Blood 4:120-129 ( (Feb) ) 1949. 4. Tzanck, A.; Soulier, J.P.; and Blatrix, C.: Deux nouvelles observations de syndromes hémorrhagiques avec présence d'un anticoagulant circulant , Rev Hémat 4:502-518 ( (July) -Sept) 1949. 5. Bartorelli, C. and Vanacore, C.: Pseudoemofilia da Anticoagulante Circolante: Revisione della Letteratura e Descrizione di un caso , Policlinico (Med) 62:251-288 ( (Oct) ) 1955. 6. Benthaus, J., and Richter, W.C.: Zum Krankheitsbild der Hemmkörperhämophilie: Ein Kasuistischer Beitrag , Deutsch Arch Klin Med 203:1-9 ( (March) ) 1956. 7. Björnberg, A.: Circulating Anticoagulants and Bullous Dermatoses , Acta Dermatovener 38:251-263, 1958. 8. von Klingmüller, G.; Leinbrock, A.; and Laumanns, U.: Pemphigus Vulgaris mit Hemmkörperhämophilie , Hautarzt 7:200-206 ( (May) ) 1956. 9. Cooke, J.V.; Anderson, J.B.; and Gamble, W.S.: Circulating Factor VIII Anticoagulant in Bullous Dermatitis , Arch Int Med 110:511-515, 1962.Crossref 10. Miller, S.P., et al: Coagulation Disorders in Cancer: I. Clinical and Laboratory Studies , Cancer 20:1452-1467 ( (Sept) ) 1967.Crossref 11. Johnson, A.J., and Tse, A.O.: Fibrinolytic Determination of Plasminogen Proactivator in Human Purified Plasminogen or Biological Fluids , in Tocantins, L.M., and Kagal, L.A. (eds.): Blood Coagulation, Hemorrhage, and Thrombosis , New York: Grune & Stratton, Inc., 1964, pp 476-477. 12. Villavicensio, J.L., and Warren, R.: Clinical Determination of Fibrinolytic Activity , Arch Surg 78:639-646 ( (April) ) 1959.Crossref 13. Johnson, A.J.; Tse, A.O.; and Newman, J.: Fibrinolytic Estimation of Streptokinase (SK) Inhibitor and Antibody and Urokinase (UK) Inhibitor in Purified Systems of Biological Fluids , in Tocantins, L.M., and Kagal, L.A. (eds.): Blood Coagulation, Hemorrhage, and Thrombosis , New York: Grune & Stratton, Inc., 1964, pp 479-482. 14. Biggs, R., and Douglas, A.S.: The Thromboplastin Generation Test , J Clin Path 6:23-29 ( (Feb) ) 1953.Crossref 15. Degos, R.: Dermatologie , Paris: Flammarion, 1953, pp 413-422. 16. Degos, R: In Discussion on Bullous Dermatoses , Acta Dermatovener, Proc 11th Int Congr Derm 1957 , pp 301-305. 17. Percival, G.H.: The Relationship Between Dermatitis Herpetiformis, Pemphigoid, and Pemphigus, on the Basis of Clinical and Histological Investigation , Acta Dermatovener, Proc 11th Int Congr Derm 1957 , pp 286-292. 18. Lapiere, S.: in MacKenna: Modern Trends in Dermatology , London: Butterworth and Co., Ltd., 1966, pp 222-246. 19. Lever, W.F.: Pemphigus and Pemphigoid , Springfield, Ill: Charles C Thomas, Publisher, 1965, pp 92-99. 20. Allen, A.C.: The Skin , St. Louis: C. V. Mosby Co., Medical Publishers, 1954, p 256. 21. Rapaport, S.I.; Ames, S.B.; and Duvall, B.J.: A Plasma Coagulation Defect in Systemic Lupus Erythematosus Arising From Hypoprothrombinemia Combined with Antiprothrombinase Activity , Blood 15:212-227 ( (Feb) ) 1960. 22. Medal, L.S., and Lisker, R.: Circulating Anticoagulants in Disseminated Lupus Erythematosus , Brit J Haemat 5:284-293 ( (July) ) 1959.Crossref 23. Bonnin, J.; Cohen, A.K.; and Hicks, M.D.: Coagulation Defects in a Case of Systemic Lupus Erythematosus With Thrombocytopenia , Brit J Haemat 2:168-179 ( (April) ) 1956.Crossref 24. Ramot, B., and Singer, K.: An Unusual Circulating Anticoagulant in Systemic Lupus Erythematosus , Acta Hemat 16:158-169 ( (March) -April) 1956.Crossref 25. Swift, S.: A Serum Anticoagulant Factor in Systemic Lupus Erythematosus , Arch Derm 74:296-299 ( (Sept) ) 1956.Crossref 26. Frick, P.G.: Acquired Circulating Anticoagulants in Systemic Collagen Disease , Blood 10:691-706 ( (July) ) 1955. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

Subepidermal Bullous Disease With a Circulating Anticoagulant

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Publisher
American Medical Association
Copyright
Copyright © 1969 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archderm.1969.01610200033006
Publisher site
See Article on Publisher Site

Abstract

Abstract A case of bullous dermatitis associated with a circulating anticoagulant is presented. The course of the disease was prolonged (three years) and characterized by frequent exacerbations separated by short, partly incomplete remissions. The morphologic pattern of the cutaneous eruption was not characteristic of any of the bullous dermatoses, though it bore some resemblance to dermatitis herpetiformis. The histopathologic findings did not allow differentiation beyond the diagnosis of subepidermal bullous dermatosis. A therapeutic trial with sulfones was inconclusive while the response to large doses of systemic steroids was fair. Studies of coagulation revealed that the anticoagulant was an inhibitor of formed thromboplastin. The anticoagulant was first demonstrated before the patient received any blood transfusion. Results of subsequent coagulation studies were within normal limits. References 1. Margolius, A., Jr.; Jackson, D.P.; and Ratnoff, O.D.: Circulating Anticoagulants , Medicine 40:145-202 ( (May) ) 1961.Crossref 2. Quick, A.J., and Stefanini, M.: Activation of Plasma Thromboplastinogen and Evidence of an Inhibitor , Proc Soc Exp Biol Med 67:111-112, 1948.Crossref 3. Dieter, D.G.; Spooner, M.; and Pohle, F.J.: Studies on an Undetermined Circulating Anticoagulant: Case Report and Laboratory Findings , Blood 4:120-129 ( (Feb) ) 1949. 4. Tzanck, A.; Soulier, J.P.; and Blatrix, C.: Deux nouvelles observations de syndromes hémorrhagiques avec présence d'un anticoagulant circulant , Rev Hémat 4:502-518 ( (July) -Sept) 1949. 5. Bartorelli, C. and Vanacore, C.: Pseudoemofilia da Anticoagulante Circolante: Revisione della Letteratura e Descrizione di un caso , Policlinico (Med) 62:251-288 ( (Oct) ) 1955. 6. Benthaus, J., and Richter, W.C.: Zum Krankheitsbild der Hemmkörperhämophilie: Ein Kasuistischer Beitrag , Deutsch Arch Klin Med 203:1-9 ( (March) ) 1956. 7. Björnberg, A.: Circulating Anticoagulants and Bullous Dermatoses , Acta Dermatovener 38:251-263, 1958. 8. von Klingmüller, G.; Leinbrock, A.; and Laumanns, U.: Pemphigus Vulgaris mit Hemmkörperhämophilie , Hautarzt 7:200-206 ( (May) ) 1956. 9. Cooke, J.V.; Anderson, J.B.; and Gamble, W.S.: Circulating Factor VIII Anticoagulant in Bullous Dermatitis , Arch Int Med 110:511-515, 1962.Crossref 10. Miller, S.P., et al: Coagulation Disorders in Cancer: I. Clinical and Laboratory Studies , Cancer 20:1452-1467 ( (Sept) ) 1967.Crossref 11. Johnson, A.J., and Tse, A.O.: Fibrinolytic Determination of Plasminogen Proactivator in Human Purified Plasminogen or Biological Fluids , in Tocantins, L.M., and Kagal, L.A. (eds.): Blood Coagulation, Hemorrhage, and Thrombosis , New York: Grune & Stratton, Inc., 1964, pp 476-477. 12. Villavicensio, J.L., and Warren, R.: Clinical Determination of Fibrinolytic Activity , Arch Surg 78:639-646 ( (April) ) 1959.Crossref 13. Johnson, A.J.; Tse, A.O.; and Newman, J.: Fibrinolytic Estimation of Streptokinase (SK) Inhibitor and Antibody and Urokinase (UK) Inhibitor in Purified Systems of Biological Fluids , in Tocantins, L.M., and Kagal, L.A. (eds.): Blood Coagulation, Hemorrhage, and Thrombosis , New York: Grune & Stratton, Inc., 1964, pp 479-482. 14. Biggs, R., and Douglas, A.S.: The Thromboplastin Generation Test , J Clin Path 6:23-29 ( (Feb) ) 1953.Crossref 15. Degos, R.: Dermatologie , Paris: Flammarion, 1953, pp 413-422. 16. Degos, R: In Discussion on Bullous Dermatoses , Acta Dermatovener, Proc 11th Int Congr Derm 1957 , pp 301-305. 17. Percival, G.H.: The Relationship Between Dermatitis Herpetiformis, Pemphigoid, and Pemphigus, on the Basis of Clinical and Histological Investigation , Acta Dermatovener, Proc 11th Int Congr Derm 1957 , pp 286-292. 18. Lapiere, S.: in MacKenna: Modern Trends in Dermatology , London: Butterworth and Co., Ltd., 1966, pp 222-246. 19. Lever, W.F.: Pemphigus and Pemphigoid , Springfield, Ill: Charles C Thomas, Publisher, 1965, pp 92-99. 20. Allen, A.C.: The Skin , St. Louis: C. V. Mosby Co., Medical Publishers, 1954, p 256. 21. Rapaport, S.I.; Ames, S.B.; and Duvall, B.J.: A Plasma Coagulation Defect in Systemic Lupus Erythematosus Arising From Hypoprothrombinemia Combined with Antiprothrombinase Activity , Blood 15:212-227 ( (Feb) ) 1960. 22. Medal, L.S., and Lisker, R.: Circulating Anticoagulants in Disseminated Lupus Erythematosus , Brit J Haemat 5:284-293 ( (July) ) 1959.Crossref 23. Bonnin, J.; Cohen, A.K.; and Hicks, M.D.: Coagulation Defects in a Case of Systemic Lupus Erythematosus With Thrombocytopenia , Brit J Haemat 2:168-179 ( (April) ) 1956.Crossref 24. Ramot, B., and Singer, K.: An Unusual Circulating Anticoagulant in Systemic Lupus Erythematosus , Acta Hemat 16:158-169 ( (March) -April) 1956.Crossref 25. Swift, S.: A Serum Anticoagulant Factor in Systemic Lupus Erythematosus , Arch Derm 74:296-299 ( (Sept) ) 1956.Crossref 26. Frick, P.G.: Acquired Circulating Anticoagulants in Systemic Collagen Disease , Blood 10:691-706 ( (July) ) 1955.

Journal

Archives of DermatologyAmerican Medical Association

Published: Feb 1, 1969

References