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Atypical Neutrophilic Dermatosis With Subcorneal IgA Deposits

Atypical Neutrophilic Dermatosis With Subcorneal IgA Deposits Abstract • A 26-year-old woman had a chronic vesiculopustular and ulcerating skin disease associated with fever and arthritis. Cutaneous biopsy specimens showed an extensive infiltration of the dermis and epidermis by neutrophils. Direct immunofluorescence (IF) revealed linear subcorneal IgA deposits. Indirect IF showed IgA antibodies reactive with the subcorneal zone of normal epidermis. The disease responded to dapsone therapy. The association between neutrophilic dermatoses, including pyoderma gangrenosum, subcorneal pustular dermatosis, and related entities, and IgA involvement, either IgA gammopathies and/or intraepidermal IgA deposits, is emphasized. Intraepidermal IgA deposits are possibly involved in the pathogenesis of our patient's condition and of other cases of unusual neutrophilic dermatoses. (Arch Dermatol 1987;123:790-795) References 1. Katz SI: Blistering skin diseases: New insights . NEngl JMed 1985;313:1657-1658.Crossref 2. Beutner EH, Chorzelski TP, Bean SF: Immunopathology of the Skin , ed 2. New York, John Wiley & Sons Inc, 1979. 3. Perry HO, Winkelmann RK: Bullous pyoderma gangrenosum and leukemia . Arch Dermatol 1972;106:901-905.Crossref 4. Horton JJ, Trounce JR, MacDonald DM: Bullous pyoderma gangrenosum and multiple myeloma . Br J Dermatol 1984;111: 227-230.Crossref 5. Ackerman AB: Histologic Diagnosis of Inflammatory Skin Diseases . Philadelphia, Lea & Febiger, 1978. 6. Marsden JR, Millard LG: Pyoderma gangrenosum, subcorneal pustular dermatosis and IgA paraproteinemia . Br JDermatol 1986;114:125-129.Crossref 7. Wolff K, Stingl G: Pyoderma gangrenosum , in Fitzpatrick TB, Eisen AZ, Wolff K, et al (eds): Update: Dermatology in General Medicine . New York, McGraw-Hill International Book Co, 1983, pp 174-183. 8. Russell Jones R: Cutaneous manifestations of paraproteinemia: Part II . Br J Dermatol 1981;104:209-220.Crossref 9. Powell FC, Schroeter AL, Su WPD, et al: Pyoderma gangrenosum and monoclonal gammopathy . Arch Dermatol 1983;119:468-472.Crossref 10. Perry HO, Brunsting LA: Pyoderma gangrenosum . Arch Dermatol 1957;75:380-386.Crossref 11. Ayres S Jr, Ayres S III: Pyoderma gangrenosum with an unusual syndrome of ulcers, vesicles, and arthritis . Arch Dermatol 1958;77:269-280.Crossref 12. Wolff K: Subkorneale pustulöse Dermatose (Sneddon-Wilkinson): Pyoderma Gangrenosum mit IgA-paraproteinämie . Dermatol Monatsschr 1971;157:842. 13. Venning VA, Ryan TJ: Subcorneal pustular dermatosis followed by pyoderma gangrenosum . Br J Dermatol 1986;115:117-118.Crossref 14. Andersen BL: Pyoderma gangrenosum associated with transient acantholytic dermatosis (pemphigus erythematosus-like) and paraproteinemia . Acta Derm Venereol 1981;61:77-79. 15. Sneddon IB, Wilkinson DS: Subcorneal pustular dermatosis . Br J Dermatol 1956;68:385-394.Crossref 16. Wallach D: Pustuloses sous-cornées et gammapathies monoclonales . Ann Med Interne 1984;135:672-676. 17. Sneddon IB, Wilkinson DS: Subcorneal pustular dermatosis . Br J Dermatol 1979;100:61-68.Crossref 18. Wallach D, Cottenot F, Pelbois G, et al: Subcorneal pustular dermatosis and monoclonal IgA . Br J Dermatol 1982;107:229-234.Crossref 19. Wallach D, Foldes C, Cottenot F: Pustulose sous-cornée, acantholyse superficielle et IgA monoclonale . Ann Dermatol Venereol 1982;109:959-963. 20. Tagami H, Iwatsuki K, Iwase Y, et al: Subcorneal pustular dermatosis with vesiculo-bullous eruption: Demonstration of subcorneal IgA deposits and a leukocyte chemotactic factor . Br J Dermatol 1983;109:581-587.Crossref 21. Burrows D, Bingham EA: Subcorneal pustular dermatosis and IgA gammopathy . Br J Dermatol 1984;111( (suppl 26) ):91-93.Crossref 22. Huff JC, Golitz LE, Kunke KS: Intraepidermal neutrophilic IgA dermatosis . N Engl J Med 1985;313:1643-1645.Crossref 23. Caughman W, Stern R, Haynes H: Neutrophilic dermatosis of myeloproliferative disorders . J Am Acad Dermatol 1983;9: 751-758.Crossref 24. Lawrence DA, Weigle WO, Spiegelberg HL: Immunoglobulins cytophilic for human lymphocytes, monocytes and neutrophils . J Clin Invest 1975;55:368-376.Crossref 25. Fanger MW, Shen L, Pugh H, et al: Subpopulations of human peripheral granulocytes and monocytes express receptors for IgA . Proc Natl Acad Sci USA 1980;77:3640-3644.Crossref 26. Van Epps DE, Williams RC Jr: Suppression of leucocyte chemotaxis by human IgA myeloma components . J Exp Med 1976; 144:1227-1242.Crossref 27. Schröder JM, Szeperalski B, Koh CJ, et al: IgA-associated inhibition of polymorphonuclear leukocyte chemotaxis in neutrophilic dermatoses . J Invest Dermatol 1981;77:464-468.Crossref 28. Stendahl O, Molin L, Dahlgren C: The inhibition of polymorphonuclear leukocyte cytotoxicity by dapsone . J Clin Invest 1978; 62:214-220.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

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Publisher
American Medical Association
Copyright
Copyright © 1987 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archderm.1987.01660300112023
Publisher site
See Article on Publisher Site

Abstract

Abstract • A 26-year-old woman had a chronic vesiculopustular and ulcerating skin disease associated with fever and arthritis. Cutaneous biopsy specimens showed an extensive infiltration of the dermis and epidermis by neutrophils. Direct immunofluorescence (IF) revealed linear subcorneal IgA deposits. Indirect IF showed IgA antibodies reactive with the subcorneal zone of normal epidermis. The disease responded to dapsone therapy. The association between neutrophilic dermatoses, including pyoderma gangrenosum, subcorneal pustular dermatosis, and related entities, and IgA involvement, either IgA gammopathies and/or intraepidermal IgA deposits, is emphasized. Intraepidermal IgA deposits are possibly involved in the pathogenesis of our patient's condition and of other cases of unusual neutrophilic dermatoses. (Arch Dermatol 1987;123:790-795) References 1. Katz SI: Blistering skin diseases: New insights . NEngl JMed 1985;313:1657-1658.Crossref 2. Beutner EH, Chorzelski TP, Bean SF: Immunopathology of the Skin , ed 2. New York, John Wiley & Sons Inc, 1979. 3. Perry HO, Winkelmann RK: Bullous pyoderma gangrenosum and leukemia . Arch Dermatol 1972;106:901-905.Crossref 4. Horton JJ, Trounce JR, MacDonald DM: Bullous pyoderma gangrenosum and multiple myeloma . Br J Dermatol 1984;111: 227-230.Crossref 5. Ackerman AB: Histologic Diagnosis of Inflammatory Skin Diseases . Philadelphia, Lea & Febiger, 1978. 6. Marsden JR, Millard LG: Pyoderma gangrenosum, subcorneal pustular dermatosis and IgA paraproteinemia . Br JDermatol 1986;114:125-129.Crossref 7. Wolff K, Stingl G: Pyoderma gangrenosum , in Fitzpatrick TB, Eisen AZ, Wolff K, et al (eds): Update: Dermatology in General Medicine . New York, McGraw-Hill International Book Co, 1983, pp 174-183. 8. Russell Jones R: Cutaneous manifestations of paraproteinemia: Part II . Br J Dermatol 1981;104:209-220.Crossref 9. Powell FC, Schroeter AL, Su WPD, et al: Pyoderma gangrenosum and monoclonal gammopathy . Arch Dermatol 1983;119:468-472.Crossref 10. Perry HO, Brunsting LA: Pyoderma gangrenosum . Arch Dermatol 1957;75:380-386.Crossref 11. Ayres S Jr, Ayres S III: Pyoderma gangrenosum with an unusual syndrome of ulcers, vesicles, and arthritis . Arch Dermatol 1958;77:269-280.Crossref 12. Wolff K: Subkorneale pustulöse Dermatose (Sneddon-Wilkinson): Pyoderma Gangrenosum mit IgA-paraproteinämie . Dermatol Monatsschr 1971;157:842. 13. Venning VA, Ryan TJ: Subcorneal pustular dermatosis followed by pyoderma gangrenosum . Br J Dermatol 1986;115:117-118.Crossref 14. Andersen BL: Pyoderma gangrenosum associated with transient acantholytic dermatosis (pemphigus erythematosus-like) and paraproteinemia . Acta Derm Venereol 1981;61:77-79. 15. Sneddon IB, Wilkinson DS: Subcorneal pustular dermatosis . Br J Dermatol 1956;68:385-394.Crossref 16. Wallach D: Pustuloses sous-cornées et gammapathies monoclonales . Ann Med Interne 1984;135:672-676. 17. Sneddon IB, Wilkinson DS: Subcorneal pustular dermatosis . Br J Dermatol 1979;100:61-68.Crossref 18. Wallach D, Cottenot F, Pelbois G, et al: Subcorneal pustular dermatosis and monoclonal IgA . Br J Dermatol 1982;107:229-234.Crossref 19. Wallach D, Foldes C, Cottenot F: Pustulose sous-cornée, acantholyse superficielle et IgA monoclonale . Ann Dermatol Venereol 1982;109:959-963. 20. Tagami H, Iwatsuki K, Iwase Y, et al: Subcorneal pustular dermatosis with vesiculo-bullous eruption: Demonstration of subcorneal IgA deposits and a leukocyte chemotactic factor . Br J Dermatol 1983;109:581-587.Crossref 21. Burrows D, Bingham EA: Subcorneal pustular dermatosis and IgA gammopathy . Br J Dermatol 1984;111( (suppl 26) ):91-93.Crossref 22. Huff JC, Golitz LE, Kunke KS: Intraepidermal neutrophilic IgA dermatosis . N Engl J Med 1985;313:1643-1645.Crossref 23. Caughman W, Stern R, Haynes H: Neutrophilic dermatosis of myeloproliferative disorders . J Am Acad Dermatol 1983;9: 751-758.Crossref 24. Lawrence DA, Weigle WO, Spiegelberg HL: Immunoglobulins cytophilic for human lymphocytes, monocytes and neutrophils . J Clin Invest 1975;55:368-376.Crossref 25. Fanger MW, Shen L, Pugh H, et al: Subpopulations of human peripheral granulocytes and monocytes express receptors for IgA . Proc Natl Acad Sci USA 1980;77:3640-3644.Crossref 26. Van Epps DE, Williams RC Jr: Suppression of leucocyte chemotaxis by human IgA myeloma components . J Exp Med 1976; 144:1227-1242.Crossref 27. Schröder JM, Szeperalski B, Koh CJ, et al: IgA-associated inhibition of polymorphonuclear leukocyte chemotaxis in neutrophilic dermatoses . J Invest Dermatol 1981;77:464-468.Crossref 28. Stendahl O, Molin L, Dahlgren C: The inhibition of polymorphonuclear leukocyte cytotoxicity by dapsone . J Clin Invest 1978; 62:214-220.Crossref

Journal

Archives of DermatologyAmerican Medical Association

Published: Jun 1, 1987

References