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Intercellular IgA Dermatosis of Childhood: Selective Deposition of Monomer IgA1 in the Intercellular Space of the Epidermis

Intercellular IgA Dermatosis of Childhood: Selective Deposition of Monomer IgA1 in the... Abstract • We describe a 7-year-old girl with recurrent pruritic vesiculopustular lesions involving the trunk, extremities, face, and oral mucosa. Histopathologic examination revealed intraepidermal bullae containing neutrophils and eosinophils, and direct immunofluorescence test showed the deposition of IgA in the intercellular space of the epidermis. Circulating IgA anti-intercellular antibodies were also detected by indirect immunofluorescence test. Immunofluorescence studies using monoclonal antibodies to human IgA subclasses showed that these IgA antibodies belonged to IgA1. Antisera against J chain and secretory component did not show any specific intercellular staining. Surface IgA+-B cells were transiently increased in the peripheral blood during the active stage of the disease. These results indicated the extragut origin of these IgA antibodies. Dapsone therapy was shown to be very effective. (Arch Dermatol. 1991;127:221-224) References 1. Wallach D, Foldès C, Cottenot F. Pustulose sous-cornée acantholyse superficielle et IgA monoclonale . Ann Dermatol Venereol. 1982;109:953-963. 2. Tagami H, Iwatsuki K, Iwase Y, Yamada M. 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 3. Burrows D, Bingham EA. Subcorneal pustular dermatosis and IgA gammopathy . Br J Dermatol. 1984;111:91-93.Crossref 4. Huff JC, Golitz LE, Kunke KS. Intraepidermal neutrophilic IgA dermatosis . N Engl J Med. 1985;313:1643-1645.Crossref 5. Hashimoto T, Inamoto N, Nakamura K, Nishikawa T. Intercellular IgA dermatosis with clinical features of subcorneal pustular dermatosis . Arch Dermatol. 1987;123:1062-1065.Crossref 6. Nishikawa T, Shimizu H, Hashimoto T. Role of IgA intercellular antibodies: report of clinically and immunologically atypical cases . In: Orfanos CE, Stadler R, Gollnick H, eds. Proceedings of the 17th World Congress of Dermatology . New York, NY: Springer-Verlag NY Inc; 1987:383-384. 7. Gollnick H, Thies W, Taud W, Orfanos CE. Intraepidermale neutrophile und eosinophile IgA-Dermatose . In: Gollnick H, Stadler R, eds. Fallvorstellungen anlässlich des 17. Weltkongresses für Dermatologie, Berlin, 1987 . Stuttgart, Germany: Schattauer; 1987:68-70. 8. Piette W, Burken RR, Ray TL. Intraepidermal neutrophilic IgA dermatosis: presence of circulating pemphigus-like IgA antibody specific for monkey epithelium . J Invest Dermatol. 1987; 88:512. 9. Beutner EH, Chorzelski TP, Wilson RM, et al. IgA pemphigus foliaceus: report of two cases and review of literature . J Am Acad Dermatol. 1989;20:89-97.Crossref 10. Saurat JH, Mérot Y, Salomon D, Didierjean L. Pemphiguslike IgA deposits and vesiculo-pustular dermatosis in a 10-year-old girl . Dermatologica . 1987;175:96-100.Crossref 11. Wright S, Phillips T, Ryan J, Leigh IM. Intraepidermal neutrophilic IgA dermatosis with colitis . Br J Dermatol. 1989;129:113-119.Crossref 12. Conley ME, Kearney JF, Lawton AR III, Cooper MD. Differentiation of human B cells expressing the IgA subclasses as demonstrated by monoclonal antibodies . J Immunol. 1980;125:2311-2316. 13. Leonard JN, Haffenden GP, Unthworth DJ, Ring NP, Holborow EJ, Fly L. Evidence that the IgA in patients with linear IgA disease is qualitatively different from that of patients with dermatitis herpetiformis . Br J Dermatol. 1984;110:315-321.Crossref 14. Wojnarowska F, Delacroix D, Gengoux P. Cutaneous IgA subclasses in dermatitis herpetiformis and linear IgA dermatosis . J Cutan Pathol. 1988;15:272-275.Crossref 15. Nomoto Y, Sakai H, Arimoto S. Increase of IgA-bearing lymphocytes in peripheral blood from patients with IgA nephropathy . Am J Clin Pathol. 1979;71:158-160. 16. Kuno-Sakai H, Sakai H, Nomoto Y, Takakura I, Kimura M. Increase of IgA-bearing lymphocytes in peripheral blood in children with Henoch-Schoenlein purpura . Pediatrics . 1979;64:918-922. 17. Hashimoto T, Ogawa MM, Konohana A, Nishikawa T. Detection of pemphigus vulgaris and pemphigus foliaceus antigens by immunoblot analysis using different antigen sources . J Invest Dermatol. 1990;94:327-331.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

Intercellular IgA Dermatosis of Childhood: Selective Deposition of Monomer IgA1 in the Intercellular Space of the Epidermis

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References (19)

Publisher
American Medical Association
Copyright
Copyright © 1991 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archderm.1991.01680020089011
Publisher site
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Abstract

Abstract • We describe a 7-year-old girl with recurrent pruritic vesiculopustular lesions involving the trunk, extremities, face, and oral mucosa. Histopathologic examination revealed intraepidermal bullae containing neutrophils and eosinophils, and direct immunofluorescence test showed the deposition of IgA in the intercellular space of the epidermis. Circulating IgA anti-intercellular antibodies were also detected by indirect immunofluorescence test. Immunofluorescence studies using monoclonal antibodies to human IgA subclasses showed that these IgA antibodies belonged to IgA1. Antisera against J chain and secretory component did not show any specific intercellular staining. Surface IgA+-B cells were transiently increased in the peripheral blood during the active stage of the disease. These results indicated the extragut origin of these IgA antibodies. Dapsone therapy was shown to be very effective. (Arch Dermatol. 1991;127:221-224) References 1. Wallach D, Foldès C, Cottenot F. Pustulose sous-cornée acantholyse superficielle et IgA monoclonale . Ann Dermatol Venereol. 1982;109:953-963. 2. Tagami H, Iwatsuki K, Iwase Y, Yamada M. 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 3. Burrows D, Bingham EA. Subcorneal pustular dermatosis and IgA gammopathy . Br J Dermatol. 1984;111:91-93.Crossref 4. Huff JC, Golitz LE, Kunke KS. Intraepidermal neutrophilic IgA dermatosis . N Engl J Med. 1985;313:1643-1645.Crossref 5. Hashimoto T, Inamoto N, Nakamura K, Nishikawa T. Intercellular IgA dermatosis with clinical features of subcorneal pustular dermatosis . Arch Dermatol. 1987;123:1062-1065.Crossref 6. Nishikawa T, Shimizu H, Hashimoto T. Role of IgA intercellular antibodies: report of clinically and immunologically atypical cases . In: Orfanos CE, Stadler R, Gollnick H, eds. Proceedings of the 17th World Congress of Dermatology . New York, NY: Springer-Verlag NY Inc; 1987:383-384. 7. Gollnick H, Thies W, Taud W, Orfanos CE. Intraepidermale neutrophile und eosinophile IgA-Dermatose . In: Gollnick H, Stadler R, eds. Fallvorstellungen anlässlich des 17. Weltkongresses für Dermatologie, Berlin, 1987 . Stuttgart, Germany: Schattauer; 1987:68-70. 8. Piette W, Burken RR, Ray TL. Intraepidermal neutrophilic IgA dermatosis: presence of circulating pemphigus-like IgA antibody specific for monkey epithelium . J Invest Dermatol. 1987; 88:512. 9. Beutner EH, Chorzelski TP, Wilson RM, et al. IgA pemphigus foliaceus: report of two cases and review of literature . J Am Acad Dermatol. 1989;20:89-97.Crossref 10. Saurat JH, Mérot Y, Salomon D, Didierjean L. Pemphiguslike IgA deposits and vesiculo-pustular dermatosis in a 10-year-old girl . Dermatologica . 1987;175:96-100.Crossref 11. Wright S, Phillips T, Ryan J, Leigh IM. Intraepidermal neutrophilic IgA dermatosis with colitis . Br J Dermatol. 1989;129:113-119.Crossref 12. Conley ME, Kearney JF, Lawton AR III, Cooper MD. Differentiation of human B cells expressing the IgA subclasses as demonstrated by monoclonal antibodies . J Immunol. 1980;125:2311-2316. 13. Leonard JN, Haffenden GP, Unthworth DJ, Ring NP, Holborow EJ, Fly L. Evidence that the IgA in patients with linear IgA disease is qualitatively different from that of patients with dermatitis herpetiformis . Br J Dermatol. 1984;110:315-321.Crossref 14. Wojnarowska F, Delacroix D, Gengoux P. Cutaneous IgA subclasses in dermatitis herpetiformis and linear IgA dermatosis . J Cutan Pathol. 1988;15:272-275.Crossref 15. Nomoto Y, Sakai H, Arimoto S. Increase of IgA-bearing lymphocytes in peripheral blood from patients with IgA nephropathy . Am J Clin Pathol. 1979;71:158-160. 16. Kuno-Sakai H, Sakai H, Nomoto Y, Takakura I, Kimura M. Increase of IgA-bearing lymphocytes in peripheral blood in children with Henoch-Schoenlein purpura . Pediatrics . 1979;64:918-922. 17. Hashimoto T, Ogawa MM, Konohana A, Nishikawa T. Detection of pemphigus vulgaris and pemphigus foliaceus antigens by immunoblot analysis using different antigen sources . J Invest Dermatol. 1990;94:327-331.Crossref

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Archives of Dermatology – American Medical Association

Published: Feb 1, 1991

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