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Epidermodysplasia Verruciformis Accompanied by Large Granular Lymphocytosis: Report of a Case and Immunological Studies

Epidermodysplasia Verruciformis Accompanied by Large Granular Lymphocytosis: Report of a Case and... Abstract • A 34-year-old man with epidermodysplasia verruciformis showed increased natural killer (NK) cell activity in his peripheral blood cells, which was found to be due to abnormal expansion of large granular lymphocytes (LGL). Surface marker analysis of LGL in a two-color immunofluorescence test demonstrated that all LGL subsets, Leu7+11-, Leu-7+11+, and Leu-7-11+, were increased. In contrast, his peripheral blood mononuclear cells showed a remarkable decrease in response to T cell mitogens, which could not be restored by depletion of the LGL subpopulation from the mononuclear cells. Immunohistologic studies for dermal distribution of LGL showed numerous Leu-7+ cells but no Leu-11+ cells at the sites of delayed hypersensitivity reaction or interferon β- or interferon γ-injected sites. These findings suggest that the elevated NK cell activity in this patient with epidermodysplasia verruciformis was caused by overall expansion of normal LGL subpopulations, but that the decreased T-cell mitogenic response represented a primary T-cell defect rather than a direct suppression by increased NK cells. It is noteworthy that despite this extraordinary increase of all LGL subpopulations in the blood, Leu-11+ NK cells never appeared in the skin. (Arch Dermatol 1989;125:660-665) References 1. Tagami H, Takigawa M, Ogino A, et al: Spontaneous regression of plane warts after inflammation: Clinical and histologic studies in 25 cases . Arch Dermatol 1977;113:1209-1213.Crossref 2. Tagami H, Oku T, Iwatsuki K: Primary tissue culture of spontaneously regressing flat warts: In vitro attack by mononuclear cells against wart-derived epidermal cells . Cancer 1985; 55:2437-2441.Crossref 3. Jablonska S, Dabrowski J, Jakubowitz K: Epidermodysplasia verruciformis as a model in studies on the role of papovaviruses in oncogenesis . Cancer Res 1972;32:583-589. 4. Glinski W, Jablonska S, Langner A, et al: Cell-mediated immunity in epidermodysplasia verruciformis . Dermatologica 1976;153:218-227.Crossref 5. Kaminski M, Pawinska M, Jablonska S, et al: Increased natural killer cell activity in patients with epidermodysplasia verruciformis . Arch Dermatol 1985;121:84-86.Crossref 6. Majewski S, Skopinska E, Jablonska S, et al: Partial defects of cell-mediated immunity in patients with epidermodysplasia verruciformis . J Am Acad Dermatol 1986;15:966-973.Crossref 7. Abo T, Miller CA, Balch CM: Characterization of human granular lymphocyte subpopulations expressing HNK-1 (Leu-7) and Leu-11 antigens in the blood and lymphoid tissues from fetuses, neonates and adults . Eur J Immunol 1984;14:616-623.Crossref 8. Abo T, Balch CM: Characterization of HNK-1+ (Leu-7) human lymphocytes: II. Distinguishing phenotypic and functional properties of natural killer cells from activated NK-like cells . J Immunol 1982;129:1758-1761. 9. Tilden AB, Abo T, Balch CM: Suppressor cell function of human granular lymphocytes identified by the HNK-1 (Leu 7) monoclonal antibody . J Immunol 1983;130:1171-1175. 10. Landay A, Gartland GL, Clement LT: Characterization of a phenotypically distinct subpopulation of Leu-2+ cells that suppresses T cell proliferative responses . J Immunol 1983;131:2757-2761. 11. Mortari F, Bains MA, Singhal SK: Immunoregulatory activity of human bone marrow: Identification of suppressor cells possessing OKM1, SSEA-1, and HNK-1 antigens . J Immunol 1986;137:1133-1137. 12. Itoh K, Tilden AB, Kumagai K, et al: Leu-11+ lymphocytes with natural killer (NK) activity are precursors of recombinant interleukin 2 (rIL 2)-induced activated killer (AK) cells . J Immunol 1985;134:802-807. 13. Pandolfi F, Semenzato G, de Rossi G, et al: HNK-1 monoclonal antibody (Leu-7) in the identification of abnormal expansions of large granular lymphocytes . Clin Exp Immunol 1983;52:641-647. 14. Itoh K, Tsuchikawa K, Awataguchi T, et al: A case of chronic lymphocytic leukemia with properties characteristic of natural killer cells . Blood 1983;61:940-948. 15. Peter HH, Friedrich W, Dopfer R, et al: NK cell function in severe combined immunodeficiency (SCID): Evidence of a common T and NK cell defect in some but not all SCID patients . J Immunol 1983;131:2332-2339. 16. Bakke AC, Gray JD, Abo W, et al: Studies on human blood lymphocytes with iC3b (Type 3) complement receptors: I. Granular, Fc-IgG receptor positive and negative subsets in healthy subjects and patients with systemic lupus erythematosus . J Immunol 1986;136:1253-1259. 17. Ennis FA, Bear AS, Rilley D, et al: Interferon induction and increased natural killer cell activity in influenza infections in man . Lancet 1981;2:891-893.Crossref 18. Keleman E, Gergely P, Lehoczy D, et al: Permanent large granular lymphocytosis in the blood of splenectomized individuals without concomitant increase of in vitro natural killer cell cytotoxicity . Clin Exp Immunol 1986;63:696-702. 19. Abo T, Sugawara S, Amenomori A, et al: Selective phagocytosis of gram-positive bacteria and interleukin 1-like factor production by a subpopulation of large granular lymphocytes . J Immunol 1986;136:3189-3197. 20. Abo T, Balch CM: A differentiation antigen of human NK and K cells identified by a monoclonal antibody (HNK-1) . J Immunol 1981;127:1024-1029. 21. Hsu SM, Raine L, Fanger H: Use of avidin-biotin-peroxidase complex (ABC) in immunoperoxidase techniques: A comparison between ABC and unlabelled antibody (PAP) procedures . J Histochem Cytochem 1981;29:577-580.Crossref 22. Androphy EJ, Dvoretzky I, Meluish AE, et al: Response of warts in epidermodysplasia verruciformis to treatment with systemic and intralesional alpha interferon . J Am Acad Dermatol 1984;11:197-202.Crossref 23. Abo T, Balch CM: In vitro propagation of cultured human natural killer cells expressing the HNK-1 differentiation antigen and spontaneous cytotoxic function . Eur J Immunol 1983;13:383-389.Crossref 24. Kohchiyama A, Oka D, Ueki H: Immunohistologic studies of squamous cell carcinoma: Possible participation of Leu-7+ (natural killer) cells as antitumor effector cells . J Invest Dermatol 1986;87:515-518.Crossref 25. Aiba S, Rokugo M, Tagami H: Immunohistologic analysis of the phenomenon of spontaneous regression of numerous flat warts . Cancer 1986;58:1246-1251.Crossref 26. Chardonnet Y, Viac J, Thivolet J: Langerhans cells in human warts . Br J Dermatol 1986;115:669-675.Crossref 27. Haftek M, Jablonska S, Szymanczyk J, et al: Langerhans cells in epidermodysplasia verruciformis . Dermatologica 1987; 174:173-179.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

Epidermodysplasia Verruciformis Accompanied by Large Granular Lymphocytosis: Report of a Case and Immunological Studies

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Publisher
American Medical Association
Copyright
Copyright © 1989 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archderm.1989.01670170074013
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Abstract

Abstract • A 34-year-old man with epidermodysplasia verruciformis showed increased natural killer (NK) cell activity in his peripheral blood cells, which was found to be due to abnormal expansion of large granular lymphocytes (LGL). Surface marker analysis of LGL in a two-color immunofluorescence test demonstrated that all LGL subsets, Leu7+11-, Leu-7+11+, and Leu-7-11+, were increased. In contrast, his peripheral blood mononuclear cells showed a remarkable decrease in response to T cell mitogens, which could not be restored by depletion of the LGL subpopulation from the mononuclear cells. Immunohistologic studies for dermal distribution of LGL showed numerous Leu-7+ cells but no Leu-11+ cells at the sites of delayed hypersensitivity reaction or interferon β- or interferon γ-injected sites. These findings suggest that the elevated NK cell activity in this patient with epidermodysplasia verruciformis was caused by overall expansion of normal LGL subpopulations, but that the decreased T-cell mitogenic response represented a primary T-cell defect rather than a direct suppression by increased NK cells. It is noteworthy that despite this extraordinary increase of all LGL subpopulations in the blood, Leu-11+ NK cells never appeared in the skin. (Arch Dermatol 1989;125:660-665) References 1. Tagami H, Takigawa M, Ogino A, et al: Spontaneous regression of plane warts after inflammation: Clinical and histologic studies in 25 cases . Arch Dermatol 1977;113:1209-1213.Crossref 2. Tagami H, Oku T, Iwatsuki K: Primary tissue culture of spontaneously regressing flat warts: In vitro attack by mononuclear cells against wart-derived epidermal cells . Cancer 1985; 55:2437-2441.Crossref 3. Jablonska S, Dabrowski J, Jakubowitz K: Epidermodysplasia verruciformis as a model in studies on the role of papovaviruses in oncogenesis . Cancer Res 1972;32:583-589. 4. Glinski W, Jablonska S, Langner A, et al: Cell-mediated immunity in epidermodysplasia verruciformis . Dermatologica 1976;153:218-227.Crossref 5. Kaminski M, Pawinska M, Jablonska S, et al: Increased natural killer cell activity in patients with epidermodysplasia verruciformis . Arch Dermatol 1985;121:84-86.Crossref 6. Majewski S, Skopinska E, Jablonska S, et al: Partial defects of cell-mediated immunity in patients with epidermodysplasia verruciformis . J Am Acad Dermatol 1986;15:966-973.Crossref 7. Abo T, Miller CA, Balch CM: Characterization of human granular lymphocyte subpopulations expressing HNK-1 (Leu-7) and Leu-11 antigens in the blood and lymphoid tissues from fetuses, neonates and adults . Eur J Immunol 1984;14:616-623.Crossref 8. Abo T, Balch CM: Characterization of HNK-1+ (Leu-7) human lymphocytes: II. Distinguishing phenotypic and functional properties of natural killer cells from activated NK-like cells . J Immunol 1982;129:1758-1761. 9. Tilden AB, Abo T, Balch CM: Suppressor cell function of human granular lymphocytes identified by the HNK-1 (Leu 7) monoclonal antibody . J Immunol 1983;130:1171-1175. 10. Landay A, Gartland GL, Clement LT: Characterization of a phenotypically distinct subpopulation of Leu-2+ cells that suppresses T cell proliferative responses . J Immunol 1983;131:2757-2761. 11. Mortari F, Bains MA, Singhal SK: Immunoregulatory activity of human bone marrow: Identification of suppressor cells possessing OKM1, SSEA-1, and HNK-1 antigens . J Immunol 1986;137:1133-1137. 12. Itoh K, Tilden AB, Kumagai K, et al: Leu-11+ lymphocytes with natural killer (NK) activity are precursors of recombinant interleukin 2 (rIL 2)-induced activated killer (AK) cells . J Immunol 1985;134:802-807. 13. Pandolfi F, Semenzato G, de Rossi G, et al: HNK-1 monoclonal antibody (Leu-7) in the identification of abnormal expansions of large granular lymphocytes . Clin Exp Immunol 1983;52:641-647. 14. Itoh K, Tsuchikawa K, Awataguchi T, et al: A case of chronic lymphocytic leukemia with properties characteristic of natural killer cells . Blood 1983;61:940-948. 15. Peter HH, Friedrich W, Dopfer R, et al: NK cell function in severe combined immunodeficiency (SCID): Evidence of a common T and NK cell defect in some but not all SCID patients . J Immunol 1983;131:2332-2339. 16. Bakke AC, Gray JD, Abo W, et al: Studies on human blood lymphocytes with iC3b (Type 3) complement receptors: I. Granular, Fc-IgG receptor positive and negative subsets in healthy subjects and patients with systemic lupus erythematosus . J Immunol 1986;136:1253-1259. 17. Ennis FA, Bear AS, Rilley D, et al: Interferon induction and increased natural killer cell activity in influenza infections in man . Lancet 1981;2:891-893.Crossref 18. Keleman E, Gergely P, Lehoczy D, et al: Permanent large granular lymphocytosis in the blood of splenectomized individuals without concomitant increase of in vitro natural killer cell cytotoxicity . Clin Exp Immunol 1986;63:696-702. 19. Abo T, Sugawara S, Amenomori A, et al: Selective phagocytosis of gram-positive bacteria and interleukin 1-like factor production by a subpopulation of large granular lymphocytes . J Immunol 1986;136:3189-3197. 20. Abo T, Balch CM: A differentiation antigen of human NK and K cells identified by a monoclonal antibody (HNK-1) . J Immunol 1981;127:1024-1029. 21. Hsu SM, Raine L, Fanger H: Use of avidin-biotin-peroxidase complex (ABC) in immunoperoxidase techniques: A comparison between ABC and unlabelled antibody (PAP) procedures . J Histochem Cytochem 1981;29:577-580.Crossref 22. Androphy EJ, Dvoretzky I, Meluish AE, et al: Response of warts in epidermodysplasia verruciformis to treatment with systemic and intralesional alpha interferon . J Am Acad Dermatol 1984;11:197-202.Crossref 23. Abo T, Balch CM: In vitro propagation of cultured human natural killer cells expressing the HNK-1 differentiation antigen and spontaneous cytotoxic function . Eur J Immunol 1983;13:383-389.Crossref 24. Kohchiyama A, Oka D, Ueki H: Immunohistologic studies of squamous cell carcinoma: Possible participation of Leu-7+ (natural killer) cells as antitumor effector cells . J Invest Dermatol 1986;87:515-518.Crossref 25. Aiba S, Rokugo M, Tagami H: Immunohistologic analysis of the phenomenon of spontaneous regression of numerous flat warts . Cancer 1986;58:1246-1251.Crossref 26. Chardonnet Y, Viac J, Thivolet J: Langerhans cells in human warts . Br J Dermatol 1986;115:669-675.Crossref 27. Haftek M, Jablonska S, Szymanczyk J, et al: Langerhans cells in epidermodysplasia verruciformis . Dermatologica 1987; 174:173-179.Crossref

Journal

Archives of DermatologyAmerican Medical Association

Published: May 1, 1989

References