水疱性類天疱瘡と鑑別困難であった抗BP180型粘膜類天疱瘡の1例

Specific target antigens have been identified in many autoimmune syndromes, and most of these diseases are caused by single target antigens. However, 2 or more target antigens are known to be involved in mucous membrane pemphigoid, which can exhibit various clinical characteristics. We report a case...

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Published in日本口腔外科学会雑誌 Vol. 58; no. 5; pp. 297 - 301
Main Authors 坂口, 和歌子, 近津, 大地, 宮本, 重樹, 安田, 卓史, 金子, 忠良
Format Journal Article
LanguageJapanese
Published 社団法人 日本口腔外科学会 20.05.2012
Subjects
口腔粘膜
多彩な臨床症状
抗BP180 型粘膜類天疱瘡
標的抗原
水疱性類天疱瘡
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ISSN0021-5163
2186-1579
DOI10.5794/jjoms.58.297

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Abstract Specific target antigens have been identified in many autoimmune syndromes, and most of these diseases are caused by single target antigens. However, 2 or more target antigens are known to be involved in mucous membrane pemphigoid, which can exhibit various clinical characteristics. We report a case of anti-BP180-type mucous membrane pemphigoid that was difficult to distinguish from bullous pemphigoid on the basis of immunological analysis. The patient was a 76-year-old man who visited our department because of gum erosion and pain. Sore gums and ulcers were observed on both the upper and lower jaws, and mild pruritic erythema was noted on the posterior wall of the oral cavity. Direct immunohistological analysis of tissues obtained from the affected jaw showed deposition of IgG and C3 in the basal membrane. An enzymelinked immunosorbent assay（ELISA）for BP180 and BP230 showed positive results, and an immunoblot assay using the recombinant BP180 protein showed positive results. In addition, an immunoblot assay using the recombinant BP180 protein showed positive results for IgG antibodies against BP180 NC16a and negative results for IgG antibodies against the C-terminal portion of BP180. The patient was suspected to have bullous pemphigoid, but because the main clinical findings were pathologic changes of the oral mucosa, the patient was given a diagnosis of anti-BP180-type mucosal pemphigoid. The symptoms improved with an improvement in oral hygiene and the application of a steroid.
AbstractList Specific target antigens have been identified in many autoimmune syndromes, and most of these diseases are caused by single target antigens. However, 2 or more target antigens are known to be involved in mucous membrane pemphigoid, which can exhibit various clinical characteristics. We report a case of anti-BP180-type mucous membrane pemphigoid that was difficult to distinguish from bullous pemphigoid on the basis of immunological analysis. The patient was a 76-year-old man who visited our department because of gum erosion and pain. Sore gums and ulcers were observed on both the upper and lower jaws, and mild pruritic erythema was noted on the posterior wall of the oral cavity. Direct immunohistological analysis of tissues obtained from the affected jaw showed deposition of IgG and C3 in the basal membrane. An enzymelinked immunosorbent assay（ELISA）for BP180 and BP230 showed positive results, and an immunoblot assay using the recombinant BP180 protein showed positive results. In addition, an immunoblot assay using the recombinant BP180 protein showed positive results for IgG antibodies against BP180 NC16a and negative results for IgG antibodies against the C-terminal portion of BP180. The patient was suspected to have bullous pemphigoid, but because the main clinical findings were pathologic changes of the oral mucosa, the patient was given a diagnosis of anti-BP180-type mucosal pemphigoid. The symptoms improved with an improvement in oral hygiene and the application of a steroid.
Author 坂口, 和歌子
近津, 大地
安田, 卓史
金子, 忠良
宮本, 重樹
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References 13) 渡辺　彩, 早乙女敦子, 他：水疱性類天疱瘡口腔粘膜に初発した症例. 皮膚病診療 30: 389-392, 2008.
17) Nie Z and Hashimoto T : IgA Antibodies of Cicatrial Pemphigoid Sera Specifically React with C-Terminus of BP180. J Invest Dermatol 112: 254-255, 1999.
7) 川島愛雄, 石倉多美子, 他：水疱性類天疱瘡29例のまとめと予後調査. 西日皮膚 39: 37-45, 1977.
14) 澤村大輔：自己免疫性水疱症におけるBP180の異なる関与. 医学のあゆみ 218: 951-955, 2006.
4) 満山陽子, 堀田隆之, 他：喉頭狭窄をきたした抗ラミニン5型瘢痕性類天疱瘡の1例. 臨皮 61: 975-978, 2007.
5) Hashimoto T, Han-Yaku H, et al : Four Japanese Patients with cicatrial pemphigoid showing both IgG and IgA antibodies against the 180kDa bullous pemphigoid pemphigoid antigen. Br J Dermatol 137: 305-306, 1997.
22) 加藤則人, 岡本啓子, 他：ニコチン酸アミドとミノサイクリンの併用療法が著効を示した水疱性類天疱瘡の2例. 皮膚臨床 21: 1281-1286, 1990.
26) Czernik A and Bystryn JC : Improvement of intravenous immunoglobulin therapy for bullous pemphigoid by adding immunosuppressive agents. Arch Dermatol 144: 658-661, 2008.
1) 藤本　亘：「粘膜類天疱瘡」の考え方. 皮膚病診療 30: 250-257, 2008.
3) Neville BW, Damm DD, et al : Oral andmaxillofacialpathology. 3rdEd, Sanders Co, St Louis, 771-774, 2009.
25) 石地尚興：自己免疫性水疱症の単純血漿交換療法. 日アフェレシス会誌 27: 117-121, 2008.
27) Kjellman P, Eriksson H, et al : A retrospective analysis of patients with bullous pemphigoid treated with methotrexate. Arch Dermatol 144: 612-616, 2008.
20) Berk MA and Lorincz AL : The treatment of bullous pemphigoid with tetracycline and Niacinamide. Arch Dermatol 122: 670-674, 1986.
16) Zillikens D, Rose PA, et al : Tight Clustering of Extracellular BP180 Epitopes Recognized by Bullous Pemphigoid Autoantibodies. J Invest Dermatol 109: 573-579, 1997.
9) 櫻井一成, 高橋由美子, 他：口腔粘膜に初発した水疱性類天疱瘡の1例. 日口外誌 38: 518-519, 1992.
19) 橋本　隆：天疱瘡と類天疱瘡. 日皮会誌 116: 1161-1171, 2006.
11) 高岡一樹, 萬野幸代, 他：口蓋粘膜に生じた水疱性類天疱瘡の1例. 日口外誌 50: 672-675, 2004.
8) 小林博人, 田辺俊英, 他：瘢痕性類天疱瘡, 水疱性類天疱瘡に対するニコチン酸アミド大量療法. 皮膚 32: 240-244, 1990.
15) Giudice GJ, Emery DJ, et al : Bullous Pemphigoid and Herpes Gestatiois Autoantibodies Recognize A Common Non-Collagenous Site on the BP180 Ectodomain. J Immunol 151: 5742-5750, 1993.
18) 國井隆英, 佐々木喜教, 他：抗BP180型　粘膜類天疱瘡の1例. 臨皮 64: 125-128, 2010.
23) 西　隆久, 海塚安郎, 他：水疱性類天疱瘡に対するニコチン酸アミドとテトラサイクリン併用療法の試み. 西日皮膚 51: 288-292, 1989.
10) 野中玲子, 小坂和己, 他：口腔粘膜病変で発症した水疱症の2例. 耳鼻臨床 91: 273-280, 1998.
2) 橋本　隆：粘膜類天疱瘡. 皮膚病診療 30: 385-388, 2008.
12) 伊藤大典, 豊田　華, 他：口腔内に初発した水疱性類天疱瘡の1例. 日口粘膜 12: 30-36, 2006.
21) 橋本　隆：類天疱瘡のテトラサイクリン療法. 皮膚臨床 37: 1063-1066, 1995.
6) Chan LS, Ahmed AR, et al : The First International Consensus on Mucous Membrane Pemphigoid. Arch Dermatol 138: 370-379, 2002.
24) 川久保　洋：ダプソン （DDS）. 皮膚臨床 49: 1239-1242, 2007.
References_xml – reference: 11) 高岡一樹, 萬野幸代, 他：口蓋粘膜に生じた水疱性類天疱瘡の1例. 日口外誌 50: 672-675, 2004.
– reference: 23) 西　隆久, 海塚安郎, 他：水疱性類天疱瘡に対するニコチン酸アミドとテトラサイクリン併用療法の試み. 西日皮膚 51: 288-292, 1989.
– reference: 5) Hashimoto T, Han-Yaku H, et al : Four Japanese Patients with cicatrial pemphigoid showing both IgG and IgA antibodies against the 180kDa bullous pemphigoid pemphigoid antigen. Br J Dermatol 137: 305-306, 1997.
– reference: 16) Zillikens D, Rose PA, et al : Tight Clustering of Extracellular BP180 Epitopes Recognized by Bullous Pemphigoid Autoantibodies. J Invest Dermatol 109: 573-579, 1997.
– reference: 8) 小林博人, 田辺俊英, 他：瘢痕性類天疱瘡, 水疱性類天疱瘡に対するニコチン酸アミド大量療法. 皮膚 32: 240-244, 1990.
– reference: 21) 橋本　隆：類天疱瘡のテトラサイクリン療法. 皮膚臨床 37: 1063-1066, 1995.
– reference: 3) Neville BW, Damm DD, et al : Oral andmaxillofacialpathology. 3rdEd, Sanders Co, St Louis, 771-774, 2009.
– reference: 14) 澤村大輔：自己免疫性水疱症におけるBP180の異なる関与. 医学のあゆみ 218: 951-955, 2006.
– reference: 6) Chan LS, Ahmed AR, et al : The First International Consensus on Mucous Membrane Pemphigoid. Arch Dermatol 138: 370-379, 2002.
– reference: 24) 川久保　洋：ダプソン （DDS）. 皮膚臨床 49: 1239-1242, 2007.
– reference: 19) 橋本　隆：天疱瘡と類天疱瘡. 日皮会誌 116: 1161-1171, 2006.
– reference: 20) Berk MA and Lorincz AL : The treatment of bullous pemphigoid with tetracycline and Niacinamide. Arch Dermatol 122: 670-674, 1986.
– reference: 18) 國井隆英, 佐々木喜教, 他：抗BP180型　粘膜類天疱瘡の1例. 臨皮 64: 125-128, 2010.
– reference: 25) 石地尚興：自己免疫性水疱症の単純血漿交換療法. 日アフェレシス会誌 27: 117-121, 2008.
– reference: 13) 渡辺　彩, 早乙女敦子, 他：水疱性類天疱瘡口腔粘膜に初発した症例. 皮膚病診療 30: 389-392, 2008.
– reference: 27) Kjellman P, Eriksson H, et al : A retrospective analysis of patients with bullous pemphigoid treated with methotrexate. Arch Dermatol 144: 612-616, 2008.
– reference: 15) Giudice GJ, Emery DJ, et al : Bullous Pemphigoid and Herpes Gestatiois Autoantibodies Recognize A Common Non-Collagenous Site on the BP180 Ectodomain. J Immunol 151: 5742-5750, 1993.
– reference: 7) 川島愛雄, 石倉多美子, 他：水疱性類天疱瘡29例のまとめと予後調査. 西日皮膚 39: 37-45, 1977.
– reference: 9) 櫻井一成, 高橋由美子, 他：口腔粘膜に初発した水疱性類天疱瘡の1例. 日口外誌 38: 518-519, 1992.
– reference: 17) Nie Z and Hashimoto T : IgA Antibodies of Cicatrial Pemphigoid Sera Specifically React with C-Terminus of BP180. J Invest Dermatol 112: 254-255, 1999.
– reference: 26) Czernik A and Bystryn JC : Improvement of intravenous immunoglobulin therapy for bullous pemphigoid by adding immunosuppressive agents. Arch Dermatol 144: 658-661, 2008.
– reference: 10) 野中玲子, 小坂和己, 他：口腔粘膜病変で発症した水疱症の2例. 耳鼻臨床 91: 273-280, 1998.
– reference: 22) 加藤則人, 岡本啓子, 他：ニコチン酸アミドとミノサイクリンの併用療法が著効を示した水疱性類天疱瘡の2例. 皮膚臨床 21: 1281-1286, 1990.
– reference: 1) 藤本　亘：「粘膜類天疱瘡」の考え方. 皮膚病診療 30: 250-257, 2008.
– reference: 2) 橋本　隆：粘膜類天疱瘡. 皮膚病診療 30: 385-388, 2008.
– reference: 4) 満山陽子, 堀田隆之, 他：喉頭狭窄をきたした抗ラミニン5型瘢痕性類天疱瘡の1例. 臨皮 61: 975-978, 2007.
– reference: 12) 伊藤大典, 豊田　華, 他：口腔内に初発した水疱性類天疱瘡の1例. 日口粘膜 12: 30-36, 2006.
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SubjectTerms 口腔粘膜
多彩な臨床症状
抗BP180 型粘膜類天疱瘡
標的抗原
水疱性類天疱瘡
Title 水疱性類天疱瘡と鑑別困難であった抗BP180型粘膜類天疱瘡の1例
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