Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans

Wataru Fujimoto, Arihiko Kanehiro, Kazue Kuwamoto-Hara, Mari Saitoh, Takashi Nakakita, Masayuki Amagai, Jirô Arata, Keiji Iwatsuki

研究成果: Article

20 引用 (Scopus)

抄録

We describe a 19-year-old male patient who presented with recalcitrant erosions limited to the orolabial and genital mucosa for 18 months. The clinicopathologic diagnosis of paraneoplastic pemphigus was confirmed by indirect immunofluorescence staining of murine bladder epithelium, and the presence of IgG autoantibodies against envoplakin, periplakin, and 170 kDa protein. Enzyme-linked immunosorbent assay (ELISA) with desmoglein 3 (Dsg3) and desmoglein 1 (Dsg1) recombinant proteins showed that the patient's sera were positive against Dsg3. Castleman's tumour was found in the pelvic cavity and resected completely. The orogenital erosions disappeared slowly after the resection of Castleman's tumour with the concomitant decrease in intercellular antibody titre and index values of Dsg ELISA. Although the patient was unaware of dyspnea on excertion, a notable air flow obstruction persisted over 17 months. The expiratory images of high resolution computed tomography showed air trapping, indicating the presence of asymptomatic but gradually progressive bronchiolitis obliterans.

元の言語English
ページ(範囲)355-359
ページ数5
ジャーナルEuropean Journal of Dermatology
12
発行部数4
出版物ステータスPublished - 2002
外部発表Yes

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Bronchiolitis Obliterans
Giant Lymph Node Hyperplasia
Pemphigus
Desmoglein 3
Desmoglein 1
Enzyme-Linked Immunosorbent Assay
Air
Indirect Fluorescent Antibody Technique
Recombinant Proteins
Dyspnea
Autoantibodies
Mucous Membrane
Urinary Bladder
Epithelium
Immunoglobulin G
Tomography
Staining and Labeling
Antibodies
Serum
Proteins

ASJC Scopus subject areas

  • Dermatology

これを引用

Fujimoto, W., Kanehiro, A., Kuwamoto-Hara, K., Saitoh, M., Nakakita, T., Amagai, M., ... Iwatsuki, K. (2002). Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans. European Journal of Dermatology, 12(4), 355-359.

Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans. / Fujimoto, Wataru; Kanehiro, Arihiko; Kuwamoto-Hara, Kazue; Saitoh, Mari; Nakakita, Takashi; Amagai, Masayuki; Arata, Jirô; Iwatsuki, Keiji.

:: European Journal of Dermatology, 巻 12, 番号 4, 2002, p. 355-359.

研究成果: Article

Fujimoto, W, Kanehiro, A, Kuwamoto-Hara, K, Saitoh, M, Nakakita, T, Amagai, M, Arata, J & Iwatsuki, K 2002, 'Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans', European Journal of Dermatology, 巻. 12, 番号 4, pp. 355-359.
Fujimoto W, Kanehiro A, Kuwamoto-Hara K, Saitoh M, Nakakita T, Amagai M その他. Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans. European Journal of Dermatology. 2002;12(4):355-359.
Fujimoto, Wataru ; Kanehiro, Arihiko ; Kuwamoto-Hara, Kazue ; Saitoh, Mari ; Nakakita, Takashi ; Amagai, Masayuki ; Arata, Jirô ; Iwatsuki, Keiji. / Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans. :: European Journal of Dermatology. 2002 ; 巻 12, 番号 4. pp. 355-359.
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abstract = "We describe a 19-year-old male patient who presented with recalcitrant erosions limited to the orolabial and genital mucosa for 18 months. The clinicopathologic diagnosis of paraneoplastic pemphigus was confirmed by indirect immunofluorescence staining of murine bladder epithelium, and the presence of IgG autoantibodies against envoplakin, periplakin, and 170 kDa protein. Enzyme-linked immunosorbent assay (ELISA) with desmoglein 3 (Dsg3) and desmoglein 1 (Dsg1) recombinant proteins showed that the patient's sera were positive against Dsg3. Castleman's tumour was found in the pelvic cavity and resected completely. The orogenital erosions disappeared slowly after the resection of Castleman's tumour with the concomitant decrease in intercellular antibody titre and index values of Dsg ELISA. Although the patient was unaware of dyspnea on excertion, a notable air flow obstruction persisted over 17 months. The expiratory images of high resolution computed tomography showed air trapping, indicating the presence of asymptomatic but gradually progressive bronchiolitis obliterans.",
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AU - Fujimoto, Wataru

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AU - Kuwamoto-Hara, Kazue

AU - Saitoh, Mari

AU - Nakakita, Takashi

AU - Amagai, Masayuki

AU - Arata, Jirô

AU - Iwatsuki, Keiji

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N2 - We describe a 19-year-old male patient who presented with recalcitrant erosions limited to the orolabial and genital mucosa for 18 months. The clinicopathologic diagnosis of paraneoplastic pemphigus was confirmed by indirect immunofluorescence staining of murine bladder epithelium, and the presence of IgG autoantibodies against envoplakin, periplakin, and 170 kDa protein. Enzyme-linked immunosorbent assay (ELISA) with desmoglein 3 (Dsg3) and desmoglein 1 (Dsg1) recombinant proteins showed that the patient's sera were positive against Dsg3. Castleman's tumour was found in the pelvic cavity and resected completely. The orogenital erosions disappeared slowly after the resection of Castleman's tumour with the concomitant decrease in intercellular antibody titre and index values of Dsg ELISA. Although the patient was unaware of dyspnea on excertion, a notable air flow obstruction persisted over 17 months. The expiratory images of high resolution computed tomography showed air trapping, indicating the presence of asymptomatic but gradually progressive bronchiolitis obliterans.

AB - We describe a 19-year-old male patient who presented with recalcitrant erosions limited to the orolabial and genital mucosa for 18 months. The clinicopathologic diagnosis of paraneoplastic pemphigus was confirmed by indirect immunofluorescence staining of murine bladder epithelium, and the presence of IgG autoantibodies against envoplakin, periplakin, and 170 kDa protein. Enzyme-linked immunosorbent assay (ELISA) with desmoglein 3 (Dsg3) and desmoglein 1 (Dsg1) recombinant proteins showed that the patient's sera were positive against Dsg3. Castleman's tumour was found in the pelvic cavity and resected completely. The orogenital erosions disappeared slowly after the resection of Castleman's tumour with the concomitant decrease in intercellular antibody titre and index values of Dsg ELISA. Although the patient was unaware of dyspnea on excertion, a notable air flow obstruction persisted over 17 months. The expiratory images of high resolution computed tomography showed air trapping, indicating the presence of asymptomatic but gradually progressive bronchiolitis obliterans.

KW - Bronchiolitis obliterans

KW - Castleman's disease

KW - Desmoglein

KW - Enzyme-linked immunosorbent assay

KW - High resolution computed tomography

KW - Paraneoplastic pemphigus

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