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

Research output: Contribution to journalArticle

20 Citations (Scopus)

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.

Original languageEnglish
Pages (from-to)355-359
Number of pages5
JournalEuropean Journal of Dermatology
Volume12
Issue number4
Publication statusPublished - 2002
Externally publishedYes

Fingerprint

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

Keywords

  • Bronchiolitis obliterans
  • Castleman's disease
  • Desmoglein
  • Enzyme-linked immunosorbent assay
  • High resolution computed tomography
  • Paraneoplastic pemphigus

ASJC Scopus subject areas

  • Dermatology

Cite this

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.

In: European Journal of Dermatology, Vol. 12, No. 4, 2002, p. 355-359.

Research output: Contribution to journalArticle

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, vol. 12, no. 4, pp. 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. In: European Journal of Dermatology. 2002 ; Vol. 12, No. 4. pp. 355-359.
@article{1776f5ea6baa4d6cbe071bc8ff6ba182,
title = "Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans",
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.",
keywords = "Bronchiolitis obliterans, Castleman's disease, Desmoglein, Enzyme-linked immunosorbent assay, High resolution computed tomography, Paraneoplastic pemphigus",
author = "Wataru Fujimoto and Arihiko Kanehiro and Kazue Kuwamoto-Hara and Mari Saitoh and Takashi Nakakita and Masayuki Amagai and Jir{\^o} Arata and Keiji Iwatsuki",
year = "2002",
language = "English",
volume = "12",
pages = "355--359",
journal = "European Journal of Dermatology",
issn = "1167-1122",
publisher = "John Libbey Eurotext",
number = "4",

}

TY - JOUR

T1 - Paraneoplastic pemphigus associated with Castleman's disease and asymptomatic bronchiolitis obliterans

AU - Fujimoto, Wataru

AU - Kanehiro, Arihiko

AU - Kuwamoto-Hara, Kazue

AU - Saitoh, Mari

AU - Nakakita, Takashi

AU - Amagai, Masayuki

AU - Arata, Jirô

AU - Iwatsuki, Keiji

PY - 2002

Y1 - 2002

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

UR - http://www.scopus.com/inward/record.url?scp=0036018519&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=0036018519&partnerID=8YFLogxK

M3 - Article

VL - 12

SP - 355

EP - 359

JO - European Journal of Dermatology

JF - European Journal of Dermatology

SN - 1167-1122

IS - 4

ER -