Non-paraneoplastic autoimmune subepidermal bullous disease associated with fatal bronchiolitis obliterans.

Abstract

Bronchiolitis obliterans is a small-airway obstructive lung disease for which immunologically mediated pathogenesis is supposed. Frequent association of bronchiolitis obliterans with paraneoplastic pemphigus is well known, but its association with other autoimmune bullous diseases has not been reported except for a case of anti-laminin-332-type mucous membrane pemphigoid in a patient with chronic graft-versus-host disease. We report a case of non-paraneoplastic autoimmune subepidermal bullous disease associated with fatal bronchiolitis obliterans in a patient without transplantation. Although the patient's serum contained immunoglobulin (Ig)A antibodies to the 180-kDa bullous pemphigoid antigen/type XVII collagen and IgG antibodies to laminin-332, diagnosis of either linear IgA bullous dermatosis or mucous membrane pemphigoid could not be made because of the failure to detect linear IgA deposition at the basement membrane zone by direct immunofluorescence and the lack of mucous membrane lesions. Physicians should be aware that autoimmune bullous diseases other than paraneoplastic pemphigus can also associate with this rare but potentially fatal lung disease.

• Authors: Mari Orime, Katsuhiro Tomiyama, Hideki Hashidate, Satoru Yoshida, Satoshi Hokari, Akiko Tsuda, Hisashi Yokoyama, Jun-Ichi Narita, Youhei Uchida, Takuro Kanekura, Riichiro Abe, Norito Ishii, Takashi Hashimoto, Kazuhiro Kawai
• Journal: The Journal of dermatology (J Dermatol) Vol. 44 Issue 4 Pg. 461-464 (Apr 2017) ISSN: 1346-8138 [Electronic] England
• PMID: 27914107 (Publication Type: Case Reports, Journal Article)
• Copyright: © 2016 Japanese Dermatological Association.

Chemical References

• Anti-Bacterial Agents
• Autoantibodies
• Autoantigens
• Cell Adhesion Molecules
• Glucocorticoids
• Immunoglobulin A
• Immunoglobulin G
• Non-Fibrillar Collagens
• collagen type XVII
• kalinin
• Vitamin B Complex
• Niacinamide
• Prednisolone
• Minocycline

Topics

• Aged
• Anti-Bacterial Agents (therapeutic use)
• Autoantibodies (blood)
• Autoantigens (immunology)
• Basement Membrane (metabolism)
• Biopsy
• Bronchiolitis Obliterans (blood, complications)
• Cell Adhesion Molecules (immunology)
• Fatal Outcome
• Fluorescent Antibody Technique, Direct
• Glucocorticoids (therapeutic use)
• Humans
• Immunoglobulin A (blood, metabolism)
• Immunoglobulin G (blood)
• Male
• Minocycline (therapeutic use)
• Niacinamide (therapeutic use)
• Non-Fibrillar Collagens (immunology)
• Paraneoplastic Syndromes (complications)
• Pemphigoid, Bullous (complications, drug therapy, immunology, pathology)
• Prednisolone (therapeutic use)
• Respiratory Insufficiency (etiology)
• Vitamin B Complex (therapeutic use)