Yoshihito Iijima1, Shun Iwai2, Nozomu Motono2, Katsuo Usuda2, Akihiro Shioya3, Shingo Takeuchi4, Shigeki Yamagishi5, Kiyoshi Koizumi5, Sohsuke Yamada3, Hidetaka Uramoto2. 1. Department of Thoracic Surgery, Kanazwa Medical University, 1-1 Daigaku, Uchinada-machi, Kahoku-gun, 920-0293, Ishikawa, Japan. s7006@nms.ac.jp. 2. Department of Thoracic Surgery, Kanazwa Medical University, 1-1 Daigaku, Uchinada-machi, Kahoku-gun, 920-0293, Ishikawa, Japan. 3. Department of Pathology and Laboratory Medicine, Kanazwa Medical University, Ishikawa, Japan. 4. Department of Thoracic Surgery, Tomei Atsugi Hospital, Kanagawa, Japan. 5. Department of Thoracic Surgery, Aidu Chuo Hospital, Fukushima, Japan.
Abstract
BACKGROUND: Immunoglobulin G4 (IgG4)-related diseases are characterized by abnormal IgG4 levels, swelling, and marked infiltration and fibrosis of the lymphocytes and IgG4-positive plasma cells, causing hypertrophic lesions or nodules. The cause is currently not well understood. IgG4-related diseases involving lesions limited to the pleura are extremely rare. Herein, we report an IgG4-related disease presenting with multiple pleural nodules confirmed by thoracoscopic surgical biopsy. CASE PRESENTATION: A 74 year-old man was referred to our department for definitive diagnosis of multiple pleural nodules after 1 year of follow-up. Computed tomography of the chest revealed multiple pleural nodules, while 2-deoxy-2-( 18F)-fluorodeoxyglucose positron emission tomography imaging exhibited tracer accumulation in the nodules. A thoracoscopic surgical biopsy was performed. Histopathological examination revealed hyalinized fibrous tissue with a high degree of plasma cell-based inflammatory cell infiltration. Immunohistochemically, IgG4-positive cells were conspicuous, accounting for 70.5% of the plasma cells. The postoperative serum IgG4 concentration was 289 mg/dL. We diagnosed the patient with an IgG4-related disease with multiple pleural nodules. The postoperative course was good, and the patient is currently being followed up. CONCLUSION: IgG4-related disease should be considered in cases presenting with multiple pleural nodules.
BACKGROUND: Immunoglobulin G4 (IgG4)-related diseases are characterized by abnormal IgG4 levels, swelling, and marked infiltration and fibrosis of the lymphocytes and IgG4-positive plasma cells, causing hypertrophic lesions or nodules. The cause is currently not well understood. IgG4-related diseases involving lesions limited to the pleura are extremely rare. Herein, we report an IgG4-related disease presenting with multiple pleural nodules confirmed by thoracoscopic surgical biopsy. CASE PRESENTATION: A 74 year-old man was referred to our department for definitive diagnosis of multiple pleural nodules after 1 year of follow-up. Computed tomography of the chest revealed multiple pleural nodules, while 2-deoxy-2-( 18F)-fluorodeoxyglucose positron emission tomography imaging exhibited tracer accumulation in the nodules. A thoracoscopic surgical biopsy was performed. Histopathological examination revealed hyalinized fibrous tissue with a high degree of plasma cell-based inflammatory cell infiltration. Immunohistochemically, IgG4-positive cells were conspicuous, accounting for 70.5% of the plasma cells. The postoperative serum IgG4 concentration was 289 mg/dL. We diagnosed the patient with an IgG4-related disease with multiple pleural nodules. The postoperative course was good, and the patient is currently being followed up. CONCLUSION: IgG4-related disease should be considered in cases presenting with multiple pleural nodules.
Authors: John P Corcoran; Emma L Culver; Rebekah M Anstey; Ambika Talwar; Charis D Manganis; Tamsin N Cargill; Robert J Hallifax; Ioannis Psallidas; Najib M Rahman; Eleanor Barnes Journal: Respir Med Date: 2017-10-10 Impact factor: 3.415