IgG4-related disease presenting as otogenic skull base osteomyelitis |
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| Authors: | Chae Dong Yim Hyo Jung An Seong Ki Ahn Dong Gu Hur Hyun-Jin Lee |
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| Affiliation: | 1. Department of Otorhinolaryngology, Gyeongsang National University Changwon Hospital, Changwon, South Korea;2. Department of Pathology, Gyeongsang National University Changwon Hospital, Changwon, South Korea;3. Department of Otorhinolaryngology, Gyeongsang National University Hospital, Gyeongsang National University College of Medicine, Jinju, Korea;1. Department of Otolaryngology - Head and Neck Surgery, University of Modena and Reggio Emilia, University Hospital of Modena, Via del Pozzo 71, Modena, Italy;2. PhD Program in Clinical and Experimental Medicine, University of Modena and Reggio Emilia, IRCCS - Arcispedale Santa Maria Nuova, Viale Risorgimento 80, Reggio Emilia, Italy;3. Department of Otolaryngology Head and Neck Surgery, Ospedale Maggiore, Largo Nigrisoli 2, Bologna, Italy;1. Department of Otolaryngology Head and Neck Surgery, Kurashiki Central Hospital, Miwa 1-1-1, Kurashiki, Okayama 710-8602, Japan;2. Kurashiki Clinical Research Institute, Miwa 1-1-1, Kurashiki, Okayama 710-8602, Japan;1. Department of Otorhinolaryngology-Head and Neck Surgery, Pusan National University Hospital, Busan, Republic of Korea;2. Department of Otorhinolaryngology-Head and Neck Surgery, The Dongnam Institute of Radiological and Medical Sciences (DIRAMS), Busan, Republic of Korea;3. Department of Otorhinolaryngology-Head and Neck Surgery, Dong-A University College of Medicine, Busan, Republic of Korea;4. Department of Otorhinolaryngology-Head and Neck Surgery, Seoul National University Bundang Hospital, Seoul National University College of Medicine, 300 Gumi‐dong, Bundang‐gu, Seongnam 13620, Republic of Korea;1. Departments of Otolaryngology, Japanese Red Cross Society Himeji Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan;2. Department of Otolaryngology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-cho, Kita-ku, Okayama City, Okayama 700-8558, Japan;3. Departments of Pathology, Japanese Red Cross Society Himeji Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan;4. Departments of Inspection technology, Japanese Red Cross Society Himeji Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan;1. Tianjin Medical University General Hospital, An Shan Road No.154, Heping District, Tianjin 300192, China;2. Tianjin First Center Hospital, Fukang Road No. 24, Nankai District, Tianjin 300192, China |
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| Abstract: | IgG4-related disease (IgG4-RD) is an emerging clinical disease entity characterized by tumefactive lesions at multiple sites with a dense lymphoplasmacytic infiltrate rich in IgG4+ plasma cells. Although almost any organ can be affected, IgG4-RD is most likely to involve the submandibular, lacrimal, or parotid glands in the head and neck region. However, skull base involvement presenting as otogenic skull base osteomyelitis (SBO) is rare. We encountered a 70-year-old male with IgG4-RD presenting primarily with severe otalgia and otorrhea. He had uncontrolled diabetes mellitus and showed clinical manifestations of otogenic SBO. Tissue immunostaining revealed typical features of increased IgG4-positive plasma cells, and hematological examination showed elevated serum IgG4 concentrations. Treatment with corticosteroids significantly improved well-being and partially resolved the lesion based on computed tomography (CT) scan. |
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