Journal of Otolaryngology Head & Neck Surgery Category: Clinical Type: Case Report
A Case of IgG4-Related Mikulicz’s Disease with Chronic Rhinosinusitis and Elevated Levels of IgG4-and IgE-Immune Complexes
- Yasushi Ota1*, Mutsunori Fujiwara2, Hirabayashi Yoji3, Rumi Ota4, Mitsuya Suzuki1
- 1 Department Of Otorhinolaryngology, Toho University, Tokyo, Japan
- 2 Department Of Pathology, Nissan Tamagawa Hospital, Tokyo, Japan
- 3 Clinical Laboratory Testing Division, Toho University Sakura Medical Center, Chiba, Japan
- 4 Okamura Otorhinolaryngology Clinic, Hiroshima, Japan
*Corresponding Author:Yasushi Ota
Department Of Otorhinolaryngology, Toho University, Tokyo, Japan
Received Date: Feb 22, 2018 Accepted Date: Apr 14, 2018 Published Date: Apr 27, 2018
We report a case of male IgG4-related Mikulicz’s disease with chronic rhinosinusitis with high serum IgG4-and IgE-Immune Complex (IC). Untreated serum IgG4 was 3,110 mg/dL, while Polyethylene Glycol (PEG)-treated serum IgG4-IC was 1,460mg/dL. Both were markedly elevated (normal IgG4 range <105 mg/dL). Serum IgE and PEG-treated serum IgE-IC was also elevated (normal IgE range <170 IU/mL) at 376 IU/mL and 237 IU/mL, respectively. IgE PEG Precipitated (PP) index was higher than normal (<20%) at 63%. We speculate that serum IgG4 and IgE represented autoantibodies for a component of the paranasal sinus mucosa and submandibular gland tissue.
Figure 1: Computed Tomography (CT) of the paranasal sinuses and neck revealed bilateral sinusitis (black arrow heads), lacrimal gland swelling (white arrow heads) and bilateral submandibular glands (white arrows).
Figure 2: Histopathological findings revealed that most lymphocytes and plasma cells that had invaded the paranasal lamina propria, as well as epithelial cells in the parasinus mucosa and lymphocytes and plasma cells that had invaded the submandibular gland were strongly positive for anti-IgG4 antibody. Some mast cells and some of the stroma in the paranasal lamina propria and submandibular gland were positive for anti-IgE antibody (black arrows).
MEASUREMENT OF ANTIGEN-SPECIFIC IGG4 AND IGE ANTIBODIES IN THE SERUM
|sIgG4 (mg/dL)||sIgE (Iµ/mL)|
|Malassezia furfur (pityrosporum orbiculare)||0.5514||<0.10|
|Enterotoxin A (S.aureus)||0.8355||<0.10|
|Enterotoxin B (S.aureus)||0.9488||<0.10|
ASSESSMENT OF IMMUNE COMPLEXES IN THE SERUM
|Serum mg/dL||Serum mg/dL||Serum mg/dL||Serum mg/dL||%|
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Citation:Ota Y, Fujiwara M, Yoji H, Ota R, Suzuki M (2018) A Case of IgG4-Related Mikulicz’s Disease with Chronic Rhinosinusitis and Elevated Levels of IgG4-and IgE-Immune Complexes. J Otolaryng Head Neck Surg 4: 019.
Copyright: © 2018 Yasushi Ota, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.