Unusual Involvement of IgG4-Related Sclerosing Disease in Lacrimal and Submandibular Glands and Extraocular Muscles.
10.3341/kjo.2012.26.3.216
- Author:
Yong Un SHIN
1
;
Young Ha OH
;
Yoon Jung LEE
Author Information
1. Department of Ophthalmology, Hanyang University College of Medicine, Seoul, Korea.
- Publication Type:Case Reports
- Keywords:
Extraocular muscles;
IgG4-related sclerosing disease;
Kuttner tumor;
Lacrimal apparatus
- MeSH:
Biopsy, Fine-Needle;
Diagnosis, Differential;
Facial Muscles/*immunology/pathology/radiography;
Follow-Up Studies;
Humans;
Immunoglobulin G/*immunology/metabolism;
Immunohistochemistry;
Lacrimal Apparatus/*immunology/metabolism/pathology;
Lacrimal Duct Obstruction/complications/diagnosis/*immunology;
Magnetic Resonance Imaging;
Male;
Middle Aged;
Sclerosis;
Submandibular Gland/*immunology/pathology/radiography;
Submandibular Gland Diseases/complications/diagnosis/*immunology;
Tomography, X-Ray Computed
- From:Korean Journal of Ophthalmology
2012;26(3):216-221
- CountryRepublic of Korea
- Language:English
-
Abstract:
Chronic sclerosing sialadenitis, also known as Kuttner tumor, is a chronic inflammatory disease of the salivary glands that is reported in a few cases in medical literature. Recent reports suggest that certain aspects of sclerosing diseases, including chronic sclerosing sialadenitis or dacryoadenitis, should be classified under immunoglobulin G4 (IgG4)-related sclerosing disease based on immunohistochemical studies. This study reports an unusual case of IgG4-related sclerosing disease appearing simultaneously in the lacrimal glands, submandibular glands, and extraocular muscles. A 56-year-old male presented with complaints of bilateral eyelid swelling and proptosis that began two years ago. Computed tomography confirmed that bilateral submandibular enlargements also existed five years ago in the subject. Orbital computed tomography and magnetic resonance imaging revealed bilateral lacrimal gland enlargement and thickening of extraocular muscles. Typical findings of chronic sclerosing dacryoadenitis were revealed upon pathologic exam of the right lacrimal gland. Immunostaining revealed numerous IgG4-positive plasma cells. Through these clinical features, we make a diagnosis of IgG4-relataed sclerosing disease in the subject.
