The diagnostic utility of biopsies from the submandibular and labial salivary glands in IgG4-related dacryoadenitis and sialoadenitis, so-called Mikulicz's disease

Abstract IgG4-related dacryoadenitis and sialoadenitis (IgG4-DS) is characterized by serum IgG4 elevation and the infiltration of IgG4-positive plasma cells in glandular tissues. For definitive diagnosis of IgG4-DS, biopsies of local lesions are recommended to exclude Sjögren's syndrome (SS), m...

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Published in:International journal of oral and maxillofacial surgery 2014-10, Vol.43 (10), p.1276-1281
Main Authors: Moriyama, M, Furukawa, S, Kawano, S, Goto, Y, Kiyoshima, T, Tanaka, A, Maehara, T, Hayashida, J.-N, Ohta, M, Nakamura, S
Format: Article
Language:English
Subjects:
Aged
Biopsy
Dentistry
Female
Humans
IgG4-related dacryoadenitis and sialoadenitis
IgG4-related disease
Immunoglobulin G - immunology
labial salivary gland biopsy
Male
Middle Aged
Mikulicz' Disease - immunology
Mikulicz' Disease - pathology
Mikulicz's disease
Salivation
Sensitivity and Specificity
submandibular gland biopsy
Surgery
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Summary:Abstract IgG4-related dacryoadenitis and sialoadenitis (IgG4-DS) is characterized by serum IgG4 elevation and the infiltration of IgG4-positive plasma cells in glandular tissues. For definitive diagnosis of IgG4-DS, biopsies of local lesions are recommended to exclude Sjögren's syndrome (SS), malignant tumours, and similar disorders. In this study, we examined the diagnostic utility of submandibular gland (SMG) and labial salivary gland (LSG) biopsies in IgG4-DS. Fourteen patients presenting with swelling of the SMG (eight females and six males) underwent both SMG and LSG biopsies. The sensitivity, specificity, and accuracy of SMG biopsies were all 100.0%. In contrast, those of LSG biopsies were 69.2%, 100.0%, and 71.4%, respectively. Thirty-three out of 61 LSG biopsies (54.1%) from all 14 patients were positive for the diagnostic criteria of IgG4-DS (IgG4-positive/IgG-positive plasma cells >0.4). None of the patients experienced complications such as facial nerve palsy, sialocele, or hyposalivation. The IgG4/IgG ratio showed no significant correlation between the LSG and SMG. The final diagnosis was IgG4-DS in 13 patients and marginal zone B-cell lymphoma (MZL) in one. These results suggest that incisional biopsy of the SMG is useful and appropriate for the definitive diagnosis of IgG4-DS, while diagnosis by LSG biopsy alone requires more caution.
ISSN:0901-5027
1399-0020
DOI:10.1016/j.ijom.2014.06.014