The diagnostic utility of submandibular gland sonography and labial salivary gland biopsy in IgG4-related dacryoadenitis and sialadenitis: Its potential application to the diagnostic criteria

Abstract

Objectives: In this study, we investigated the diagnostic utility of submandibular gland (SMG) sonography and labial salivary gland (LSG) biopsy as a less invasive procedure for diagnosing IgG4-related dacryoadenitis and sialadenitis (IgG4-DS)

Methods: Sixty-eight patients with suspected IgG4-DS by presenting swelling of elevated serum IgG (>1747 mg/dl) and/or swelling glands underwent SMG sonography, LSG biopsy and measurement for serum IgG4. SMG sonographic diagnosis was determined by the following characteristic changes; ‘hypoechoic areas of a nodal pattern with high vascularity’ and/or ‘hypoechoic areas of a reticular pattern in the superficial part’.

Results: Thirty-one patients were diagnosed with IgG4-DS, 5 with IgG4-RD unaccompanied by lacrimal and salivary gland lesions, 28 with Sjögren’s syndrome, and 4 with malignant lymphoma. The sensitivity, specificity, and accuracy of SMG sonography and LSG biopsy were 100%, 83.8%, 91.2% and 64.5%, 73.8%, 75.0%, respectively. Moreover, those of SMG sonography and LSG biopsy combined with serum IgG4 concentration (>135 mg/dl) were 100%, 94.6%, 97.1% and 64.5%, 91.9%, 79.4%, respectively.

Conclusion: LSG biopsy needs to be extremely careful to diagnose IgG4-DS because of its low sensitivity. SMG sonography is sufficient for the diagnosis of IgG4-DS, especially when combined with serologic analysis. Thus, SMG sonography could adapt to the diagnostic criteria of IgG4-DS as a non-invasive method.

Keywords:

• IgG4-related dacryoadenitis and sialadenitis
• submandibular gland sonography
• labial salivary gland biopsy
• diagnostic criteria

Acknowledgements

We thank Helen Jeays, BDSc AE, from Edanz Group (www.edanzediting.com/ac) for editing a draft of this manuscript.

Conflict of interest

None.

Additional information

Funding

This work was supported in part by grants from the Ministry of Education, Culture, Sports, Science, and Technology of Japan (18K09747, 17H04671, 17H01603, 26293430, 15H06491), the ‘Research on Measures for Intractable Diseases’ project, a matching fund subsidy from the Ministry of Health, Labour and Welfare, Japan (H26-026, H26-064), the ‘Practical Research Project for Rare/Intractable Diseases’, and a matching fund subsidy from the Japan Agency for Medical Research and Development (H29-058).