A validated method of labial minor salivary gland biopsy for the diagnosis of Sjögren's syndrome : Method of LSGB for the Diagnosis of SS

To analyze the safety of our biopsy technique and the effectiveness of minor salivary gland biopsy (MSGB) for the diagnosis of Sjögren's syndrome (SS) and amyloidosis. We conducted a retrospective analysis of 452 patients with suspected SS and 50 with suspected amyloidosis and negative periumbilical fat aspiration analysis who underwent MSGB at a single center. Diagnostic evaluation for SS included Schirmer's test, unstimulated whole salivary flow, detection of antinuclear antibodies and anti-SSA/SSB, erythrocyte sedimentation rate, C-reactive protein, IgM rheumatoid factor, and serology for hepatitis C virus. For all biopsy samples, a cumulative focus score on multilevel sections was calculated. SS was diagnosed according to American-European Consensus Group (AECG) criteria. Histologic evaluation for amyloidosis was performed using Congo red staining and polarized-light microscopy. Adverse events were recorded on a questionnaire immediately after the procedure and 7 days, 14 days, and 6 months thereafter. Sixty-four patients (12.7%) reported transient adverse events: 40 paresthesias lasting or =1, and 87 (94.5%) of 93 satisfied the AECG criteria. Classification of SS was possible for 124 (32.8%) of 378 patients. In 51 (41%) of 124, MSGB was essential to reach the number of criteria needed for classification. Of 50 patients evaluated for amyloidosis, 10 (20%) had positive Congo red staining. MSGB is a simple, safe, and reliable tool for the diagnosis of SS and amyloidosis, and therefore is suitable for more extensive application.

This study proposes a revision of the literature on the current techniques employed in salivary gland biopsy. A systematic review of the literature between January 1990 and January 2010 was conducted using MEDLINE, Embase and the Cochrane Central Register of Controlled Trials. The search terms were: 'biopsy AND parotid AND Sjögren'; 'biopsy AND sublingual salivary gland AND Sjögren'; 'biopsy AND minor salivary gland AND Sjögren'; 'biopsy AND labial salivary gland AND Sjögren' and 'biopsy AND salivary glands AND connective disorders'. No study reporting submandibular salivary gland biopsy was found; 3 studies reported sublingual salivary gland biopsy; 1 study reported palate biopsy; 4 studies reported parotid gland biopsy and 21 studies reported minor salivary gland biopsy. Biopsy of salivary glands must be performed as last investigation and only when the other items are not complete enough to satisfy the diagnosis. The knowledge of complications and sequelae may be useful in order to minimize the risk.