Diagnostic value of minor salivary gland biopsy for Sjögren's syndrome in children: A monocentric retrospective study over 10years

Abstract

Objectives: Minor salivary gland biopsy (MSGB) is a minimally invasive test used in suspected autoimmune diseases. Our study goal was to evaluate diagnostic performance of MSGB in children suspected of Sjögren's syndrome (SS).

Methods: We did a retrospective monocentric study on patients under 18years old who had a MSGB between October 2011 and December 2021 at Bicêtre University Hospital. Biopsy was considered positive if focus score was positive or Chisholm-Mason score was superior to 2.

Results: Sixty-six MSGB were analysed in SS indication, 23/66 (35%) were positive and 13/23 (57%) were associated with a confirmed final diagnosis. MSGB was sensitive and specific (Se=81%, Sp=80), but with low positive and negative predictive value (57% and 7%). Twenty of 64 MSGB also had positive anti-SSA antibodies. Anti-SSA antibodies improved MSGB specificity (98%).

Conclusion: MSGB demonstrates good sensitivity and specificity, but the predictive values and likelihood ratios make it a poor screening test. However, when combined with other criteria such as the presence of anti-SSA antibodies, the specificity of MSGB is improved. A larger prospective study should be carried out to support our results and clarify the good indications for MSGB.

Keywords: Minor salivary gland biopsy; Paediatric rheumatology; Sjögren's syndrome.