Abstract
PurposeTo determine whether the severity of salivary-gland involvement, assessed using salivary gland ultrasonography [SGUS], histological focus score, or the unstimulated whole salivary flow [UWSF], was associated with the response to rituximab in patients with primary Sjögren’s syndrome [pSS].Materials and MethodsAmong the 120 patients with pSS enrolled in the randomised TEARS trial of rituximab versus placebo, 35 underwent either centralised minor salivary-gland biopsy or SGUS at inclusion. The echostructure of each parotid and submandibular gland was graded on a scale of 0 to 4. Histologic minor salivary gland involvement was assessed by the focus score. Among rituximab-treated patients with available data (n = 14), half met the Sjögren’s Syndrome Responder Index [SSRI]-30 definition of a response at week 24.ResultsThe SGUS score correlated positively to the focus score [r = 0.61] and negatively to the UWSF [r = -0.68]. The focus score was not correlated to the UWSF. The median total SGUS grade at inclusion was 9 [6-11] in responders versus 16 [11-16] in non-responders [p = 0.04]. The proportion of SSRI-30 responders was 0% among patients with SGUS grade 4 and 88% among those with SGUS grade ≤3. Low baseline SGUS scores were associated with sicca-related outcomes improvement, but not with fatigue or biological improvement. Median baseline focus score was 0.3 [0.0–1.3] in the responders versus 4.0 [2.7–5.3] in the non-responders [p = 0.02]. Baseline UWSF was not associated with the response rate.ConclusionIn patients with pSS, the highest SGUS grade or a high histological focus score is associated with absence of a response to a single rituximab course after 6 months. Further studies, including more patients and different treatment strategies, are required to confirm the clinical utility of these potential biomarkers in pSS.
Highlights
Primary Sjögren’s syndrome [pSS] is a systemic autoimmune disease clinically characterised by intense fatigue and by oral and ocular dryness, reflecting lymphocytic infiltration and subsequent dysfunction of the exocrine glands
Among the 120 patients with pSS enrolled in the randomised TEARS trial of rituximab versus placebo, 35 underwent either centralised minor salivary-gland biopsy or SGUS at inclusion
In patients with pSS, the highest SGUS grade or a high histological focus score is associated with absence of a response to a single rituximab course after 6 months
Summary
Primary Sjögren’s syndrome [pSS] is a systemic autoimmune disease clinically characterised by intense fatigue and by oral and ocular dryness, reflecting lymphocytic infiltration and subsequent dysfunction of the exocrine glands. The larger TEARS trial [6] failed to detect a therapeutic effect of rituximab, indicating either lack of efficacy of this agent or low sensitivity to change of the primary endpoint. We recently developed the Sjögren’s Syndrome Response Index (SSRI)-30, a data-driven composite index of treatment response including symptoms (oral and ocular dryness and fatigue) and objective measurements (salivary flow and erythrocyte sedimentation rate) [7]. In the TEARS trial, about half the rituximab-treated patients achieved the SSRI-30 versus 10–20% in the placebo arm [7]
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