Background/Purpose: Sublingual frenulum abnormalities are reported in systemic sclerosis (SSc), but the exact nature and clinical relevance of such abnormalities is unknown.  Laser speckle contrast imaging (LSCI) is an emerging method for assessing tissue perfusion but has not been applied to the gastrointestinal tract (GIT) in SSc. We report a feasibility study evaluating clinical associations and inter-rater agreement using a novel scoring chart to assess sublingual abnormalities in SSc patients and healthy controls (HC). We also report preliminary findings and associations following objective assessment of sublingual perfusion using LSCI in SSc.

Methods: Ten patients fulfilling 2013 ACR/EULAR classification criteria for SSc and 8 HC were recruited from two different SSc clinics. SSc patients completed the GIT 2.0 questionnaire and baseline demographics were documented. A 0-2 Likert scale was used to assess sublingual frenulum thickness, frenulum length, mucosa pallor and the presence of sublingual and labial telangiectases.  A composite index of scores for each domain was calculated for each subject. Photographs of the sublingual frenulum were obtained under standardised conditions to allow blinded assessment by 2 separate assessors of sublingual abnormalities without knowledge of GIT symptoms. Baseline perfusion (in arbitrary flux units [fu]) of the frenulum was assessed using LSCI in subjects with SSc.

Results: Each of the frenulum measures differed between HC and SSc (Mann-Whitney U, p =0.0003).  No HC had a composite score of greater than 2.  Positive associations were identified between clinical frenulum assessment (rater 1), domains of GIT 2.0, disease duration and LSCI perfusion in SSc.  Inter-rater agreement for individual domains of frenulum scoring chart and composite index revealed moderate agreement for assessment of frenulum (Table 1) with a weighted kappa for the total score is 0.53 (moderate agreement). When LSCI was applied to the frenulum, the perfusion index was 2308 fu (1810-3203).  The perfusion index correlated negatively with disease duration, -0.64 (p < 0.05) and with social component of the GIT 2.0, –0.67 (p =0.05).

Conclusion: This feasibility study confirms the presence of clinical sublingual abnormalities in SSc, which can be categorised using a simple scoring chart with moderate inter-rater agreement. Perfusion of the sublingual frenulum can be assessed by LSCI and may correlate with disease duration and GIT 2.0 self-report. Efforts to refine image capture and scoring shall be undertaken to improve blinded inter-rater agreement before embarking on a larger study exploring the significance of clinical and LSCI perfusion sublingual abnormalities in SSc.

Table 1: Frenulum Assessment

 

	Score			Inter-rater Agreement Cohen’s Kappa (95% CI)
Parameter	0	1	2	Rater 1 vs 2	Rater 1 vs 3	Rater 2 vs 3
Sublingual frenulum thickness	Normal	Intermediate thickening	Severe thickening	0.64 (0.28-1.0)	1.0 (1.0-1.0)	0.74 (0.40-1.0)
Sublingual frenulum       length	Normal	Intermediate shortening	Severe Shortening	0.60 (0.28- 0.64)	0.89 (0.68- 1.0)	0.44 (.13-0.75 )
Sublingual mucosa pallor	Normal	Intermediate pallor	Severe pallor	0.56 (0.11-0.99)	0.76 (0.47-1.0)	0.42 (0.06-0.77)
Buccal and labial telangiectases	None visible	1-6 visible	>7 visible	0.53 (0.28-0.79)	1.0 (1.0-1.0)	0.55 (0.11-0.99)
Total Score in 4 categories 0 1-3 3-5 >5				0.53 (0.28-0.79)	1.0 (1.0-1.0)	0.62 (0.37-0.87)

---

« Back to 2014 ACR/ARHP Annual Meeting

ACR Meeting Abstracts - https://acrabstracts.org/abstract/a-feasibility-study-of-subjective-and-objective-assessment-of-sublingual-abnormalities-in-systemic-sclerosis/