Intraoperative Visualization and Treatment of Salivary Gland Dysfunction in Sjögren’s Syndrome Patients Using Contrast-Enhanced Ultrasound Sialendoscopy (CEUSS)
Abstract
:1. Introduction
2. Materials and Methods
2.1. Study Setting and Ethics
2.2. Patient and Public Involvement
2.3. Participants and Eligibility Criteria
2.4. Intervention
2.5. Outcomes
2.6. Participant Timeline
2.7. Data Collection Methods
2.7.1. Saliva Flow Rates and Analysis
2.7.2. Xerostomia Inventory (XI) Score
2.7.3. European League against Rheumatism (EULAR) Sjögren’s Syndrome Patient Reported Index (ESSPRI)
2.7.4. Clinical Oral Dryness Score (CODS)
2.7.5. Pain Score
2.7.6. Evaluation of the Major Salivary Glands by US
2.8. Sample Size Analysis and Statistical Methods
3. Results
3.1. Primary Outcome Measures
3.2. Secondary Outcome Measures
4. Discussion
5. Conclusions
Supplementary Materials
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
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Enrolment | Baseline Assessment | Intervention Post-Intervention Assessment | |||||
---|---|---|---|---|---|---|---|
Timepoint | T-6 | T-4 | T0 | T1 | T2 | T8 | T16 (Closeout) |
Enrolment: | |||||||
Eligibility screen | X | ||||||
Informed consent | X | ||||||
Intervention: | |||||||
CEUSS | X | ||||||
Assessments: | |||||||
Safety | X | X | X | X | |||
Technical feasibility and applicability | X | ||||||
UWSF, SWSF | X | X | X | X | X | X | |
CODS | X | X | X | X | X | ||
XI | X | X | X | X | X | ||
ESSPRI | X | X | X | X | X | ||
MPQ | X | X | X | X | X | ||
Salivary gland topography with SGUS | X | X | X |
Mean (S.D.) | Median (IQR) | ||
---|---|---|---|
Patient variables | Age, years | 47.7 (18.7) | 49.5 (28.5–62.5) |
Female gender, n | 10 | ||
Disease variables | Disease duration, years a | 8.3 (8.4) | 4.0 (2.0–17.5) |
Primary SS, n b | 4 | ||
Secondary SS, n b | 6 | ||
Positive salivary gland biopsy, n | 4 | ||
Autoantibodies to anti-SSA, n | 7 | ||
Baseline measurements | UWSF (mL/min) | 0.13 (0.09) | 0.10 (0.07–0.22) |
pSS | 0.12 (0.06) | 0.09 (0.15–0.50) | |
sSS | 0.15 (0.11) | 0.16 (0.03–0.25) | |
SWSF (mL/min) | 0.50 (0.43) | 0.41 (0.18–0.75) | |
pSS | 0.39 (0.27) | 0.32 (0.17–0.67) | |
sSS | 0.59 (0.52) | 0.49 (0.15–0.97) | |
XI Score | 45.20 (3.55) | ||
pSS | 45.75 (3.86) | ||
sSS | 44.83 (3.66) | ||
ESSPRI (all domains) c | 21.5 (5.3) | ||
pSS | 17.8 (5.1) | ||
sSS | 21.5 (5.3) | ||
ESSPRI (dryness domain) | 7.8 (1.6) | ||
pSS | 7.5 (1.7) | ||
sSS | 8.0 (1.7) | ||
CODS | 4.0 (1.6) | ||
pSS | 4.5 (1.3) | ||
sSS | 3.8 (1.7) | ||
Hoçevar score | 20.4 (11.9) | 21.5 (7.0–29.5) | |
pSS | 21.8 (13.0) | 24.5 (8.3–32.5) | |
sSS | 19.5 (13.3) | 20.0 (7.0–30.3) | |
Pain score | 38.4 (32.8) | ||
pSS | 9.3 (12.3) | ||
sSS | 57.8 (26.7) |
Median (IQR) | Mean (S.D.) | p Value (Compared to Baseline) | ||
---|---|---|---|---|
UWSF (mL/min) All patients n = 10 | T-4 | 0.10 (0.07–0.22) | 0.13 (0.09) | -- |
T1 | 0.18 (0.06–0.35) | 0.26 (0.29) | 0.037 * | |
T2 | 0.22 (0.10–0.49) | 0.29 (0.24) | 0.007 * | |
T8 | 0.22 (0.15–0.50) | 0.38 (0.44) | 0.028 * | |
T16 | 0.24 (0.09–0.40) | 0.26 (0.21) | 0.074 | |
UWSF (mL/min) Primary SS n = 4 | T-4 | 0.09 (0.08–0.17) | 0.12 (0.06) | -- |
T1 | 0.10 (0.05–0.27) | 0.14 (0.13) | 0.72 | |
T2 | 0.18 (0.12–0.37) | 0.22 (0.14) | 0.07 | |
T8 | 0.20 (0.08–0.37) | 0.22 (0.16) | 0.14 | |
T16 | 0.18 (0.08–0.34) | 0.20 (0.14) | 0.47 | |
UWSF (mL/min) Secondary SS n = 6 | T-4 | 0.16 (0.03–0.25) | 0.15 (0.11) | -- |
T1 | 0.28 (0.06–0.57) | 0.34 (0.34) | 0.028 * | |
T2 | 0.25 (0.08–0.69) | 0.33 (0.30) | 0.046 * | |
T8 | 0.27 (0.15–0.91) | 0.49 (0.54) | 0.12 | |
T16 | 0.26 (0.10–0.54) | 0.30 (0.25) | 0.12 | |
SWSF (mL/min) All patients n = 10 | T-4 | 0.41 (0.18–0.75) | 0.50 (0.43) | -- |
T1 | 0.60 (0.20–1.15) | 0.70 (0.57) | 0.06 | |
T2 | 0.55 (0.24–0.93) | 0.68 (0.55) | 0.07 | |
T8 | 0.61 (0.27–0.97) | 0.73 (0.64) | 0.047 * | |
T16 | 0.66 (0.17–0.84) | 0.66 (0.52) | 0.33 | |
SWSF (mL/min) Primary SS n = 4 | T-4 | 0.32 (0.17–0.67) | 0.39 (0.27) | -- |
T1 | 0.26 (0.16–0.61) | 0.34 (0.26) | 0.47 | |
T2 | 0.36 (0.16–0.46) | 0.33 (0.16) | 0.72 | |
T8 | 0.35 (0.19–0.59) | 0.38 (0.22) | 0.72 | |
T16 | 0.35 (0.15–0.66) | 0.40 (0.27) | 1.00 | |
SWSF (mL/min) Secondary SS n = 6 | T-4 | 0.49 (0.15–0.97) | 0.59 (0.52) | -- |
T1 | 0.83 (0.44–1.61) | 0.93 (0.62) | 0.028 * | |
T2 | 0.78 (0.51–1.50) | 0.92 (0.61) | 0.028 * | |
T8 | 0.68 (0.48–1.76) | 0.97 (0.73) | 0.028 * | |
T16 | 0.70 (0.49–1.37) | 0.85 (0.59) | 0.25 |
Mean (S.D.) | p Value (Compared to Baseline) | 95% Ci | |||
---|---|---|---|---|---|
Upper Limit | Lower Limit | ||||
Xerostomia Inventory | T-4 | 45.20 (3.55) | -- | -- | -- |
T1 | 37.00 (8.33) | 0.04 * | 0.31 | 16.10 | |
T2 | 37.80 (7.57) | 0.031 * | 0.57 | 14.23 | |
T8 | 34.40 (7.027) | 0.003 * | 3.89 | 17.71 | |
T16 | 34.20 (8.84) | 0.021 * | 1.49 | 20.51 | |
ESSPRI (all domains) | T-4 | 21.50 (5.26) | -- | -- | -- |
T1 | 16.30 (4.22) | 0.018 * | 0.81 | 9.59 | |
T2 | 17.10 (5.69) | 0.001 * | 1.93 | 6.87 | |
T8 | 16.80 (4.76) | 0.044 * | 0.10 | 9.30 | |
T16 | 16.80 (5.63) | 0.10 | −0.66 | 10.06 | |
ESSPRI (dryness domain) | T-4 | 7.80 (1.62) | -- | -- | -- |
T1 | 5.80 (2.44) | 0.34 | −0.96 | 4.96 | |
T2 | 4.80 (2.35) | 0.015 * | 0.54 | 5.46 | |
T8 | 4.50 (2.01) | 0.015 * | 0.60 | 6.00 | |
T16 | 3.70 (1.83) | 0.001 * | 1.74 | 6.46 | |
Clinical Oral Dryness | T-4 | 4.00 (1.58) | -- | -- | -- |
T1 | 3.33 (1.23) | 0.22 | −0.24 | 1.57 | |
T2 | 2.33 (1.00) | 0.011 * | 0.39 | 2.94 | |
T8 | 1.89 (0.60) | 0.017 * | 0.37 | 3.85 | |
T16 | 1.78 (0.67) | 0.036 * | 0.13 | 4.32 | |
VAS pain score | T-4 | 38.4 (32.8) | -- | -- | -- |
T1 | 40.8 (32.0) | 1.00 | −41.55 | 36.75 | |
T2 | 48.0 (34.3) | 1.00 | −48.02 | 28.82 | |
T8 | 44.2 (35.1) | 1.00 | −46.03 | 34.43 | |
T16 | 34.5 (31.7) | 1.00 | −17.77 | 25.57 |
Median (IQR) | Mean (S.D.) | p Value (Compared to Baseline) | ||
---|---|---|---|---|
Total Hoçevar score | T-4 | 21.5 (7.0–29.5) | 20.4 (11.9) | -- |
T1 | 22.0 (6.0–27.5) | 19.1 (11.4) | 0.29 | |
T16 | 20.0 (6.0–29.0) | 19.3 (11.8) | 0.20 | |
Parenchymal echogenicity | T-4 | 0.5 (0.0–2.3) | 1.2 (1.5) | -- |
T1 | 0.5 (0.0–2.3) | 1.1 (1.5) | 0.66 | |
T16 | 0.5 (0.0–2.3) | 1.2 (1.6) | 1.0 | |
Homogeneity | T-4 | 5.5 (2.3–10.0) | 6.1 (3.5) | -- |
T1 | 6.0 (2.9–8.75) | 6.0 (3.5) | 0.71 | |
T16 | 5.5 (2.0–8.8) | 6.0 (3.7) | 0.71 | |
Presence of hypo-echogenic areas | T-4 | 8.0 (2.3–12.0) | 7.6 (4.5) | -- |
T1 | 8.0 (2.0–11.0) | 7.3 (4.2) | 0.32 | |
T16 | 7.5 (2.0–11.0) | 7.3 (4.1) | 0.32 | |
Hyper-echogenic reflections | T-4 | 4.0 (0.0–5.0) | 3.1 (2.3) | -- |
T1 | 2.5 (0.0–4.3) | 2.3 (2.2) | 0.11 | |
T16 | 2.0 (0.0–4.3) | 2.3(2.5) | 0.11 | |
Clearness of the salivary gland border | T-4 | 2.0 (0.0–4.3) | 2.4 (2.1) | -- |
T1 | 2.0 (0.0–4.3) | 2.4 (2.2) | 1.00 | |
T16 | 2.0 (0.0–4.5) | 2.5 (2.3) | 0.32 |
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Karagozoglu, K.H.; Mahraoui, A.; Bot, J.C.J.; Cha, S.; Ho, J.-P.T.F.; Helder, M.N.; Brand, H.S.; Bartelink, I.H.; Vissink, A.; Weisman, G.A.; et al. Intraoperative Visualization and Treatment of Salivary Gland Dysfunction in Sjögren’s Syndrome Patients Using Contrast-Enhanced Ultrasound Sialendoscopy (CEUSS). J. Clin. Med. 2023, 12, 4152. https://doi.org/10.3390/jcm12124152
Karagozoglu KH, Mahraoui A, Bot JCJ, Cha S, Ho J-PTF, Helder MN, Brand HS, Bartelink IH, Vissink A, Weisman GA, et al. Intraoperative Visualization and Treatment of Salivary Gland Dysfunction in Sjögren’s Syndrome Patients Using Contrast-Enhanced Ultrasound Sialendoscopy (CEUSS). Journal of Clinical Medicine. 2023; 12(12):4152. https://doi.org/10.3390/jcm12124152
Chicago/Turabian StyleKaragozoglu, K. Hakki, Anissa Mahraoui, Joseph C. J. Bot, Seunghee Cha, Jean-Pierre T. F. Ho, Marco N. Helder, Henk S. Brand, Imke H. Bartelink, Arjan Vissink, Gary A. Weisman, and et al. 2023. "Intraoperative Visualization and Treatment of Salivary Gland Dysfunction in Sjögren’s Syndrome Patients Using Contrast-Enhanced Ultrasound Sialendoscopy (CEUSS)" Journal of Clinical Medicine 12, no. 12: 4152. https://doi.org/10.3390/jcm12124152