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  1. Ellinor Sydow,
  2. Sabien Severi,
  3. Kristien Van der Elst,
  4. Patrick Verschueren,
  5. Jan Lenaerts,
  6. Rene Westhovens,
  7. Ellen De Langhe
  1. University Hospitals Leuven, Rheumatology, Leuven, Belgium


Background Systemic sclerosis (SSc) is a severe autoimmune disease and fibrotic cutaneous involvement of hands and face is a typical characteristic. Oral involvement with reduced oral aperture is frequent and associated with impaired food intake, oral hygiene and secondary dental problems. Several studies have shown that stretching (placing the thumbs in opposite corners of the mouth hand, pulling outward) and oral augmentation (tongue depressors between the back molars) exercises can increase oral aperture but is often hampered by low adherence rates.

Objectives The aim of this descriptive explorative mixed method study was to explore feasibility, patient satisfaction and effectiveness of two exercise programs, Therabite and orofacial exercises, in systemic sclerosis associated microstomia.

Methods We included adult patients suffering from systemic sclerosis (fulfilling the ACR/EULAR 2013 criteria) and microstomia (maximal oral aperture <40mm). We discerned two groups: Group A exercised with a passive jaw motion device (Therabite®), and Group B performed mouth-stretching exercises. Patients were expected to exercise for 10 minutes, 3 times/day for 3 months. Patients were contacted 4 times by telephone to address encountered problems and completed an exercise diary to document the adherence rate. Patients were evaluated at baseline, 3 months (period without intervention), 6 months (after 3 months of intervention) and at 9 months (post-intervention visit). At time point 6 months, semi-structured one-to-one interviews were conducted. Interviews were recorded, transcribed verbatim and systematically analyzed using Qualitative Analysis Guide of Leuven.

Results We included 6 women and 3 men, with a median age of 60 years (range 40-75) and a median disease duration of 8 years (range 3-22). At time point 6 months, all patients in group A (n=4) and 4 patients in group B (n=5) improved with a median of 9mm (range 2-10) and 7mm (range 4-11), respectively. One patient had a decrease of 2mm. The compliance, measured as the ratio of executed exercises relative to the planned number of exercises ranged between 63.7% and 98,9% in group A and between 48.5% and 97,4% in group B. Details are shown in Table 1. In the follow-up period, we documented maintenance of the observed increase in oral aperture in those patients that continued exercising daily. In all others, maximal oral aperture declined again. All 9 patients attended the interview. Three main themes emerged from the data: drivers, challenges and perceived improvement. Patients highlighted several drivers to perform the exercises at home, such as the motivation to improve current disability cause by microstomia. Furthermore, they equally highlighted several challenges regarding feasibility, such as the struggle to exercise multiple times a day. Most of the patients were hoping that they could keep their improvement. They were willing to continue practicing if necessary, but with a lower frequency.

Conclusion This study suggests that both types of intervention can improve maximal oral aperture. The adherence to therapy was higher than expected but none of the patients considered it feasible to continue practicing 3 times/day in the long-term resulting in a decline of improvement post intervention. This is the first study to report the feasibility of the exercises for the patients and can be very useful for health professionals giving guidance. Future studies are needed in order to define exercise programs that are feasible and can be sustained in the long term.

Disclosure of Interests Ellinor Sydow: None declared, Sabien Severi: None declared, Kristien Van der Elst: None declared, Patrick Verschueren Grant/research support from: Unrestricted Pfizer Grant for Early RA research, Jan Lenaerts: None declared, Rene Westhovens Grant/research support from: Bristol-Myers Squibb, Consultant for: Celltrion, Galapagos-Gilead, Ellen De Langhe: None declared

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