Systemic sclerosis (SSc) is a severe autoimmune disease often leading to fibrotic cutaneous involvement of the face. Reduced oral aperture is associated with impaired food intake, oral hygiene and secondary dental problems. Stretching and oral augmentation exercises can increase oral aperture but are often hampered by low adherence rates. The aim of this mixed method study was to explore feasibility, patient satisfaction and effectiveness of two exercise programs in SSc-associated microstomia.
Adult patients (<18 years) suffering from systemic sclerosis (fulfilling the ACR/EULAR 2013 criteria) and microstomia (maximal oral aperture <40mm) were randomized to two groups. Group A exercised with a passive jaw motion device (Therabiteâ), and Group B performed mouth-stretching exercises. Patients were expected to exercise for 10 minutes, 3 times/day for 3 months, completed an exercise diary and were contacted 4 times by telephone. 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 month 6 semi-structured one‐to‐one interviews were conducted, recorded, transcribed verbatim and analyzed using Qualitative Analysis Guide of Leuven (QUAGOL).
We included 6 women and 3 men, median age 60 years (range 40-75) and median disease duration 8 years (range 3-22). At 6 months, all patients in group A (n=4) and 4 in group B (n=5) improved with a median of 9mm (range 2-10) and 7mm (range 4-11), respectively. The proportion of executed 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. Maintenance of the increase in oral aperture was noted in patients that continued to exercise daily. All 9 patients attended the interview that revealed three themes: drivers, challenges and perceived improvement.
Both interventions improve maximal oral aperture. The adherence to therapy was high but none of the patients considered it feasible to continue practicing 3 times/day. Future studies are needed in order to define feasible long-term exercise programs.