Adjunctive Molecular Hydrogen Therapy Modulates T Cell Markers and Reduces Anti-Ro Antibody in Refractory Oral Ulcers of Behçet's Disease and Sjögren's Syndrome: A Case Report.

[BACKGROUND/AIM] Behçet's Disease is a systemic vasculitis affecting both venous and arterial vessels, characterized by painful oral and genital ulcers. Primary Sjögren's Syndrome is an autoimmune disorder mainly targeting exocrine glands, especially the salivary and lacrimal glands, with possible systemic involvement. Molecular hydrogen has emerged as a potential adjuvant therapy due to its antioxidant and immunomodulatory effects. This report evaluates the efficacy of molecular hydrogen as adjunctive therapy for refractory oral ulcers in a patient with Behçet's disease and primary Sjögren's syndrome.

[CASE REPORT] A 67-year-old Taiwanese woman with Behçet's disease and primary Sjögren's syndrome had recurrent painful oral ulcers and mucosal atrophy since 2018, meeting the International Study Group (ISG) criteria. In 2020, she fulfilled the 2016 European League Against Rheumatism (EULAR)/American College of Rheumatology (ACR) criteria for Sjögren's syndrome based on Schirmer's test (0/0 mm), positive anti-Ro antibody (29 U/ml), and labial salivary gland biopsy findings. Despite long-term, hydroxychloroquine, colchicine, and monthly high-dose intravenous vitamin C and glutathione, her oral ulcers recurred with petechiae and bleeding. In January 2024, oral molecular hydrogen capsules were added as adjunctive therapy. Over several months, she showed marked clinical improvement, with less mucosal bleeding and decreased anti-Ro antibody levels. Immunophenotyping revealed increased naïve T helper Fas cell surface death receptor-positive (Fas) and cytotoxic T cell programmed death-1-positive (PD-1) subsets, suggesting immunomodulatory effects. The therapy was well tolerated, with no major adverse events. By August 2025, oral ulcers had markedly improved, and systemic disease remained stable on maintenance with hydroxychloroquine, azathioprine, corticosteroid infusions, and hydrogen therapy.

[CONCLUSION] Molecular hydrogen may aid refractory oral ulcers in Behçet's and primary Sjögren's syndromes, with clinical and immunologic benefits.