Anifrolumab in Refractory Oral Manifestations in Systemic Lupus Erythematosus: A Case Report and Literature Review.

Abstract

Oral manifestations are a frequent yet underrecognized feature of systemic lupus erythematosus (SLE), contributing substantially to patient morbidity and reduced quality of life. They are insufficiently represented in clinical outcome measures and therapeutic guidelines, despite their clinical relevance. We report the case of a 39-year-old woman with SLE who developed severe, refractory tongue ulcers and glossodynia persisting for three years, unresponsive to hydroxychloroquine (HCQ), azathioprine (AZA), colchicine, methotrexate (MTX), and glucocorticoids (GC). Belimumab was discontinued due to gastrointestinal intolerance and lack of efficacy on oral lesions. The patient was subsequently treated with anifrolumab 300 mg intravenously every four weeks. After the first infusion, oral ulcers resolved, arthritis improved, and corticosteroids were discontinued. Within three months, the tongue lesions had completely healed, enabling unrestricted oral intake and full resumption of professional activities. Clinical remission was maintained on anifrolumab and hydroxychloroquine alone at four months of follow-up. This case highlights the therapeutic challenge of refractory oral lesions in SLE, a manifestation not adequately addressed in current EULAR (2023 update) or ACR (2025) guidelines. While conventional immunosuppressants such as azathioprine, methotrexate, and mycophenolate mofetil remain options, their efficacy for isolated oral lesions is limited. Emerging evidence, including case reports and small series, suggests that anifrolumab may provide significant benefit for mucosal involvement. The key learning point is that refractory tongue involvement may represent a dominant and treatment-resistant manifestation of SLE, yet it can respond to targeted biologic therapy such as anifrolumab and our report adds to this growing body of evidence. Prospective comparative studies and registry analyses are required to define anifrolumab's effectiveness specifically for refractory oral manifestations and to compare outcomes with other biologics.