Ocular Manifestations in Adult-Onset Still’s Disease: A Rare Case of Bilateral Uveitis and Updated Literature Review

Background Adult-onset Still’s disease (AOSD) is a systemic autoinflammatory disorder with a broad clinical spectrum. Ocular involvement is extremely rare and typically associated with chronic disease or posterior segment inflammation. To date, acute bilateral anterior uveitis has not been reported as an initial manifestation of AOSD. Case presentation We report the case of a 30-year-old patient who presented with fever, urticarial rash, arthralgia, and acute bilateral ocular pain and photophobia. Comprehensive infectious and autoimmune workups were negative. Dermatologic and histopathologic findings, elevated inflammatory markers, and markedly increased IL-6 and sCD25 levels supported the diagnosis of AOSD. The patient responded rapidly to systemic corticosteroids and IL-6 blockade with tocilizumab, achieving full clinical and ophthalmologic remission. Discussion This case represents the first report of acute bilateral anterior uveitis as the initial clinical manifestation of AOSD. The ocular inflammation was likely driven by cytokine dysregulation, particularly IL-6, which may compromise the blood-ocular barrier. The favorable response to tocilizumab further supports IL-6 as a key pathogenic factor. Considering this, we conducted a focused literature review, identifying four additional cases of ocular involvement in AOSD. These cases underline the importance of prompt diagnosis and immunomodulatory treatment to preserve vision and control systemic disease. Conclusion This case expands the known clinical spectrum of AOSD and underscores the need to consider it in patients presenting with bilateral anterior uveitis and systemic inflammatory signs. IL-6 inhibition may be an effective therapeutic approach in such atypical presentations.