Orbital myositis in systemic lupus erythematosus (SLE) is a rare entity with risk of serious complications. Timely treatment with effective immunosuppressors is desirable. We report a case of a 32-year-old female patient with SLE who presented with an acute ocular pain and extraorbital muscle thickening, consistent with orbital myositis. Association with SLE was made after exclusion of other aetiologies. Due to refractoriness to steroids, off-label rituximab was initiated with clinical and imaging parameter improvement.

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