Purpose: We report a case of refractory atopic keratoconjunctivitis (AKC) which was successfully treated with subcutaneous immunotherapy (SCIT). Case Report: A 22-year-old woman presented with severe allergic conjunctivitis for one and a half year. She failed to respond to conventional topical anti-allergic medications, topical corticosteroid, as well as topical cyclosporine A. Therefore, oral corticosteroids had to be prescribed to control the exacerbation for 1 year. Due to refractory AKC and to avoid long-term corticosteroid use, we referred her to an allergy clinic for considering the role of SCIT. Allergology investigations showed positive skin prick test and strongly elevated serum-specific IgE to Dermatophagoides farinae (Der f) and Dermatophagoides pteronyssinus (Der p). She received a conventional protocol of SCIT using Der f and Der p allergen extracts. Results: The patient’s ocular signs and symptoms were dramatically improved 2 months after the initiation of SCIT, and oral corticosteroids could be discontinued within 3 months of the treatment. She was maintained with mast cell stabilizers and preservative-free tears without any episodes of exacerbation. Conclusions: SCIT may contribute to successful outcomes in controlling symptoms and preventing exacerbation in AKC patient. It should be considered as an alternative or even a primary treatment for patients with refractory AKC. However, the optimal SCIT protocol must be discussed with an allergist on an individual basis for the best outcome.