To describe a case of severe dupilumab-associated blepharoconjunctivitis with giant papillae treated with high potency corticosteroid eyedrops, without discontinuing or reducing dupilumab therapy.A 22-year-old Latin American female with a long history of severe atopic dermatitis (AD) with no ocular involvement presented 20 weeks after starting treatment with dupilumab injections with blurry vision, multiple chalazia, eyelid swelling and severe conjunctival injection in both eyes. She also reports having a hordeolum 2 months prior and severely dry eyes starting 2 weeks prior. Slit-lamp examination revealed severe conjunctivitis with macroscopically visible giant papillae in the right lower tarsal conjunctiva. The diagnosis of severe dupilumab-associated blepharoconjunctivitis was made and difluprednate 0.05% eyedrops two times a day for 7 days was initiated. Given the severity of her AD and her marked skin improvement with dupilumab, it was decided to continue dupilumab without reducing the dose. At 2-day follow-up, conjunctival injection had markedly improved, and at 2-month follow-up, her examination was unremarkable. Currently, our patient only uses dexamethasone 0.1% drops few times a week as per needed for occasional eye irritation.As dupilumab injections begin to claim a rightful place in medicine, the ophthalmic community may start encountering dupilumab-associated ocular surface disease all more often and potentially play an important role in identifying, characterizing and treating the adverse ocular effects from this novel medication.