Marginal corneal infiltrates as an ocular manifestation of acute generalized exanthematous pustulosis

PURPOSE: To report a case of keratoconjunctivitis with marginal corneal infiltrates in a patient with acute generalized exanthematous pustulosis (AGEP) secondary to trimethoprim-sulfamethoxazole. OBSERVATIONS: A 63-year-old female presented with a diffuse pustular skin rash and bilateral keratoconjunctivitis with marginal corneal infiltrates. Skin biopsy led to the diagnosis of AGEP secondary to trimethoprim-sulfamethoxazole use. Treatment of the ocular findings with topical corticosteroids and lubrication led to near-full resolution after two weeks. CONCLUSIONS AND IMPORTANCE: To the best of our knowledge, this is the first reported association between AGEP and keratoconjunctivitis with marginal corneal infiltrates. A hypersensitivity reaction to a foreign antigen is implicated in the pathogenesis of both AGEP and sterile marginal infiltrates, and we suggest that the patient's underlying hypersensitivity process associated with AGEP accounted for the ocular findings.