Resolution of a neurotrophic keratopathy associated hypopyon with cenegermin

PURPOSE: We present a novel case of a neurotrophic keratopathy associated inflammatory hypopyon that resolved after initiation of therapy with cenegermin (Oxervate; Dompe, Milan, Italy), a recombinant human nerve growth factor (rhNGF). This finding illustrates the potential of cenegermin in advanced inflammatory neurotrophic disease. OBSERVATIONS: A 60-year-old female with a history of herpes zoster keratitis was evaluated in our clinic for stage 2 neurotrophic keratopathy. One month later, she presented emergently with a large epithelial defect, infiltrate, and hypopyon. Three separate sets of corneal cultures returned negative. She was treated with oral antivirals and aggressive topical antibiotics with no clinical improvement. Given the presumed diagnosis of stage 3 neurotrophic keratopathy with a sterile hypopyon, she was started on cenegermin 6 times daily for 8 weeks in the absence of a corticosteroid. By 2 weeks after starting cenegermin, the epithelial defect, infiltrate, and hypopyon sizes had improved. Within 4 weeks of starting cenegermin, the hypopyon had clinically resolved. The patient was subsequently started on topical corticosteroid drops for the last 4 weeks of cenegermin therapy. Examination at the conclusion of 8 weeks of cenegermin treatment revealed a closed epithelium and minimal scar. Best-corrected visual acuity with contact lens overrefraction was 20/70. Over the course of 7 months of continued follow-up, the cornea remained epithelialized without recurrent corneal infiltration or hypopyon. CONCLUSIONS AND IMPORTANCE: While cenegermin has been previously shown to be an effective treatment for neurotrophic keratopathy associated epithelial defects, resolution of a neurotrophic keratopathy associated inflammatory hypopyon with cenegermin is novel.