A 75-year-old male patient with asthma presented with left eye vision loss for 1 year and progressive nasal obstruction, anosmia, and purulent eyelid and nasal discharge for several months. There was no light perception vision with an afferent pupillary defect in the left eye. Examination demonstrated nasal dorsum widening, sino-cutaneous fistulae, bilateral proptosis, and left eye chemosis with corneal keratinization in the setting of prior perforation (Fig. 1A). Nasal endoscopy showed severe, bilateral polyposis (Fig. 1B). MRI revealed an extensive sinonasal process with left globe disfigurement and severe, postobstructive, sinus mucocele formation (Fig. 2, top). The patient underwent bilateral functional endoscopic sinus surgery. Intraoperatively extensive sinonasal polyposis and inspissated mucinous debris were encountered. Histopathology demonstrated nasal polyps with dense eosinophilic infiltration and cultures grew polymicrobial bacteria. He underwent left eye evisceration, left lower eyelid ectropion repair, and second-stage sinus surgery. Postoperatively, there were improvements in pain, breathing, and smell. Nasal endoscopy displayed improved sinus patency, although with rapid polyp recurrence. Given concern for mucocele formation and potential impact on the right eye, bimonthly subcutaneous dupilumab was initiated. Following 5 doses, there was dramatic improvement, with symptom resolution, nasal bridge narrowing, sinonasal polyposis reduction, and fistulae closure (Fig. 1C). Subsequent CT showed dramatic polyposis involution and sinus clearing (Fig. 2, bottom). Severe chronic sinusitis with polyposis may exert mass effect on local structures including the brain, optic nerves, and globes. Among patients with severe, refractory, or recurrent polyposis, subcutaneous dupilumab may provide a dramatic clinical response.FIG. 1FIG. 2