Abstract
RATIONALE: Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) are severe, life-threatening hypersensitivity reactions most often induced by systemic medications. Reports of SJS/TEN caused by topical ophthalmic sulfonamide-derived carbonic anhydrase inhibitors, such as brinzolamide, are extremely rare. PATIENT CONCERNS: A 31-year-old man underwent pars plana vitrectomy for traumatic retinal detachment in the right eye and subsequently developed secondary glaucoma. He was treated with topical brinzolamide 1% twice daily. On day 6 of treatment, he developed high-grade fever and erythema on the upper limbs, which rapidly progressed to widespread edematous erythema, bullae, epidermal detachment, and erosions involving 99% of the total body surface area. Ocular involvement was asymmetric: the right eye showed eyelid ulceration and conjunctival hyperemia, while the left eye had more severe manifestations, including marked conjunctival chemosis and extensive corneal epithelial sloughing. DIAGNOSIS: Laboratory tests revealed leukopenia, elevated C-reactive protein, abnormal liver enzymes, and hypoalbuminemia. Blood cultures grew Escherichia coli and Streptococcus species. Causality assessment demonstrated a Naranjo score of 7 and an ALDEN score of 5, indicating a probable relationship between brinzolamide and SJS/TEN overlap syndrome. INTERVENTIONS: The patient was treated with intravenous immunoglobulin, systemic corticosteroids, broad-spectrum antibiotics, antifungal therapy, supportive care, and ocular surface protection. OUTCOMES: Over 4 weeks, cutaneous lesions resolved with residual hyperpigmentation. The left eye developed symblepharon and dense corneal scarring, requiring penetrating keratoplasty, resulting in postoperative visual acuity improvement to 0.05. The right eye remained without light perception due to preexisting retinal detachment. LESSONS: Topical ocular administration of sulfonamide-based drugs may trigger life-threatening systemic hypersensitivity reactions. Thorough allergy history and human leukocyte antigen genotyping should be considered before prescribing. Early recognition of prodromal symptoms, particularly ocular signs, and prompt multidisciplinary intervention are critical to preventing severe complications and irreversible vision loss.