https://doi.org/10.1177/1120672118803534 European Journal of Ophthalmology 1–4 © The Author(s) 2018 Article reuse guidelines: sagepub.com/journals-permissions DOI: 10.1177/1120672118803534 journals.sagepub.com/home/ejo EJO European Journal of Ophthalmology Introduction Behçet’s disease (BD) is a chronic systemic inflamma- tory vasculitis of unknown etiology. Non necrotizing inflammatory damage involves many organ systems and patient may present with oral ulcers, genital ulcers, joint disease, uveitis, recurrent venous thrombosis, intracra- nial sinus thrombosis, gastrointestinal complications, and erythema nodosum. 1 Mucocutaneous ulcers have been found to be most common presenting illness followed by genital ulcers and ocular inflammation. 1 Eye involve- ment in BD is common and usually presents with panu- veitis with mainly posterior segment inflammation. 2 Severe kerato-conjunctivitis without other features of eye involvement has been reported infrequently. 3 Our patient presented with severe bilateral keratitis followed by development of seizures, which was subsequently diag- nosed as neuro-BD. Case summary A 19-year-old Nepalese male presented to the emergency services of our tertiary-care center with complaints of ocu- lar discomfort, photophobia, watering, and redness in both the eyes (OU) (left > right eye) for the past 1 week. The patient had been diagnosed as viral conjunctivitis else- where and was started on topical moxifloxacin 0.5% and fluorometholone 0.1%. Since there was no improvement in the symptoms, the patient was referred for a second opinion. On examination, his visual acuity was 6/18 and 6/60 in right eye (OD) and left eye (OS), respectively. Slit-lamp examination revealed bilateral diffuse conjuncti- val congestion with palpebral conjunctival follicular reac- tion. There was presence of stromal infiltrates with overlying punctate epitheliopathy and was located densely at the termination of limbal vessels at peripheral cornea OU. The stromal infiltrates were more dense, diffuse and were progressing centripetally OS (Figure 1). Corneal sen- sations were normal OU. These corneal findings were Immune keratitis: An unusual primary presentation of neuro-Behçet’s disease Jitender Jinagal 1 , Aniruddha Agarwal 1 , Avinash Negi 1 , Gaurav Gupta 1 , Vikas Sharma 2 , Parul Chawla Gupta 1 and Jagat Ram 1 Abstract A 19-year-old Nepalese male presented with complaints of bilateral ocular discomfort, photophobia, watering, and redness for 1 week. Visual acuity was 6/12 and 6/60 in the right and left eye, respectively. On biomicroscopic examination, presence of peripheral stromal infiltrates with conjunctival follicles was noted; infiltrates progressed to involve central cornea with further decrease in vision over next few days. After ruling out infectious keratitis, detailed systemic examination and laboratory investigations were diagnostic of neuro-Behçet’s disease. Patient responded to systemic steroidal and immunosuppressive therapy characterized by corneal healing and visual acuity improvement to 6/6 and 6/9. Although rare, but neuro-Behçet’s disease can primarily present as bilateral immune keratitis and every case of bilateral keratitis needs early systemic evaluation after ruling out infective etiologies. Keywords Behçet disease, bilateral keratitis, immune keratitis, peripheral keratitis, neuro-Behçet Date received: 8 July 2018; accepted: 4 September 2018 1 Advanced Eye Centre, Department of Ophthalmology, Postgraduate Institute of Medical Education & Research (PGIMER), Chandigarh, India 2 Department of Rheumatology, Postgraduate Institute of Medical Education & Research (PGIMER), Chandigarh, India Corresponding author: Jagat Ram, Advanced Eye Centre, Department of Ophthalmology, Postgraduate Institute of Medical Education & Research (PGIMER), Sector 12, Chandigarh 160012, India. Email: drjagatram@gmail.com 803534EJO 0 0 10.1177/1120672118803534European Journal of OphthalmologyJinagal et al. case-report 2018 Case Report