Purpose: To document a case involving a patient previously diagnosed with Behçet's disease which proved unresponsive to multiple immunomodulatory therapies, and was subsequently diagnosed with secondary cytomegalovirus retinitis and appropriately treated., Methods: This is a case report focused on the images., Results: A 39-year-old female, previously diagnosed with Behçet's disease unresponsive to multiple immunomodulatory therapies, sought a second opinion at our clinic due to more floaters and a scotoma in her left eye for a few months. Her right eye had become blind as a result of multiple glaucoma and vitreoretinal surgeries. Her best corrected visual acuity was 20/60 in the left eye. A Slit lam examination of the left eye showed 1+ cells and flare in the anterior chamber along with 1+ cells in the anterior vitreous with no vitreous haze. Dilated fundoscopy of the left eye reveled an atrophic lesion in the inferior macula. Fluorescein angiography demonstrated a mixed hypo- and hyperfluorescent lesion in the left eye. Optical coherence tomography macula demonstrated an atrophic lesion in the inferior macula of the left eye. All laboratory findings were predominantly negative or within the normal range, except for the presence of antibodies to VZV and CMV in the blood. Polymerase chain reaction analysis of the vitreous sample uncovered the presence of CMV, leading to appropriate curative and prophylactic treatment for the patient., Conclusions and Importance: In patients with resistant noninfectious uveitis, particularly those experiencing underlying disease reactivation, the possibility of infections, especially opportunistic ones, should be taken into consideration., Competing Interests: Declaration of conflicting interestsThe authors declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: C. Stephen Foster declares the following: Consultancies with Aldeyra Therapeutics (Lexington, MA), Allakos (Redwood City, CA), Bausch & Lomb Surgical, Inc. (Rancho Cucamonga, CA), Eyegate Pharma (Waltham, MA), Genentech (South San Francisco, CA), Novartis (Cambridge, MA), and pSivida (Watertown, MA). Grants or grants pending with Aciont (Salt Lake City, UT), Alcon (Aliso Viejo, CA), Aldeyra Therapeutics (Lexington, MA), Bausch & Lomb (Rochester, NY), Clearside Biomedical (Alpharetta, GA), Dompé pharmaceutical (Milan, Italy), Eyegate Pharma (Waltham, MA), Mallinckrodt pharmaceuticals (Staines-upon-Thames, UK), Novartis Pharmaceuticals (Cambridge, MA), pSivida (Watertown, MA), and Santen (Osaka, Japan). Payment for lectures including service on speaking bureaus: Alcon (Aliso Viejo, CA), Allergan (Dublin, Ireland), Mallinckrodt pharmaceuticals (Staines-upon-Thames, UK). Stock or Stock Options: Eyegate Pharma (Waltham, MA). Arash Maleki has nothing to declare.