Reversible Charles Bonnet Syndrome After Oculoplastic Procedures

Individuals with Charles Bonnet syndrome (CBS) typically have severe visual loss and experience visual hallucinations yet have no psychiatric disease. Visual impairment often is due to end-stage glaucoma or macular degeneration. We report 3 cases of CBS in patients who underwent an oculoplastic surgical procedure. One patient experienced binocular visual distortion due to excessive topical ophthalmic ointment, and 2 patients experienced monocular visual impairment from patching. Visual hallucinations resolved once vision returned to baseline. We highlight the possibility of transient CBS in postoperative patients who have temporary iatrogenic vision impairment in one or both eyes.

Clinical Correspondence Reversible Charles Bonnet Syndrome After Oculoplastic Procedures Robert A. Beaulieu, MD, Diana A. Tamboli, MD, Blair K. Armstrong, MD, R. Nick Hogan, MD, PhD, Ronald Mancini, MD, FACS Abstract: Individuals with Charles Bonnet syndrome (CBS) typically have severe visual loss and experience visual hallucinations yet have no psychiatric disease. Visual impairment often is due to end-stage glaucoma or macular degeneration. We report 3 cases of CBS in patients who underwent an oculoplastic surgical procedure. One patient experienced binocular visual distortion due to excessive topical ophthalmic ointment, and 2 patients experienced monocular visual impairment from patching. Visual hallucinations resolved once vision returned to baseline. We highlight the possibility of transient CBS in postoperative patients who have temporary iatrogenic vision impairment in one or both eyes. Journal of Neuro-Ophthalmology 2018;38:334-336 doi: 10.1097/WNO.0000000000000477 © 2016 by North American Neuro-Ophthalmology Society P atients with deteriorating vision can experience complex visual hallucinations despite normal cognitive function and retain insight that the hallucinations are not real. This condition is known as the Charles Bonnet syndrome (CBS). Classically, individuals who experience these hallucinations have advanced age, decreased visual sensory input, no demonstrable mental illnesses, and symptoms during times of low arousal, fatigue, or stress (1,2). Visual impairment most commonly is associated with degenerative eye diseases, but has been described with a variety of lesions along the visual pathway (1-3). Once thought to be a rare occurrence, larger reviews suggest that CBS affects 11%-27% of visually handicapped people (1,4-6). Although most cases of CBS occur in patients with bilateral and permanent visual impairment, there are reports of reversible CBS in patients with temporary vision loss in either one or both eyes. Department of Ophthalmology, University of Texas Southwestern Medical Center, Dallas, Texas. The authors report no conflicts of interest. Address correspondence to Ronald Mancini, MD, FACS, Department of Ophthalmology, 5323 Harry Hines Boulevard, Dallas, TX 75390; E-mail: [e-mail] 334 We describe 3 patients seen by the oculoplastic surgeons at a large tertiary referral center. These patients experienced visual hallucinations due to both monocular and binocular visual distortion and were diagnosed with CBS, all with resolution of symptoms once vision returned to baseline. CASE REPORTS Patient 1 A 73-year-old man underwent bilateral upper eyelid blepharoplasty, ptosis repair, and lower lid ectropion repair. His preoperative visual acuity was 20/25 in each eye. The procedures were performed without complication, and the patient was instructed to place erythromycin ointment in both eyes and on his upper eyelid incisions postoperatively. Later on the day of surgery, he presented to the emergency department complaining of seeing "interlocking transparent links of rope rotating around the door handle in my bedroom." He also reported seeing a cartoon of Bugs Bunny and Donald Duck on his cell phone screen when the screen was turned off, and recounted an "old time black and white movie" playing on the entryway overhang of a restaurant. All his hallucinations were vivid and clear with sharp borders. Most were in color with the exception of the movie. Each hallucination would last for 1-3 minutes. On examination, he was noted to have excessive ointment in his eyes with visual acuity of 20/100, right eye, and 20/200, left eye. Ophthalmoscopy was normal bilaterally. Carotid artery ultrasonography and cardiac echography were within normal limits. When examined on postoperative day 3, the patient reported continuation of the visual disturbances. His vision was again noted to be 20/100 in the right eye and 20/200 in the left eye with considerable ointment in his eyes. He reported applying ointment in his eyes 4 times per day. Each hallucination would last for 1-3 minutes and would occur a dozen times each day. Beaulieu et al: J Neuro-Ophthalmol 2018; 38: 334-336 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Clinical Correspondence The images were present from the evening of his surgery to the morning of postoperative day 4. On discontinuing ophthalmic ointment, the patient had clear vision with cessation of hallucinations. When examined on postoperative day 6, visual acuity was 20/20, right eye, and 20/40, left eye, and he has remained stable without recurrence of visual hallucinations. Patient 2 A 78-year-old man with a history of 3 previous lower eyelid operations for laxity and ectropion complained of left eye epiphora. He underwent a left lower eyelid retraction repair, ectropion repair, full-thickness skin graft, and temporary reverse Frost suture tarsorrhaphy. His medical history was significant for a cerebrovascular accident with residual leftsided facial weakness, but no cognitive deficits. Preoperative visual acuity was 20/20, right eye, and 20/40, left eye. The surgical procedures were completed without complications, and a pressure patch was placed over the left eye and was not removed until postoperative day 6. The patient reported visual hallucinations from the evening of his surgery until 2 days after removal of the eye patch and Frost suture. They did not recur thereafter. He described one hallucination as seeing a miniature deck of cards scattered on a table with the cards "turned up," and recalled lying in bed and when looking up it appeared as if he was looking through the branches and leaves of a tree. He also reported seeing broken glass with sharp edges scattered on the floor (Fig. 1). The objects were stationary and in color. The hallucinations would last for approximately 5 minutes and would disappear when the patient would look away or think about something else. They occurred approximately 5-8 times per day. Patient 3 An 82-year-old man with right lower eyelid cicatricial ectropion and exposure keratopathy underwent repair with lateral tarsal strip and full-thickness skin graft with a reverse Frost suture tarsorrhaphy. His medical history included mitral valve repair, defibrillator placement, kidney disease, but no history of mental illness. His preoperative visual acuity was 20/40, right eye and 20/30, left eye. After eyelid surgery, a pressure patch was placed over the right eye and was not removed until 1 week later. Vision in the unoperated eye remained stable. Visual hallucinations started on postoperative day 2 and were comprised of 2-dimensional purple patterns. These patterns were stationary, and he would see them when he was in his bathroom and in church. He recalled one episode in which the carpet turned into a floral pattern and extending in front of him as he walked. He also reported seeing a large 3-dimensional flowering bush which was green and purple and extended from the floor to the ceiling. The hallucinations would occur 5-6 times per day lasting 2-5 minutes, and occurred Beaulieu et al: J Neuro-Ophthalmol 2018; 38: 334-336 FIG. 1. Patient 2. Hallucinations this patient experienced include a scattered deck of cards (A), appearance of leaves on branches of a tree (B), and broken glass with jagged edges (C). with his eyes open and closed, and resolved when he shifted gaze. The hallucinations stopped immediately after removal of the left eye patch and the Frost suture. 335 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Clinical Correspondence DISCUSSION We describe 3 patients who experienced CBS after an oculoplastic procedure with transient monocular occlusion or transient binocular blurring secondary to ophthalmic ointment. Our patients experienced vividly colored hallucinations, some of which were animated, mostly during times of low stimulation, consistent with typical features of episodes in CBS (7). The exact mechanism of CBS hallucinations is not fully understood. The leading theory is that sensory deafferentation of the vision-associated areas of the cerebral cortex leads to the release of hallucinatory activity (2,4,6,8,9). There are no established diagnostic criteria for CBS, but generally accepted guidelines proposed by Gold and Rabins (10) include the presence of complex visual hallucinations without other sensory hallucinations or delirium as well as insight into the unreal nature of the hallucinations. The hallucinations may be episodic, periodic, or chronic and can range from mundane, inanimate objects or people to complex scenes (1). In general, low levels of sensory deprivation and low levels of arousal appear to contribute to hallucinations (1,2). There is no universally effective treatment and medication is rarely useful (2). Reassurance and attempted at maximizing visual function are the mainstays of therapy (2). There are few reports of CBS occurring after surgery. Khadavi et al (3) described a patient with acute, reversible CBS after unilateral eye patching following right lower eyelid cicatricial ectropion repair. Hallucinations started 2 days postoperatively and stopped once the patch was removed and vision returned to baseline. Jackson and Madge (11) documented CBS in a patient after unilateral eye patching and tarsoconjunctival flap and full-thickness skin graft. Hallucinations started immediately postoperatively in the surgical recovery area and lasted 2 days after patch removal. Gander et al (12) also described a case of CBS in the setting of unilateral vision loss after Frost suture placement during orbital floor repair, CBS resolved once the Frost suture was removed. Temporary visual impairment can lead to complex visual hallucination, and because many patients do not report these findings to their physician, it could be argued that CBS may affect a much larger patient population. Discussions about the possibility of developing visual hallucinations with patients undergoing ocular surgery, especially those that will be patched or with significant impairment in 336 one or both eyes after the procedure, could lead to less anxiety and concerns postoperatively in patients with otherwise healthy eyes. STATEMENT OF AUTHORSHIP Category 1: a. Conception and design: R. Mancini, D. Tamboli, and R. Beaulieu; b. Acquisition of data: R. Mancini, D. Tamboli, B. Armstrong, and R. Beaulieu; c. Analysis and interpretation of data: R. Mancini, R. N. Hogan, D. Tamboli, B. Armstrong, and R. Beaulieu. Category 2: a. Drafting the manuscript: D. Tamboli, B. Armstrong, and R. Beaulieu; b. Revising it for intellectual content: R. Mancini, R. N. Hogan, D. Tamboli, B. Armstrong, and R. Beaulieu. Category 3: a. Final approval of the completed manuscript: R. Mancini, R. N. Hogan, D. Tamboli, B. Armstrong, and R. Beaulieu. REFERENCES 1. 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Beaulieu et al: J Neuro-Ophthalmol 2018; 38: 334-336 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.