|Year : 2023 | Volume
| Issue : 2 | Page : 547-548
Unusual bilateral corneal edema seen in a case of Behcet's like syndrome
Sikha Misra1, Chinthala Navyasree2, Somasheila Murthy1
1 Cornea Department, L. V. Prasad Eye Institute, Bhubaneswar, Odisha, India
2 Department of Ophthalmology, IMS and SUM Hospital, Bhubaneswar, Odisha, India
|Date of Submission||16-Nov-2022|
|Date of Acceptance||02-Jan-2023|
|Date of Web Publication||28-Apr-2023|
Cornea Department, L. V. Prasad Eye Institute, Bhubaneswar, Odisha
Source of Support: None, Conflict of Interest: None
Keywords: Behcets, bilateral, cornea edema
|How to cite this article:|
Misra S, Navyasree C, Murthy S. Unusual bilateral corneal edema seen in a case of Behcet's like syndrome. Indian J Ophthalmol Case Rep 2023;3:547-8
|How to cite this URL:|
Misra S, Navyasree C, Murthy S. Unusual bilateral corneal edema seen in a case of Behcet's like syndrome. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Jun 5];3:547-8. Available from: https://www.ijoreports.in/text.asp?2023/3/2/547/375014
A 32-year-old male was referred from the immunology department with a 3-day history of both eyes (BES) ocular pain with extreme photophobia, oral, genital ulcers, and eruptive skin lesions. He had similar episodes 2 years ago, which improved with topical therapy. Visual acuities were 6/24 (pinhole 6/9) and 6/60 (pinhole 6/12) in the right and left eyes, respectively. Slit-lamp examination showed diffuse corneal edema with ruptured bullae in the left eye [Figure 1]a and [Figure 1]b and no other findings. Fundus was normal with no evidence of vasculitis. The ocular condition rapidly improved with hypertonic saline, lubricants, and intravenous dexamethasone. One day later, his vision was 6/9 BES [Figure 1]c, [Figure 1]d, [Figure 1]e, [Figure 1]f. Investigations revealed human leukocyte antigen (HLA)-B52 positivity. Oral steroids were continued in tapering doses. One month later, his edema had completely resolved [Figure 2]a and [Figure 2]b, intra-ocular pressures were normal and specular microscopy showed normal counts [Figure 2]c and [Figure 2]d.
|Figure 1: (a) Slit-lamp photograph of the right eye showing corneal edema (black arrow) at the first visit. (b) The left eye showing diffuse corneal edema (black arrow) with ruptured bullae (white arrow). (c) Fundus photograph of the right eye showing normal fundus at presentation. (d) Slit-lamp photograph of the right eye showing decreased edema on day 2. (e) The left eye similarly showing decreased edema and healing epithelial bullae. (f) Fundus photograph of the left eye showing normal fundus on day 2|
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|Figure 2: (a) Slit-lamp photograph of the right eye showing resolved edema at 1-month follow-up. (b) Similar findings observed in the left eye with a clear cornea. (c) Secular microscopy of the right eye showing normal cell count and morphology. (d) Similar findings noted in the specular image of the left eye|
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| Discussion|| |
Behcet's disease (BD) is a multisystemic autoimmune disorder of unknown etiology, characterized by oral and genital ulcers and skin, vascular, and ocular inflammatory manifestations, requiring aggressive immunomodulator therapy., The most important predisposing genetic factor is HLA-B51, but HLA B52 has also been reported. What was remarkable in our case was the dramatic improvement of corneal edema. The cause of such edema can be acute presentation of immunologic phase of BD or a coincidental finding.
Certain medications such as chlorpromazine, tamoxifen, and amantadine can cause sudden corneal edema. Trauma and toxins, such as plant sap injury or other chemicals, also affect the cornea, but there was no history of drug intake, trauma, or exposure to toxins in our patient. Normal specular counts ruled out endothelial dystrophy. We therefore hypothesize that this presentation could have been due to sudden immunological storm, with ulcerative lesions of oral, genital, and corneal epithelium; and rapidly improved due to systemic steroids but we have no evidence, and this unusual finding could have been coincidental.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Hyderabad Eye Research Foundation provided the financial support.
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2]