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Year : 2023  |  Volume : 3  |  Issue : 2  |  Page : 547-548

Unusual bilateral corneal edema seen in a case of Behcet's like syndrome

1 Cornea Department, L. V. Prasad Eye Institute, Bhubaneswar, Odisha, India
2 Department of Ophthalmology, IMS and SUM Hospital, Bhubaneswar, Odisha, India

Date of Submission16-Nov-2022
Date of Acceptance02-Jan-2023
Date of Web Publication28-Apr-2023

Correspondence Address:
Sikha Misra
Cornea Department, L. V. Prasad Eye Institute, Bhubaneswar, Odisha
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJO.IJO_3021_22

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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 10];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 Top

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.[1],[2] The most important predisposing genetic factor is HLA-B51,[3] but HLA B52 has also been reported.[4] 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.[5] 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.

  References Top

Davatchi F. Diagnosis/Classification criteria for Behcet's disease. Patholog Res Int 2012;2012:607921. doi: 10.1155/2012/607921.  Back to cited text no. 1
Kaçmaz RO, Kempen JH, Newcomb C, Gangaputra S, Daniel E, Levy-Clarke GA, et al. Ocular inflammation in Behçet disease: Incidence of ocular complications and of loss of visual acuity. Am J Ophthalmol 2008;146:828-36.  Back to cited text no. 2
De Menthon M, Lavalley MP, Maldini C, Guillevin L, Mahr A. HLA-B51/B5 and the risk of Behçet's disease: A systematic review and meta-analysis of case-control genetic association studies. Arthritis Rheum 2009;61:1287-96.  Back to cited text no. 3
Arber N, Klein T, Meiner Z, Pras E, Weinberger A. Close association of HLA-B51 and B52 in Israeli patients with Behçet's syndrome. Ann Rheum Dis 1999;50:351-3.  Back to cited text no. 4
Sahyoun J, Sabeti S, Robert M drug-induced corneal deposits: An up-to-date review BMJ Open Ophthalmol 2022;7:e000943. doi: 10.1136/bmjophth-2021-000943.  Back to cited text no. 5


  [Figure 1], [Figure 2]


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