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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 379-381

Recurrent herpes simplex virus nummular keratitis in a case of irritable bowel syndrome and seronegative ankylosing spondylitis


1 The Cornea Institute, L V Prasad Eye Institute, Kallam Anji Reddy Campus, Hyderabad, India
2 Jhaveri Microbiology Centre, LV Prasad Eye Institute, Kallam Anji Reddy Campus, Hyderabad, India
3 Wagh Eye Clinic, Pune, Maharashtra and Ruby Hall Clinic, Grant Medical Foundation, Pune, Maharashtra, India

Date of Submission07-Oct-2021
Date of Acceptance15-Dec-2021
Date of Web Publication13-Apr-2022

Correspondence Address:
Somasheila I Murthy
L.V. Prasad Eye Institute, L.V. Prasad Marg, Banjara Hills, Hyderabad - 500 034, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_2575_21

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  Abstract 


A 35-year-old male presented with a 16-month history of left-eye recurrent redness, photophobia, and blurring in his left eye. Systemically, he suffered from irritable bowel syndrome and HLA-B27-negative spondyloarthropathy. The cornea showed multiple nummular epithelial and anterior stromal lesions. The aqueous sample was positive for herpes simplex virus-1 DNA (glycoprotein-D gene) by polymerase chain reaction. The lesions resolved with oral acyclovir and topical dexamethasone 0.01% in tapering doses; however, he continued to have recurrent periodic redness even at 1 year. We hypothesize that coexisting systemic immune-mediated conditions may have a role in altering the disease course leading to its prolongation in our case.

Keywords: Dexamethasone eye drops, HSV nummular keratitis, HSV-1 keratitis treatment, PCR in viral detection, systemic associations in HSV-1 keratitis


How to cite this article:
Bhadra P, Murthy SI, Sharma S, Wagh S, Sheba E. Recurrent herpes simplex virus nummular keratitis in a case of irritable bowel syndrome and seronegative ankylosing spondylitis. Indian J Ophthalmol Case Rep 2022;2:379-81

How to cite this URL:
Bhadra P, Murthy SI, Sharma S, Wagh S, Sheba E. Recurrent herpes simplex virus nummular keratitis in a case of irritable bowel syndrome and seronegative ankylosing spondylitis. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 18];2:379-81. Available from: https://www.ijoreports.in/text.asp?2022/2/2/379/342967



Ocular herpes simplex virus-1 (HSV-1) infection can present as various forms of keratitis, ranging from mild (nummular keratitis, dendritic ulcer) to blinding (necrotizing stromal keratitis, neurotrophic keratitis).[1] Nummular keratitis, a less-common manifestation, is characterized by fine granular or coin-shaped lesions and can cause significant visual discomfort.[2] Predominantly immune-mediated, they persist for years despite antiviral therapy. We report a case of nummular keratitis in a patient with systemic immune-mediated diseases which offered a therapeutic challenge.


  Case Report Top


A 35-year-old pharmacist presented with a 16-month history of recurrent pain, redness, and blurring of vision in the left eye (OS) since September 2019. He had undergone bilateral LASIK 3 years earlier with a good outcome. There was no history of conjunctivitis, rash on the forehead, fever with generalized rash, or contact lens wear at onset. He has a systemic history of irritable bowel disease for many years, which was managed by dietary modification and probiotics. He also had HLA-B27-negative ankylosing spondylitis detected 2 years ago and was on treatment with sulfasalazine therapy and oral non-steroidal anti-inflammatory medication (but not on oral steroids or immunomodulators as he was unable to tolerate these). His rheumatologist had withheld parenteral immunomodulators in view of the ocular infection. He reported that the ocular lesions would rapidly subside, and vision would improve with topical steroids, but the problem would recur almost immediately on steroid taper. He had been treated at various time points with prednisolone acetate 1%, loteprednol etabonate 0.5%, and fluorometholone 0.1% eye drops for recurrences, and although his condition improved, he developed steroid response with intraocular pressure (IOP) increasing to 40 mm Hg, which fortunately responded well to antiglaucoma medications. Topical cyclosporine 0.01% and tacrolimus 0.03% had also been used as steroid-sparing agents but had no effect on the disease. He had also received oral valacyclovir but could tolerate this for a short period of 2 weeks beyond which he discontinued due to gastrointestinal intolerance. Finally, his condition reached an equilibrium with topical dexamethasone 0.01% (formulated) eye drops once daily, which controlled the symptoms without raising the IOP. He stopped dexamethasone 1 week prior to presentation. The uncorrected visual acuity was 20/20, N6 in both eyes. The right eye was essentially normal except for the presence of the scar of the LASIK flap. The left eye showed intact epithelium and multiple flat subtle and indistinct nummular lesions at the level of the superficial anterior stroma temporally and centrally [Figure 1]a and [Figure 1]b. Corneal sensations were normal. A LASIK-flap scar was noted. The rest of the examination was normal, and the optic nerve showed a vertical cup-disc ratio of 0.3:1 The IOP was 14 mm Hg in both eyes. Anterior segment optical coherence tomography (AS-OCT) showed increased reflectivity in the superficial stroma and placoid hyperreflective areas corresponding to the nummular lesions [Figure 1]c. Diagnostic aqueous tap was done and was positive for HSV-1 DNA (glycoprotein-D gene) by conventional PCR [Figure 1]d. Varicella zoster virus (VZV) and cytomegalovirus (CMV) infections were ruled out by real-time PCR. Oral acyclovir (ACV) 400 mg was started at two times a day for 2 weeks after consulting his gastroenterologist followed by once daily for 6 months, and topical dexamethasone 0.01% was stepped up to six times a day and tapered with careful monitoring of his IOP. At 6 months, visual acuity was maintained at 20/20, N6. Slit-lamp examination showed a quiet eye and faint scarring of the lesions [Figure 2]a and [Figure 2]b. The AS-OCT showed corresponding reflectivity of the scars [Figure 2]c and [Figure 2]d. He continued to report recurrent redness and discomfort on stopping the eye drops and had to continue the same on an as per need basis. The nummular lesions did not recur even at 1 year. Both his irritable bowel disease and spondyloarthropathy were well controlled.
Figure 1: (a) Multiple flat, nummular lesions from the limbu s up to central cornea, with relatively clear spaces (asterisks delineate the limbal lesion and the central edge). (b) Slit section subepithelial and anterior stromal location. (c) Anterior segment optical coherence tomography shows a placoid-hyperreflective lesion (asterisk). (d)Agarose gel electrophoresis showing HSV-1 amplicon (V-328/2020) in conventional PCR

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Figure 2: (a) Six-month follow-up shows resolution of the lesions. (b)Increased scarring is noted at the edge of the LASIK flap (asterisk). (c and d) Anterior segment optical coherence tomography shows linear-hyperreflective lesions located in the peripheral anterior stroma (asterisks) corresponding to scars

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  Discussion Top


Nummular keratitis is commonly associated with adenoviral keratitis.[2] The differential diagnosis includes herpes zoster opthalmicus,[3] Epstein–Barr virus,[4] post-varicella zoster virus (VZV) systemic infection,[5] and acanthamoeba keratitis, where subepithelial infiltrates similar to nummular lesions can be noted in the very early stages.[6] Other viral cases reported are archipelago keratitis,[7] which is a variant of superficial stromal keratitis in HSV.

Our patient had a history of undergoing LASIK 3 years ago. Reactivation of HSV infections after LASIK has been well reported in the literature.[8] However, the nummular variant has not been reported. Additionally. most of these reports pertain to onset within a few weeks to months after LASIK, and as in our case, it is unlikely to have any true causality.

The presence of immune disorders is intriguing in our case. The role of immune deviation in the propagation of the HSV-1 keratitis or its recurrences can be an underlying pathological mechanism here. Holland et al.[9] reported seven cases of bilateral HSV keratitis in their cohort of 544 cases and all were associated with some form of immune deviation, including one case of ankylosing spondylitis with Crohn's disease. They observed that these cases had more recurrences and needed prolonged therapy and had a final poorer visual outcome.

The role of polymerase chain reaction (PCR) to detect viral load in ocular surface specimens is well established.[10] We subjected aqueous fluid to real-time PCR for HSV-1, cytomegalovirus, and VZV, of which only HSV-1 was positive. The detection of the viral genome in aqueous may not be considered as conclusive evidence of etiology, but a positive PCR result is supportive of the etiological diagnosis in this case.


  Conclusion Top


To conclude, HSV-1 nummular keratitis is a rare entity and, as in our case, can be associated with underlying systemic immune deviation.

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, Hyderabad, Telangana, India.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Valerio GS, Lin CC. Ocular manifestations of herpes simplex virus. Curr Opin Ophthalmol 2019;30:525-31.  Back to cited text no. 1
    
2.
Rao SK, Srinivas SP, Nair AK. Nummular keratitis–A modern-day conundrum. TNOA J Ophthalmic Sci Res 2019;57:308-10.  Back to cited text no. 2
  [Full text]  
3.
Li JY. Herpes zoster ophthalmicus: Acute keratitis. Curr Opin Ophthalmol 2018;29:328-33.  Back to cited text no. 3
    
4.
Pinnolis M, McCulley JP, Urman JD. Nummular keratitis associated with infectious mononucleosis. Am J Ophthalmol 1980;89:791-4.  Back to cited text no. 4
    
5.
Denier M, Gabison E, Sahyoun M, Labetoulle M, Dureau P, Cochereau I, et al. Stromal keratitis after varicella in children. Cornea 2020;39:680-4.  Back to cited text no. 5
    
6.
Reinhard T, Schilgen G, Steinert M, Hacker J, Sundmacher R. Nummular infiltrates in acanthamoeba keratitis. Acta Ophthalmol Scand 2003;81:541-3.  Back to cited text no. 6
    
7.
Gabison EE, Alfonsi N, Doan S, Racine L, Sultan G, Baudouin C, et al. Archipelago keratitis: A clinical variant of recurrent herpetic keratitis? Ophthalmology 2007;114:2000-5.  Back to cited text no. 7
    
8.
Moshirfar M, Welling JD, Feiz V, Holz H, Clinch TE. Infectious and noninfectious keratitis after laser in situ keratomileusis: Occurrence, management, and visual outcomes. J Cataract Refract Surg 2007;33:474-83.  Back to cited text no. 8
    
9.
Holland EJ, Huang AJ. Bilateral herpetic keratoconjunctivitis. Ophthalmology 2003;110:493-6.  Back to cited text no. 9
    
10.
Subhan S, Jose RJ, Duggirala A, Hari R, Krishna P, Reddy S, et al. Diagnosis of herpes simplex virus-1 keratitis: Comparison of Giemsa stain, immunofluorescence assay and polymerase chain reaction. Curr Eye Res 2004;29:209-13.  Back to cited text no. 10
    


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