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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 376-378

Severe marginal sterile keratitis in a patient with staphylococcal endophthalmitis after phacoemulsification


1 Department of Ophthalmology, National Taiwan University Hospital, Taipei, Taiwan
2 Department of Ophthalmology, National Taiwan University Hospital, Taipei, Taiwan; Hamilton Glaucoma Center, Shiley Eye Institute and Viterbi Family Department of Ophthalmology, University of California, San Diego, California, USA
3 Department of Ophthalmology; Advanced Ocular Surface and Corneal Nerve Research Center, National Taiwan University Hospital; Department of Ophthalmology, College of Medicine, National Taiwan University, Taipei, Taiwan

Date of Submission19-Apr-2021
Date of Acceptance19-Oct-2021
Date of Web Publication13-Apr-2022

Correspondence Address:
Wei-Li Chen
Department of Ophthalmology, National Taiwan University Hospital, No. 7, Chun-Shan S. Rd., Taipei, 100
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_924_21

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  Abstract 


Postoperative endophthalmitis is sight-threatening, severe inflammation in posterior vitreous cavity and anterior chamber. Marginal keratitis with infiltrates or ulcers following endophthalmitis can be infectious or sterile, but the etiologies related to intraocular infection have rarely been reported. Here we present an unusual case of severe marginal keratitis with sterile infiltrates concomitant with staphylococcal endophthalmitis after phacoemulsification. Creamy white ring infiltrates at the corneolimbal junction was found with severe vitritis. With antibiotics treatment, endophthalmitis improved while the corneal infiltrates progressed. After corneal specimens yielded negative results, the corneal infiltrates improved under proper topical corticosteroid without marked sequelae. In conclusion, noninfectious, immune-related marginal sterile keratitis may develop in patients with staphylococcal endophthalmitis after phacoemulsification.

Keywords: Cornea, endophthalmitis, immunological reaction, marginal sterile keratitis, staphylococcus


How to cite this article:
Hou YT, Hsieh BJ, Wu JH, Chen WL. Severe marginal sterile keratitis in a patient with staphylococcal endophthalmitis after phacoemulsification. Indian J Ophthalmol Case Rep 2022;2:376-8

How to cite this URL:
Hou YT, Hsieh BJ, Wu JH, Chen WL. Severe marginal sterile keratitis in a patient with staphylococcal endophthalmitis after phacoemulsification. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 18];2:376-8. Available from: https://www.ijoreports.in/text.asp?2022/2/2/376/343002



Marginal sterile keratitis is a form of peripheral curvilinear infiltrates in superficial cornea located at the peripheral cornea where lids cross. It can be associated with the inflammatory reaction against staphylococcus antigen and the long-standing presence of blepharoconjunctivitis.[1] The pathogenesis of marginal sterile keratitis is postulated to be associated with the reactions to corneal antigens, circulating immune complex deposition, and hypersensitivity reactions to exogenous antigens.[2] Unlike the avascular central cornea, the limbus and peripheral cornea receive abundant blood supply from capillary arcades, allowing for the accumulation of autoantibodies and other immune complexes.[3] Here, we present an unusual case of severe marginal sterile keratitis with corneal infiltrates concomitant with staphylococcal endophthalmitis after phacoemulsification.


  Case Report Top


A 65-year-old woman presented to the emergency department 3 days after smooth phacoemulsification appeared in her left eye. She had no systemic diseases, nor any autoimmune diseases, and no history of blepharitis or eyelid problems associated with ocular complications. Slit-lamp examination was unremarkable, and no corneal wound leakage was noted on postoperative day 1. The initial examination revealed left eye visual acuity of hand motion, and intraocular pressure was 12 mmHg. Slit-lamp examination showed corneal edema with creamy white ring-like infiltrates at the upper corneolimbal junction and satellite infiltrates at the lower corneolimbal junction without epithelial defect [Figure 1]a and [Figure 1]b. Anterior chamber and vitreous revealed severe inflammatory reaction. The posterior chamber intraocular lens was in place, but the fundus was not visible due to the dense vitritis. Postoperative infectious endophthalmitis was suspected, and emergency pars plana vitrectomy (PPV) combined with retina photocoagulation, posterior capsulotomy, and intravitreal injection of vancomycin and ceftazidime were performed. Postoperatively, the patient was prescribed the topical fortified antibiotics vancomycin (33 mg/mL) and ceftazidime (50 mg/mL) given hourly, alongside oral levofloxacin (500 mg) taken twice daily. Vitreous culture yielded Staphylococcus epidermidis, although the smear and culture of corneal infiltrates had negative findings. Over the course of a week, the inflammation in the vitreous and anterior chamber improved gradually. However, the peripheral corneal infiltrates progressed and spread circumferentially along the limbus [Figure 2]a,[Figure 2]b,[Figure 2]c,[Figure 2]d. Repeated smears and cultures of the corneal infiltrates were performed, with negative results obtained. Laboratory testing for autoimmune diseases, including complete blood count and differential count, rheumatoid factor, C3 C4, C-reactive protein, antinuclear antibody, erythrocyte sedimentation rate, was done. All laboratory results were within normal limits. Marginal sterile keratitis with corneal infiltrate which is caused by hypersensitivity to staphylococcal antigen was therefore suspected. Topical usage of 1% prednisone acetate eye drops (Pred Forte, Allergan, Irvine, CA) four times per day, with tapering of a topical antibiotic, was prescribed. Marked improvement of corneal infiltrates was observed in the following few days [Figure 3]a and [Figure 3]b. After 3 weeks of topical steroid treatment, the corneal lesion and the intraocular inflammation were almost completely resolved. The patient's vision recovered to 20/60 without recurrence of corneal and intraocular inflammation during the 2-year follow-up period, and only mild peripheral corneal thinning and opacity were reported.
Figure 1: (a and b) Three days after phacoemulsification, corneal edema with creamy white ring-like infiltrates at upper corneolimbal junction, and satellite infiltrates at the lower corneolimbal junction were found. No associated epithelial defects were noted. Mild corneal neovascularization was found with some intracorneal hemorrhage in the upper corneolimbal junction

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Figure 2: (a-d) Three days after PPV and the intravitreal and topical application of fortified antibiotics, the peripheral corneal infiltrates progressed and spread circumferentially along the limbus, despite the improvement of intraocular inflammation. No epithelial defect was noted on top of the peripheral corneal infiltration

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Figure 3: (a and b) Seven days after the application of a topical steroid, the peripheral corneal infiltrate completely resolved

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  Discussion Top


Infectious endophthalmitis after phacoemulsification is a serious, vision-threatening intraocular inflammation, usually resulting from the exogenous spread of microorganisms into the eye. According to a previous review, the incidence ranges from 0.02% to 0.71%.[4] Bacteria and fungi were the most common pathogens. The primary sources of most pathogens are the patients' eyelids and conjunctiva, which usually harbor coagulase-negative staphylococci, Staphylococcus aureus, Propionibacterium acnes, and Streptococcus species. Other less common but potentially important sources of pathogens include defects in sterilization techniques; and contamination of surgical instruments.[5] For staphylococcal infection following phacoemulsification, the most common strain observed is the coagulase-negative staphylococci, although the more virulent infections usually involve Staphylococcus aureus.[6]

In this case, we collected the vitreous sample via vitrectomy under aseptic technique. The pathogen yielded from vitreous was Staphylococcus epidermidis. Nevertheless, repeated cultures from the corneal infiltrate yielded negative results. The incidence of postoperative infectious keratitis is much lower than that of postoperative endophthalmitis, and it often presents as an infiltration on the clear corneal wound.[7] However, the corneal lesion in our case had no direct contact with the corneal wound, implying the possibility of a noninfectious keratitis. The improvement of the corneal infiltrates after the application of a topical steroid also indicated a possible immune-related reaction. Therefore, sterile marginal keratitis was suspected.

Marginal sterile keratitis is considered as the product of an inflammatory reaction to staphylococcal antigens instead of a direct staphylococcal corneal infection. Corneal ulcer scraping revealed absence of organisms, while Staphylococcus aureus is frequently isolated from the eyelid margin, with long-standing staphylococcal blepharitis.[8] A study of 180 marginal catarrhal ulcers identified 133 positive findings of coagulase positive Staphylococcus aureus over the eyelid and conjunctiva, suggesting a type III hypersensitivity.[9] Immune complex at the peripheral cornea formed by antigen in the tear film and antibody from limbal vessels may have activated the complement pathway, resulting in peripheral stromal opacity with epithelial breakdown. Considering the positive result of Staphylococcus epidermidis in vitreous sample without significant staphylococcal blepharitis, the severe inflammation underlying endophthalmitis, and the predisposition of the peripheral cornea to immune reaction, we suspected that the antigen of Staphylococcus epidermidis might have resulted in marginal sterile keratitis-like hypersensitivity in the patient's peripheral cornea.

We obtained several unique findings; our case stood out from a typical marginal keratitis case. First, the patient had no history of blepharitis, and the marginal infiltrates did not occur before phacoemulsification. Second, the nearly confluent, 360° marginal infiltrates were different from the typical marginal sterile keratitis usually found in 2, 4, 8, and 10 o'clock positions. Third, the coincidental presentation of staphylococcal endophthalmitis and the treatment response to steroids pointed to a possible immunological relationship between the lesions and the staphylococcal antigen, instead of an infectious etiology.


  Conclusion Top


In conclusion, the possibility of marginal sterile keratitis with corneal infiltrate should be considered in patients with staphylococcal endophthalmitis after phacoemulsification. Detailed history-taking and careful differential diagnosis through microbiological work-up are vital. Topical steroid usage and careful monitoring of possible infectious conditions are crucial steps in treating this rare disease.

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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Hoffman J, Hassan A. Severe staphylococcal marginal keratitis presenting with hypopyon. BMJ Case Rep 2015;2015:bcr2015211979.  Back to cited text no. 1
    
2.
Boto-de-los-Bueis A, del Hierro Zarzuelo A, García Perea A, de Pablos M, Pastora N, Noval S. Staphylococcus aureus blepharitis associated with multiple corneal stromal microabscess, stromal edema, and uveitis. Ocul Immunol Inflamm 2015;23:180-3.  Back to cited text no. 2
    
3.
Dana MR, Qian Y, Hamrah P. Twenty-five-year panorama of corneal immunology: Emerging concepts in the immunopathogenesis of microbial keratitis, peripheral ulcerative keratitis, and corneal transplant rejection. Cornea 2000;19:625-43.  Back to cited text no. 3
    
4.
Nowak MS, Grzybowski A, Michalska-Małecka K, Szaflik JP, Kozioł M, Niemczyk W, et al. Incidence and characteristics of endophthalmitis after cataract surgery in Poland, during 2010-2015. Int J Environ Res Public Health 2019;16:2188.  Back to cited text no. 4
    
5.
Speaker MG, Milch FA, Shah MK, Eisner W, Kreiswirth BN. Role of external bacterial flora in the pathogenesis of acute postoperative endophthalmitis. Ophthalmology 1991;98:639-49; discussion 650.  Back to cited text no. 5
    
6.
Gentile RC, Shukla S, Shah M, Ritterband DC, Engelbert M, Davis A, et al. Microbiological spectrum and antibiotic sensitivity in endophthalmitis: A 25-year review. Ophthalmology 2014;121:1634-42.  Back to cited text no. 6
    
7.
Cosar CB, Cohen EJ, Rapuano CJ, Laibson PR. Clear corneal wound infection after phacoemulsification. Arch Ophthalmol 2001;119:1755-9.  Back to cited text no. 7
    
8.
Cohn H, Mondino BJ, Brown SI, Hall GD. Marginal corneal ulcers with acute beta streptococcal conjunctivitis and chronic dacryocystitis. Am J Ophthalmol 1979;87:541-3.  Back to cited text no. 8
    
9.
Thygeson P. Marginal corneal infiltrates and ulcers. Trans Am Acad Ophthalmol Otolaryngol 1947;51:198-209.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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