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      A Rare Microsporidial Infection in Lamellar Corneal Tissue, following Transepithelial Photorefractive Keratectomy

      case-report

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          Abstract

          The aim of this study was to report a unique case of microsporidial keratitis over deep anterior lamellar keratoplasty after transepithelial photorefractive keratectomy surgery that was successfully treated with therapeutic lamellar keratoplasty without recurrence at King Khaled Eye Specialist Hospital in Riyadh, Saudi Arabia. The patient presented with recurrent attacks of eye pain, redness, photophobia, and decreased vision. The patient was initially treated as a case of presumed herpetic keratouveitis using antiviral medication and topical steroids with partial improvement. During the last episode, the condition deteriorated and patient underwent therapeutic lamellar keratoplasty. Histopathology indicated an infected graft with evidence of microsporidial infection. The patient was discharged with complete corneal epithelial healing and no signs of recurrence during follow-up. Microsporidial infection is a rare cause of stromal keratitis that affects both immunocompetent and immunosuppressed patients. Microsporidia should be suspected after surface ablation refractive surgery if the patient presents with recurrent symptoms of keratoconjunctivitis or stromal keratitis that are partially responsive to topical steroid therapy.

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          Most cited references15

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          Emerging prevalence of microsporidial keratitis in Singapore: epidemiology, clinical features, and management.

          To investigate the incidence and epidemiologic factors involved in the development of microsporidial keratitis. The association of host immune status and clinical pattern, clinical features, and the role of fluoroquinolone monotherapy in treatment are also examined. Retrospective, noncomparative case series. All cases (124 patients, 134 eyes) of microsporidial keratitis confirmed with modified trichrome stain positive of corneal scrape over a 4-year period. Epidemiologic factors were observed. Host immune status with human immunodeficiency virus (HIV) serology and CD4/CD8 analysis was performed when consent was obtained. Visual acuity (VA) and slit-lamp examination throughout the course of keratitis was recorded. Treatment used included topical fluoroquinolones (ciprofloxacin 0.3%, moxifloxacin 0.5%, gatifloxacin 0.5%, levofloxacin 0.5%, or norfloxacin 0.3%) as monotherapy or in combination with topical fumagillin and/or systemic albendazole. Where corneal edema developed, ultrasound corneal pachymetry was recorded. Demographic features and epidemiologic factors, including host immune status. Clinical features and disease course, including the response to different therapeutic regimes. Patients ranged in age from 11 to 68 years (mean, 31.9; median, 30) with a male:female ratio of 8:1 (females n = 17 [13.7%]). We performed HIV serology and CD4/CD8 in 45.9% of cases (n = 57); all the cases tested were negative with normal T-cell indices. Epidemiologic factors included soil exposure (50%), contact lens wear (21.1%), and topical steroid treatment (17.1%). The VA on presentation ranged from 20/20 to 20/100 (median, 20/30) with no loss in lines of VA on resolution. Common features were follicular papillary conjunctivitis and coarse punctate epithelial lesions in 3 patterns--diffuse, peripheral, and paracentral--evolving into nummular keratitis before resolution. Resolution occurred in 99% of cases on topical fluoroquinolone monotherapy. Four patients had recurrent disease that resolved with repeat fluoroquinolone or fluoroquinolone/oral albendazole combination. Two new clinical features were identified--diffuse endotheliitis (19.4%) with corneal edema and limbitis. This study identifies an increasing incidence of microsporidial keratitis in Singapore with a strong correlation with prior soil exposure. Diffuse endotheliitis and limbitis have not been described and resolves with topical steroid therapy. Topical fluoroquinolone monotherapy is a valid treatment option.
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            Microsporidial keratitis: need for increased awareness.

            Since the devastation of the European silk worm industry in the 19th century, microsporidia have been recognized as important organisms. An enormous literature is available on their biology, phylogeny, classification, disease profile, diagnosis, and treatment; however, it is only recently that ophthalmologists have begun to take note of these organisms. The last two decades have seen several publications related to ocular microsporidiosis, in particular those forms affecting the cornea. Both immunocompetent and immunocompromised patients are at risk of developing corneal infections that may range from self limiting mild keratoconjunctivitis to severe stromal keratitis recalcitrant to medical treatment. Exposure to soil, muddy water, and minor trauma are possible risk factors. Although reliable prevalence data are lacking, recent studies indicate a high prevalence of microsporidial keratoconjunctivitis in the rainy season, especially in India and other countries with similar climates. For instance, a high prevalence has been documented in Singapore. We bring together the information available on ocular microsporidiosis.
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              Microsporidial keratoconjunctivitis in healthy individuals: a case series.

              To present a series of 6 cases of microsporidial keratoconjunctivitis in healthy, nonimmunocompromised individuals. Retrospective, noncomparative case series. Six individuals with unilateral keratoconjunctivitis. Cornea epithelial scrapings were taken and evaluated by modified trichome staining. Blood was taken for human immunodeficiency virus (HIV) enzyme-linked immunosorbent assay in all cases and for CD4 and CD8 T-lymphocyte counts in 5 cases. The individuals were evaluated based on symptoms, visual acuity, slit-lamp biomicroscopy, and pathologic examination of the corneal scrapings. All cases occurred in men whose ages ranged from 16 to 37 years. Initial symptoms included unilateral pain and redness. All experienced subsequent worsening of symptoms and blurring of vision after using topical steroids prescribed by general practitioners. Slit-lamp biomicroscopy revealed coarse, multifocal, punctate epithelial keratitis in all 6 cases, anterior stromal infiltrates in 2 cases, with accompanying conjunctivitis in all cases. Modified trichrome staining of corneal epithelial scrapes revealed pinkish to red spores characteristic of microsporidia in all cases. Results of an HIV enzyme-linked immunosorbent assay were negative in all cases, and CD4 and CD8 T-lymphocyte counts and ratios were normal in all 5 tested cases. On diagnosis, topical steroid therapy was stopped in all cases. Treatment with topical Fumidil B (bicyclohexylammonium fumagillin; Leiter's Park Ave Pharmacy, San Jose, CA) together with oral albendazole was given in 3 cases, oral albendazole alone in a single case, and broad-spectrum antibiotic treatment with topical norfloxacin or chloramphenicol in two cases. Two cases had keratic precipitates with mild cellular activity in the anterior chamber and one such case was restarted subsequently on topical steroids. All six cases showed resolution of epithelial keratitis but with residual visually inconsequential subepithelial scars by the end of 1 month of treatment. Microsporidial keratoconjunctivitis can occur more commonly than expected in healthy, nonimmunocompromised individuals. Topical steroids seem to contribute to the persistence of this infection and may be a predisposing factor in these cases by creating a localized immunocompromised state. The clinical course is variable and may be self-limiting with cessation of topical steroid use.
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                Author and article information

                Journal
                Case Rep Ophthalmol
                Case Rep Ophthalmol
                COP
                COP
                Case Reports in Ophthalmology
                S. Karger AG (Basel, Switzerland )
                1663-2699
                4 April 2023
                Jan-Dec 2023
                4 April 2023
                : 14
                : 1
                : 127-133
                Affiliations
                [a ]Anterior Segment Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia
                [b ]College of Medicine, King Saud University, Riyadh, Saudi Arabia
                [c ]Pathology Department, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia
                Author notes
                Correspondence to: Rawan AlShabeeb, alshabeeb.rawan@ 123456gmail.com
                Article
                528894
                10.1159/000528894
                10074259
                37034379
                f35ea484-7019-4c1d-bf62-46a44484c4c7
                © 2023 The Author(s). Published by S. Karger AG, Basel

                This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC) ( http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.

                History
                : 14 May 2022
                : 20 December 2022
                : 2023
                Page count
                Figures: 3, References: 15, Pages: 7
                Funding
                The authors have no funding received.
                Categories
                Case Report

                microsporidia,stromal keratitis,keratoconjunctivitis,deep anterior lamellar keratoplasty

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