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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 4  |  Page : 640-642

Atypical keratitis caused by Leifsonia aquatica with “cracked windshield” appearance and disabling ocular pain


Tecnologico de Monterrey, School of Medicine and Health Sciences, Institute of Ophthalmology and Visual Sciences, Monterrey, Mexico

Date of Submission29-Sep-2020
Date of Acceptance15-Mar-2021
Date of Web Publication09-Oct-2021

Correspondence Address:
Dr. Julio C Hernandez-Camarena
Tecnologico de Monterrey, School of Medicine and Health Sciences, Institute of Ophthalmology and Visual Sciences, Monterrey, Av. Batallon de San Patricio #112. Col. Real de San Agustin, N.L. CP. 66278
Mexico
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_3115_20

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  Abstract 


Microbial keratitis is a potentially sight-threatening complication of contact lens (CL) wear. Microbiological investigations, close follow-up, and culture-guided treatment are mandatory to achieve a favorable outcome. Poor CL hygiene and exposure to contaminated water, associated with severe ocular pain, are hallmarks of Acanthamoeba keratitis (AK). However, AK is characterized by an insidious onset. We report a case of an acute onset CL-related keratitis associated with L. aquatica, a Gram-positive bacterium. A history of river water exposure and disabling ocular pain, which resembled AK but with unusual slit-lamp findings, made the diagnosis challenging.

Keywords: Bacterial keratitis, contact lens, Leifsonia aquatica


How to cite this article:
Hernandez-Camarena JC, Ruiz-Lozano RE, Andrade-Leal SE, Roman-Zamudio M, Valdez-Garcia JE. Atypical keratitis caused by Leifsonia aquatica with “cracked windshield” appearance and disabling ocular pain. Indian J Ophthalmol Case Rep 2021;1:640-2

How to cite this URL:
Hernandez-Camarena JC, Ruiz-Lozano RE, Andrade-Leal SE, Roman-Zamudio M, Valdez-Garcia JE. Atypical keratitis caused by Leifsonia aquatica with “cracked windshield” appearance and disabling ocular pain. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Oct 21];1:640-2. Available from: https://www.ijoreports.in/text.asp?2021/1/4/640/327659



There are approximately 40 million contact lens (CL) wearers in the United States, of whom 93% are soft CL users.[1],[2] Of those, more than 90% report at least one form of misusing their CLs, such as overnight use, poor lens hygiene, or exposure to potentially contaminated water sources including pools, rivers, and seawater.[3] The annual incidence of infectious keratitis ranges from two to 20 cases per 10,000 CL users, with an incidence of 25.4% among soft CL users.[1],[3] Despite bacterial keratitis being the most common form of CL-related infection, other microorganisms must be considered when atypical clinical presentations are observed. Herein we present the case of an atypical CL-related keratitis associated with Leifsonia aquatica, initially presenting with signs and symptoms that resembled other virulent microorganisms (e.g., Acanthamoeba). To our knowledge, no previous case of CL-related keratitis associated with Leifsonia aquatica has been reported. Informed consent was obtained to publish clinical images.


  Case Report Top


A 27-year-old woman presented to our cornea service with a 12-h history of disabling ocular pain and vision loss in her right eye (RE). A 10-year history of extended soft CL wear was relevant during the medical interrogation. She reported swimming in a river wearing her CLs three days earlier. The best-corrected visual acuity (BCVA) was counting fingers at 1-meter in the RE with 20/20 vision in the left eye (LE). Examination of the RE revealed a central corneal dendriform lesion with surrounding stromal edema. The lesion exhibited a “cracked windshield” appearance [Figure 1]. Corneal scrapes were routinely obtained using a calcium alginate swab and inoculated in five solid culture media (sheep blood agar, chocolate agar, mannitol salt agar, Sabouraud agar, and Löwenstein–Jensen medium) and a liquid medium (brain-heart infusion) under the appropriate atmospheric conditions (37°C/5% CO2). Gram, Giemsa, Ziehl-Neelsen, and Acridine orange stains were obtained as well. Empirical treatment with moxifloxacin 0.5% and natamycin 5% eyedrops every hour was prescribed. After 24 h, the patient denied feeling pain or having visual improvement. Cultures and stains were negative at that time. On day two, the corneal infiltrate and stromal edema worsened, and ill-defined satellite lesions were observed [Figure 2]a. Cultures yielded confluent growth of Leifsonia aquatica in sheep blood and chocolate agar, described by the microbiologist as convex formations with round borders, non-hemolytic, opaque, and with a yellow-creamy coloration. The organism was identified with 96% probability as Leifsonia aquatica using the API Coryne system (bioMérieux, Marcy l'Etoile, France) and confirmed with matrix-assisted laser desorption ionization time-of-flight mass spectrometry (MALDI-TOF MS). Antibiogram showed resistance to penicillin G (MIC >32 μg/ml), intermediate susceptibility to vancomycin, meropenem, and imipenem (MIC 4 μg/ml), and acceptable sensitivity to cefotaxime (MIC 1 μg/ml) and linezolid (MIC 0.75 μg/ml). On day three, prior to any change of the antibiotic regimen, the patient reported experiencing pain and also improvement in vision (BCVA 20/200). A slit lamp exam revealed reduction in the corneal infiltrates and stromal edema [Figure 2]b. At this point, no changes were made to the antibiotic regimen. On day five, her pain significantly improved, and a slit lamp exam revealed a marked reduction in the corneal infiltrates and stromal edema [Figure 3]a. Moxifloxacin eyedrops were tapered to one drop every 4 hours and natamycin was suspended. After 2 weeks, the patient reported no pain and the BCVA improved to 20/80. Aiming to reduce corneal scarring, loteprednol etabonate 0.2% drops every 8 hours were prescribed. Up to day 30, fungal and non-nutrient agar cultures were still unrewarding, and the slit lamp evaluation revealed a mid-stromal leukoma in the RE with resolution of the peripheral infiltrates [Figure 3]b. BCVA was 20/50. At this point, moxifloxacin eyedrops were suspended. After 2 months, corneal findings and BCVA remained unchanged and topical steroids were suspended. Corneal OCT showed an anterior and mid-stromal opacification [Figure 3]c. A scleral lens trial and a deep anterior lamellar keratoplasty (DALK) were offered as potential solutions for improving her BCVA; however, the patient chose to delay further management.
Figure 1: Slit-lamp examination photographs of the right eye. Initial presentation showing mid-stromal dendritiform corneal infiltrate with stromal edema resembling a “cracked windshield”

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Figure 2: (a) Day 2. Right eye shows the increased size of the stromal infiltrate/edema with irregular borders and appearance of new satellite peripheral infiltrates. Moderate to severe conjunctival hyperemia is observed. (b) Day 3. Picture depicts density reduction of the central and peripheral infiltrates and improvement of the conjunctival hyperemia

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Figure 3: (a) Day 5. Right eye slit-lamp photograph shows improvement in density of infiltrates continues. (b) Day 30. A central stromal corneal infiltrate with no inflammatory infiltrates is observed. (c) Corneal optical coherence tomography of the right eye with thickness of 501 μm including epithelial, opacity, and residual thickness of 63 μm, 129 μm, 309 μm, respectively

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  Discussion Top


L. aquatica is a non-spore forming Gram-positive rod that resides in water habitats. Evidence of human infection is scant, with reported cases associated with septicemia in immunosuppressed, end-stage renal disease patients who are hemodialysis dependent.[4],[5] However, a case of systemic L. aquatica infection in a healthy patient who underwent surgery for retinal detachment is reported.[5] In our patient, the history of CL wear and exposure to potentially contaminated water associated with disabling ocular pain at presentation, as well as the atypical dendriform-like corneal infiltrate, raised suspicions of Acanthamoeba keratitis (AK).[2],[3] To complicate the situation, the clinical evolution and the posterior “cracked windshield” appearance surrounding the corneal infiltrate, as well as the emergence of satellite infiltrates with irregular margins, suggested a non-tuberculous mycobacteria (NTM) infection or fungal keratitis. Hence, in addition to topical moxifloxacin as an empirical treatment, topical natamycin was also initiated. In this regard, the acid-fast bacilli of mycobacteria species stain by the Ziehl-Nielsen method in almost 90% of the cases but was negative in our case; thus, this diagnostic tool conceivably aided with a quick dismissal of this etiology.[6],[7] In addition, the acute-onset severe pain at presentation was a clinical manifestation against fungal and NTM keratitis.[3] Regarding AK, approximately 85% of cases are related to soft CL use. Furthermore, CL wear, environmental water exposure, and severe pain made AK part of the differential diagnosis.[3] Also, at early stages, AK is usually confined to the corneal epithelium with pseudo-dendrites and punctate keratopathy. Lid edema and perineural infiltrates, present in up to 60% of AK cases, were not present in our case.[8] Furthermore, the acute disease onset is not a typical feature of AK keratitis. Therefore, since cultures were positive only for L. aquatica and clinical improvement with moxifloxacin was observed, we decided not to use further treatment or include any of the antibiotics proven to be effective on the susceptibility tests (i.e., cefotaxime or linezolid), and, at the moment, we also decided to discontinue natamycin and observe the clinical course. The use of topical steroids, although controversial, was initiated, aiming for reduction of corneal scarring after 2 weeks of continuous improvement, resolution of the corneal infiltrates, and visual acuity stabilization.

The data we have presented here have been limited by the absence of PCR and/or RNA sequencing methods to obtain a definitive molecular identification of the microorganism. Also, since our patient responded favorably to empiric therapy with moxifloxacin, which was not included in the susceptibility tests, we cannot rule out the presence of a nonidentified bacteria responsible for the keratitis, despite the confluent growth of Leifsonia aquatica in two different culture media. Nevertheless, the descriptions made by the microbiologists, which agree with the pattern and characteristics of culture growth in previous reports, and the patient's exposure to environmental water, the history of CL use, and the clinical picture rendered a high possibility for culture findings of Leifsonia aquatica.[4],[5] However, when available, PCR and ancillary molecular tests should be performed to identify the Leifsonia species.

Finally, as with other instances of atypical keratitis, the recommendation in the scenario of a patient with a history of CL use and water or soil exposure, corneal infiltrates, and pain is a full microbiological evaluation, considering unusual pathogens such as Acanthamoeba and NTM; also, judicious empirical therapy must be performed. Despite the expanded armamentarium of anti-infective drugs to manage CL-related microbial keratitis, patient education regarding the acceptable use of CLs is of utmost importance. Avoiding exposure to potentially contaminated water bodies (e.g., oceans, streams, pools, natural ponds) while wearing CLs, proper routine cleaning with proven CL solutions, limiting the hours of daily use, periodic eye examinations and exchange of CLs, and avoiding nighttime use are mandatory recommendations for CL users to decrease the risk of associated complications.[8]


  Conclusion Top


Ultimately, this case has the goal of encouraging general ophthalmologists and cornea specialists to consider unusual presentations of known diseases and etiologies (in this case, keratitis associated with a rare Gram-positive microorganism) before deliberating over unusual etiologies (AK or NTM keratitis), to perform the clinical exercise of identifying key clinical features of microorganisms that may carry a severe eye-threatening clinical course, to always address eye infections (including keratitis) by conducting an orderly and exhaustive microbiological investigation, and to observe in detail (preferably with photographic documentation) the clinical course before making changes in the antimicrobial therapy.

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.
Fleiszig SMJ, Kroken AR, Nieto V, Grosser MR, Wan SJ, Metruccio MME, et al. Contact lens-related corneal infection: Intrinsic resistance and its compromise. Prog Retin Eye Res 2020;76:100804.  Back to cited text no. 1
    
2.
Cope JR, Collier SA, Rao MM, Chalmers R, Mitchell GL, Richdale K, et al. Contact lens wearer demographics and risk behaviors for contact lens-related eye infections--United States, 2014. MMWR Morb Mortal Wkly Rep 2015;64:865-70.  Back to cited text no. 2
    
3.
Alipour F, Khaheshi S, Soleimanzadeh M, Heidarzadeh S, Heydarzadeh S. Contact lens-related complications: A review. J Ophthalmic Vis Res 2017;12:193-204.  Back to cited text no. 3
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4.
Porte L, Soto A, Andrighetti D, Dabanch J, Braun S, Saldivia A, et al. Catheter-associated bloodstream infection caused by Leifsonia aquatica in a haemodialysis patient: A case report. J Med Microbiol 2012;61:868-73.  Back to cited text no. 4
    
5.
Han L, Lei JE, Wang X, Guo LT, Kang QY, He L, et al. Septicemia caused by Leifsonia aquatica in a healthy patient after retinal reattachment surgery. J Clin Microbiol 2013;51:3886-8.  Back to cited text no. 5
    
6.
Kheir WJ, Sheheitli H, Abdul Fattah M, Hamam RN. Nontuberculous mycobacterial ocular infections: A systematic review of the literature. Biomed Res Int 2015;2015:164989.  Back to cited text no. 6
    
7.
Girgis DO, Karp CL, Miller D. Ocular infections caused by non-tuberculous mycobacteria: Update on epidemiology and management. Clin Exp Ophthalmol 2012;40:467-75.  Back to cited text no. 7
    
8.
Carnt N, Stapleton F. Strategies for the prevention of contact lens-related Acanthamoeba keratitis: A review. Ophthalmic Physiol Opt 2016;36:77-92.  Back to cited text no. 8
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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