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Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 49-51

An unusual case of Candida parapsilosis causing infectious crystalline keratopathy in Steven–Johnson syndrome

1 Department of Ophthalmology, Army Hospital (Research and Referral), New Delhi, India
2 Department of Cornea & Anterior Segment Services, Army Hospital (Research and Referral), New Delhi, India
3 Department of Ophthalmology, Command Hospital, Lucknow, Uttar Pradesh, India

Date of Submission03-Jun-2021
Date of Acceptance17-Aug-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Prof. Alok Sati
Professor and Senior Advisor, Cornea and Anterior Segment Services, Department of Ophthalmology, Army Hospital (Research and Referral), New Delhi - 110 001
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_1544_21

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A 41-year-old female, a known case of Steven–Johnson syndrome (SJS) with ocular involvement, had a pthisical right eye (RE) and a history of mucous membrane grafting (MMG) in the left eye. Two weeks post-MMG, the patient developed a pearly white crystalline anterior stromal infiltrate suggestive of infectious crystalline keratopathy (ICK). The Gram stain showed gram-positive, round-to-oval budding cells and the culture on sheep blood agar showed smooth white yeast colonies. VITEK 2 compact automated microbiology system-enabled species-level identification was Candida parapsilosis in addition to the antifungal sensitivity. Based on the sensitivity report, the patient responded well to oral fluconazole and topical voriconazole with corneal scar formation. Clinical and microbiological evaluations of ICK in an unusual setting like SJS is prudent for a successful outcome.

Keywords: Candida parapsilosis, infectious crystalline keratopathy, Steven–Johnson syndrome

How to cite this article:
Mishra P, Mishra SK, Sati A, Kumar SV, Kumar P, Chauhan R. An unusual case of Candida parapsilosis causing infectious crystalline keratopathy in Steven–Johnson syndrome. Indian J Ophthalmol Case Rep 2022;2:49-51

How to cite this URL:
Mishra P, Mishra SK, Sati A, Kumar SV, Kumar P, Chauhan R. An unusual case of Candida parapsilosis causing infectious crystalline keratopathy in Steven–Johnson syndrome. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Aug 14];2:49-51. Available from: https://www.ijoreports.in/text.asp?2022/2/1/49/334886

Infectious crystalline keratopathy (ICK), an uncommon, indolent corneal pathology, reported by Gorovoy in 1983, is characterized by arborizing, needle-like, intrastromal opacities in the absence of apparent inflammation.[1] Multiple organisms including fungal and bacterial entities have been implicated in ICK, however, Candida parapsilosis (C. parapsilosis) has so far been reported only thrice in varied settings, i.e., phacoemulsification, penetrating keratoplasty, and Descemet Stripping Automated Endothelial Keratoplasty (DSAEK).[2] Herein, we report for the first time C. parapsilosis causing ICK in a patient with Steven–Johnson syndrome (SJS) which was managed successfully with medical management and the infection was controlled.

  Case Report Top

In 2018, this 41-year-old female developed SJS following oral intake of an antibiotic and developed bilateral chronic ocular surface disease as its sequelae. On her first visit to our institute, her right eye (RE) was pthisical whereas the left eye (LE) had severe blepharospasm for which she underwent mucous membrane grafting (MMG) in both the upper and lower eyelids. Post-MMG, she was prescribed eye drops, prednisolone acetate, 1% six times a day, cyclosporine 0.05% twice daily, and preservative-free lubricating eye drops. The patient responded to the above management with marked improvement in the symptoms and her distant visual acuity (DVA) improved from 1/60 to 4/60. Two weeks later, the patient complained of painful diminution of vision and severe photophobia in the LE. The ocular examination of the LE revealed DVA as Counting Fingers Close to Face (CFCF), MMG in situ, mild conjunctival congestion, central corneal epithelial defect measuring 3 mm × 3.5 mm overlying a pearly white anterior stromal crystalline infiltrate with multiple needle-like projections [Figure 1]. The lens was cataractous and details of the posterior segment could not be visualized.
Figure 1: Slit-lamp photograph showing features suggestive of infectious crystalline keratopathy

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The corneal scrapings from the lesion were subjected to smear preparation and culture sensitivity. The smear preparation was done for the Potassium Hydroxide (KOH) mount and Gram stain, and the culture was inoculated on sheep blood agar, chocolate agar, and Sabouraud dextrose agar (SDA). The Gram stain showed multiple, gram-positive, round, budding cells in clusters and scattered singly [Figure 2], and the culture on the sheep blood agar showed smooth white yeast colonies with discrete margins 24 h after incubation at 37°C [Figure 3]. Antifungal sensitivity and species identification as C. parapsilosis were done using yeast reagent card on VITEK 2 compact automated microbiology system. The organism was sensitive to fluconazole ( Minimum Inhibitory Concentration [MIC]: <=0.5), voriconazole (MIC: <=0.12), caspofungin (MIC: 0.5), micafungin (MIC: 1.0), amphotericin (MIC: 0.5), and flucytosine (MIC: <=1.0).
Figure 2: Gram stain photograph showing multiple gram-positive, round, budding cells in clusters and scattered singly

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Figure 3: Creamy white colonies on sheep blood agar showing Candida spp

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Based on the antifungal sensitivity report, treatment was stepped up with the addition of tab fluconazole (150 mg, twice daily), eye drops, voriconazole (1%, 1 hourly) and homatropine (2% three times a day). One week later, voriconazole was tapered to 2 hourly, and at 6 weeks, the infected area was replaced by scar tissue [Figure 4], however, voriconazole and tab fluconazole were continued for 4 more weeks to ensure complete eradication of the Candida spp.
Figure 4: Slit-lamp photograph showing scar formation after the treatment

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  Discussion Top

C. parapsilosis, an otherwise normal human commensal, acquires pathogenicity through the hydrolytic enzymes by adhering to biotic/abiotic surfaces and through the formation of a biofilm.[2] Although a majority of fungal corneal infections are by filamentous fungi, compromised corneas along with multiple alterations in host defense are prone to yeast infections, usually by Candida spp.[3],[4] The most common isolated yeast in infections is Candida albicans, however, it seems that C. parapsilosis is being found in increasing frequency.[5] ICK is a distinctive clinical entity and is commonly associated with Streptococcus spp, however, though extremely rare, there are case reports of clinical ICK from the culture-proven C. parapsilosis.[2]

Despite vulnerability to microbial keratitis, keratitis due to fungi is not very common in SJS as has been observed in the studies discussing the profile of microbial keratitis in SJS.[6] In addition, the reporting of keratitis due to Candida in SJS is rare and so far no case has been reported wherein C. parapsilosis was isolated as the causative organism.

For C. parapsilosis to cause ICK, a compromised cornea and localized immunosuppression are required,[7] which in our patient was probably attributed to lid keratinization causing central corneal epithelial defect, chronic dry eyes, and prolong usage of topical steroids.

Apart from the rarity of ICK in SJS, ICK is also difficult to treat considering the tenacious nature of the causative organism. The sensitive drug needs to be administered in a higher concentration to have an effect on an organism inside the biofilm.[8] Based on the above facts our diagnostic and therapeutic armamentarium included a meticulous microbiological evaluation and administration of voriconazole on an hourly basis initially and two hourly subsequently to have adequate therapeutic concentration inside the biofilm.

  Conclusion Top

ICK in SJS is not a common presentation and isolating C. parapsilosis as the causative organism is even rarer. Managing a patient of SJS is a multidisciplinary approach and by the time the patient is referred to an ophthalmologist, a majority of these patients already have late-onset ocular complications.[9] Adding to the delay in diagnosing and instituting appropriate treatment is the clinical presentation of C. parapsilosis keratitis which shows a great diversity also. Clinical features suggestive of ICK may be attributed to multiple organisms that are more commonly isolated than C. parapsilosis, so the need for a complete microbiological examination of microbial keratitis in compromised corneas should always be emphasized.[7]

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.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Gorovoy MS, Stern GA, Hood I, Allen C. Intrastromal noninflammatory bacterial colonization of a corneal graft. Arch Ophthalmol 1983;101:1749-52.  Back to cited text no. 1
Sati A, Singh L, Mishra SK, Kumar SV, Dhar SK. Infectious crystalline keratopathy caused by Candida parapsilosis in a turgid host cornea. Indian J Ophthalmol 2021;1:246-8.  Back to cited text no. 2
Chirinos-Saldana P, Bautista de Lucio VM, Hernandez-Camarena JC, Navas A, Ramirez-Miranda A, Vizuet-Garcia L, et al. Clinical and microbiological profile of infectious keratitis in children. BMC Ophthalmol 2013;13:54.  Back to cited text no. 3
Tanure MA, Cohen EJ, Sudesh S, Rapuano CJ, Laibson PR. Spectrum of fungal keratitis at Wills Eye Hospital, Philadelphia, Pennsylvania. Cornea 2000;19:307-12.  Back to cited text no. 4
Rhem MV, Wilhelmus KR, Fornt RL. Infectious crystalline keratopathy caused by Candida parapsilosis. Cornea 1996;15:543-4.  Back to cited text no. 5
Sharma N, Venugopal R, Singhal D, Maharana PK, Sangwan S, Satpathy G. Microbial keratitis in Stevens-Johnson syndrome: A prospective study. Cornea 2019;38:938-42.  Back to cited text no. 6
Bourcier T, Touzeau O, Thomas F, Chaumeil C, Baudrimont M, Borderie V, et al. Candida parapsilosis keratitis. Cornea 2003;22:51-5.  Back to cited text no. 7
Fulcher TP, Dart JK, McLaughlin-Borlace L, Howes R, Matheson M, Cree I. Demonstration of biofilm in infectious crystalline keratopathy using ruthenium red and electron microscopy. Ophthalmology 2001;108:1088-92.  Back to cited text no. 8
Kompella VB, Sangwan VS, Bansal AK, Garg P, Aasuri MK, Rao GN. Ophthalmic complications and management of Stevens-Johnson syndrome at a tertiary eye care centre in south India. Indian J Ophthalmol 2002;50:283-6.  Back to cited text no. 9
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