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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 53-56

Clinical profile of infectious crystalline keratopathy – Case series with varied risk factors and etiology


1 Fellow, Cornea and Refractive Services, Aravind Eye Hospital, Puducherry, India
2 Fellow, Vitreoretinal Services, Aravind Eye Hospital, Puducherry, India
3 Consultant, Cornea and Refractive Services, Aravind Eye Hospital, Puducherry, India

Date of Submission08-Feb-2021
Date of Acceptance30-Jun-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Dr. Josephine S Christy
Department of Cornea and Refractive Services, Aravind Eye Hospital, Cuddalore Road, Thavalakuppam . 605 007, Puducherry
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_341_21

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  Abstract 


This case series of six patients with infectious crystalline keratopathy (ICK) aims to highlight the varying risk factors, clinical presentation, and its outcome. We also report a post DALK (deep anterior lamellar keratoplasty) and post corneal tattooing infection uniquely presenting as ICK. The most common etiological organism (four out of six) was the Streptococcus species. Two other rare organisms identified were Burkholderia cepacia and Candida albicans. Five out of six patients healed with medical treatment with an overall average duration of 7 weeks (2–14 weeks), and only one patient required therapeutic penetrating keratoplasty.

Keywords: Burkholderia cepacia, corneal tattoo infection, DALK infection, infectious crystalline keratopathy


How to cite this article:
Khanna V, Chhabra K, Christy JS. Clinical profile of infectious crystalline keratopathy – Case series with varied risk factors and etiology. Indian J Ophthalmol Case Rep 2022;2:53-6

How to cite this URL:
Khanna V, Chhabra K, Christy JS. Clinical profile of infectious crystalline keratopathy – Case series with varied risk factors and etiology. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Jan 23];2:53-6. Available from: https://www.ijoreports.in/text.asp?2022/2/1/53/334942



Infectious crystalline keratopathy (ICK) is characterized by needle-like branching opacities and an absence of corneal or anterior segment inflammation.[1] Because of its rare occurrence, understanding of this disease has been predominantly from case reports. In this case series of six patients, we aim to describe the newer possible risk factors, various types of clinical presentation, and treatment outcomes.


  Case Series Top


The retrospective study included six patients who were diagnosed as ICK in a tertiary eye care hospital in South India from 2015 to 2020. Diagnosis of ICK was based on clinical examination. The patients were advised to discontinue topical steroids and were initiated on empirical 0.5% moxifloxacin eye drops hourly along with cycloplegics. Following culture growth, therapy was modified according to the antibiotic sensitivity report and clinical response. Patients not responding to topical medications underwent therapeutic penetrating keratoplasty (TPK).

The average age of presentation was 59 years (44–72 years). Five out of six patients were males. Ocular comorbidities in the affected eye included secondary glaucoma post penetrating keratoplasty (PKP) in three patients and pseudophakic bullous keratopathy (PBK) in one patient. Risk factors such as previous full-thickness keratoplasty were seen in three patients and deep anterior lamellar keratoplasty (DALK) in one patient [Figure 1]a, [Figure 1]b, [Figure 1]c. Three patients had a history of prolonged usage of bandage contact lens for decompensated cornea, whereas one patient had a history of corneal tattooing 6 weeks before presentation [Figure 2]c and [Figure 2]d. Five out of six patients were on topical steroids for an average period of 12 months (5 months to 3 years), and three patients were on antiglaucoma medications for an average period of 21 months (3–5 years) [Table 1]. The average time between the last corneal procedure and the development of ICK was 7.3 months (1.5 months to 2 years).
Figure 1: Streptococcus viridans keratitis – Case 2: (a) Inferior dense stromal infiltrate with needle-like branching borders; (b) full-thickness stromal involvement at third week; (c) corneal scar with vascularization at 14th week

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Figure 2: Burkholderia cepacia keratitis – Case 1: (a) Superficial crystalline stromal infiltrate at presentation; (b) healed ulcer at 6 weeks with residual 1 mm epithelial defect and stromal scarring. Candida albicans keratitis – Case 6: (c) Superficial infiltrate with crystalline borders; (d) complete healing with loss of tattoo pigments at sixth week

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Table 1: Summary of Casewise Demographics, Risk Factors, Clinical Characteristics, and Final Outcome

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Four out of six patients presented with an epithelial defect, and all patients had stromal infiltrate with the characteristic needle-like branching opacities [Figure 1]a, [Figure 2]a, [Figure 2]c and [Figure 3]a, [Figure 3]b. Intracorneal hypopyon was present in two patients [Figure 3]a and [Figure 3]c. Organisms isolated on culture were Streptococcus viridans in two, Streptococcus pneumoniae in two, Burkholderia cepacia, and Candida albicans in one patient each. Although most of the organisms showed drug resistance to commonly used first-line antibiotics, all patients recovered with appropriate antimicrobial treatment [Figure 2]b except for one patient, who needed TPK. The average duration of topical treatment was 7 weeks (2–14 weeks) [Table 2].
Figure 3: Streptococcus pneumoniae keratitis – Case 5: (a) Anterior stromal infiltrate with characteristic crystalline borders and intracorneal hypopyon. Streptococcus viridans keratitis – Case 3: (b) Inferior anterior stromal infiltrate with fine crystalline borders; (c) intracorneal hypopyon at third week

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Table 2: Summary of Microbiological Analysis and Antibiotic Sensitivity Pattern

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  Discussion Top


ICK is a rare form of indolent infectious keratitis. Although the true incidence of ICK is difficult to determine, an increase in ICK cases has been recorded recently due to increase in PKP surgeries.[2] Long-term topical steroid post PKP is considered as one of the most important risk factors for ICK.[3] In contrast to the belief that with the advent of lamellar keratoplasty, incidence of ICK might decrease,[4] our series had one patient with infection post DALK. Chua and Sandford-Smith[5] reported the first case of ICK in lamellar keratoplasty, which presented as a focal suture infiltrate that healed within 2 weeks. But in our patient it presented as a central infiltrate progressing to involve the entire stroma and took 14 weeks for complete healing. One of the cases in our series developed fungal ICK postcorneal tattooing procedure in a nonseeing eye, which has never been reported earlier. Delayed epithelial healing, prolonged inflammation, and toxicity associated with the “dye method” of corneal tattooing can be considered as a risk factor for the development of ICK.

Although topical steroids and anesthetics have been considered as risk factors for ICK in previous literature, association with glaucoma medications has not been reported.[6] Long-term use of antiglaucoma medications with preservatives and corneal edema secondary to frequent intraocular pressure fluctuations in these patients can result in an unhealthy corneal epithelium. This along with local immunosuppression secondary to topical steroids may give microorganisms easy access to the corneal stroma.

Along with the common presentation of anterior stromal infiltrate, two cases presented with intracorneal hypopyon. One patient developed intracorneal hypopyon within 4 weeks of presentation in his second corneal graft, whereas the other presented in the first week in his first corneal graft. While the former required therapeutic keratoplasty due to a prolonged clinical course, the latter healed within 2 weeks of presentation.

Apart from the most common etiological organism Streptococci, fungal species too have the ability to produce a biofilm, which aids in the formation of ICK.[7] Similarly, we found Candida albicans as the causative organism in one of our cases that responded well to medical treatment. Burkholderia cepacia was another rare organism (gram negative bacteria) isolated, that most often causes pneumonia in immunocompromised individuals and is known for its natural resistance to many first-line topical antibiotics.[8] Antibiotic resistance to fluoroquinolones was seen in most of our patients [Table 2]. This could be explained by their long-term use along with steroids post keratoplasty and the indolent nature of the organisms causing ICK.

Unlike the study by Sharma et al.,[9] which reported that more than 50% of cases required PKP for control of infection, our case series demonstrated good recovery with topical medications. Delayed healing was seen in one patient post DALK surgery, which could be due to various factors such as interface infection, poor bioavailability of drugs, and drug resistance. The other patient who required surgical intervention had a deep stromal infiltrate that compromised drug penetration and added poor glycemic control that worsened healing.


  Conclusion Top


To conclude, timely targeted therapy after performing a good microbiological workup is the key to faster recovery in ICK.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Stern GA. Infectious crystalline keratopathy. Int Ophthalmol Clin 1993;33:1-7.  Back to cited text no. 1
    
2.
Reiss GR, Campbell RJ, Bourne WM. Infectious crystalline keratopathy. Surv Ophthalmol 1986;31:69-72.  Back to cited text no. 2
    
3.
Hollander DA, Clay EL, Sidikaro Y. Infectious crystalline keratopathy associated with intravitreal and posterior sub-tenon triamcinolone acetonide injections. Br J Ophthalmol 2006;90:656.  Back to cited text no. 3
    
4.
Porter AJ, Lee GA, Jun AS. Infectious crystalline keratopathy. Surv Ophthalmol 2018;63:480-99.  Back to cited text no. 4
    
5.
Chua VW, Sandford-Smith JH. Infectious crystalline keratopathy after stitch removal in a lamellar corneal graft. Eye 2000;14:797-9.  Back to cited text no. 5
    
6.
Kintner JC, Grossniklaus HE, Lass JH, Jacobs G. Infectious crystalline keratopathy associated with topical anesthetic abuse. Cornea 1990;9:77-80.  Back to cited text no. 6
    
7.
Elder MJ, Matheson M, Stapleton F, Dart JK. Biofilm formation in infectious crystalline keratopathy due to Candida albicans. Cornea 1996;15:301-4.  Back to cited text no. 7
    
8.
Ornek K, Ozdemir M, Ergin A. Burkholderia cepacia keratitis with endophthalmitis. J Med Microbiol 2009;58:1517-8.  Back to cited text no. 8
    
9.
Sharma N, Vajpayee RB, Pushker N, Vajpayee M. Infectious crystalline keratopathy. CLAO J 2000;26:40-3.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2]



 

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