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CASE REPORT
Ahead of print publication  

Klebsiella keratitis presenting as a ring infiltrate in an adolescent girl


1 Department of Ophthalmology, Government Medical College, Chandigarh, India
2 Department of Ophthalmology, Maharishi Markandeshwar Institute of Medical Sciences and Research, Ambala, Haryana, India
3 Department of Microbiology, Maharishi Markandeshwar Institute of Medical Sciences and Research, Ambala, Haryana, India

Date of Submission07-Aug-2022
Date of Acceptance29-Sep-2022
Date of Web Publication05-Jan-2023

Correspondence Address:
Richa Dhiman,
Department of Ophthalmology, Government Medical College, Sector-32, Chandigarh - 160 047
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/tjo.TJO-D-22-00101

  Abstract 


The purpose of the study was to report a unique case of Klebsiella keratitis presenting as a ring infiltrate in an adolescent girl. A 16-year-old girl presented with decreased vision in the right eye preceding an episode of fever with a rash associated with burning micturition. The patient was examined after taking appropriate consent. The slit-lamp examination revealed a ring-shaped corneal infiltrate with an epithelial defect in her right eye. Corneal scrapings were sent for microbiological evaluation which revealed Gram-negative rods and culture identified it as extended-spectrum beta-lactamase-producing Klebsiella pneumoniae colonies. The patient showed a good response to topical fortified amikacin and tobramycin. For her systemic complaints, the pediatrician did a thorough investigative workup out of which blood culture showed growth of K. pneumoniae. Hence, intravenous antibiotics were given based on the antibiogram report and the patient recovered. After 2 weeks, a paracentral infiltrate in her left eye was noted followed by anterior uveitis. The patient responded well to the topical course of steroids along with aminoglycosides. Four months later, she had a recurrence of anterior uveitis in the right eye preceded by fever. Blood investigations were negative. Hence, a diagnosis of recurrent uveitis secondary to endogenous infection was made and the patient was successfully treated with a short course of topical steroids. The patient is on follow-up for the past 6 months and maintaining the best-corrected visual acuity of 20/20 OU with normal intraocular pressure and quiet anterior chamber (AC). This is the first clinical report describing a ring infiltrate in endogenous Klebsiella keratitis and emphasizes thorough workup for prompt treatment.

Keywords: Amikacin, endogenous infection, extended-spectrum beta-lactamase-producing, Klebsiella keratitis, ring infiltrate, tobramycin



How to cite this URL:
Dhiman R, Sharma N, Chauhan J. Klebsiella keratitis presenting as a ring infiltrate in an adolescent girl. Taiwan J Ophthalmol [Epub ahead of print] [cited 2023 Jan 28]. Available from: https://www.e-tjo.org/preprintarticle.asp?id=367069




  Introduction Top


Klebsiella is a Gram-negative, encapsulated, nonmotile rod or coccobacilli, and a facultative anaerobe. It belongs to the Enterobacteriaceae family and consists of seven species among which Klebsiella pneumoniae and Klebsiella oxytoca are the two most common pathogens. It is a well-known opportunistic pathogen, responsible for causing nosocomial infections such as urinary tract infections, gastrointestinal infections, and pneumonia. Further, the emergence of antibiotic resistance among Klebsiella species is on a rise. Klebsiella and Escherichia coli are found to be the most common pathogenic microorganisms developing resistance to broad-spectrum beta-lactam antibiotics through extended-spectrum beta-lactamase (ESBL).[1]

Among ophthalmological isolates, the percentage of ESBL-producing Enterobacteriaceae is still rare. It usually affects immunocompromised or debilitated patients or the corneas with a preexisting pathological condition. The most serious manifestation is the catastrophic endogenous endophthalmitis. There have also been reports of Klebsiella keratitis secondary to Klebsiella conjunctivitis, refractive surgery, cataract surgery, and contact lens use.[2],[3],[4],[5],[6],[7] The clinical presentation described in the literature is usually a central or paracentral infiltrate with or without hypopyon. Here, for the first time, we are reporting a case of ESBL-producing K. pneumoniae keratitis in an adolescent girl presenting as a ring infiltrate with no history of trauma or ocular surgery who was successfully treated with fortified tobramycin and amikacin.


  Case Report Top


A 16-year-old girl presented with pain, redness, watering, and blurred vision in her right eye for 4 days. There was no history of ocular trauma, surgery, or contact lens use. She had a history of fever 15 days before this episode associated with burning micturition, papulomacular rash over the lower abdomen and groin, and vague joint pains for which she took symptomatic treatment elsewhere (no previous records available). Currently, the patient was using topical antifungals and antibiotics in the right eye for the past 2 days.

On examination, her best-corrected visual acuity (BCVA) was hand movements close to the face in the right eye and 20/20 in the left eye. A slit-lamp examination of the right eye showed intense circumcorneal congestion and a ring-shaped corneal infiltrate with an epithelial defect measuring around 6 mm × 6 mm. The anterior chamber was quiet with no hypopyon and the lens was phakic [Figure 1]a. Fundus examination revealed hazy media due to corneal involvement with a disc hazily seen and a retina attached. The left eye appeared normal. Corneal sensations were intact in both eyes. Corneal scrapings were obtained and sent for microbiological analysis. Gram stain of corneal scrapings revealed Gram-negative rods and culture yielded heavy growth of K. pneumoniae [Figure 2] later identified as ESBL producers by VITEK (automated method for antibiotic sensitivity testing). Antibiotic susceptibility testing determined by agar disk diffusion (Kirby–Bauer method) showed that the isolates were sensitive to amikacin, gentamicin, tobramycin, tigecycline, and trimethoprim-sulfamethoxazole. The patient was started on fortified amikacin (2.5%) and fortified tobramycin (1.3%) hourly with cycloplegic drops in the right eye.
Figure 1: Slit-lamp image of (a) ring-shaped infiltrate in the right eye on presentation, (b) resolving to infiltrate at 1 week, (c) scarring infiltrate with mild corneal edema at 2 weeks, (d) completely scarred infiltrate at 1 month, (e) recurrence of anterior uveitis with inflammatory hypopyon at 4 months, (f) resolution of uveitis after steroid treatment, (g) paracentral stromal infiltrate of the left eye at 1 week, (h) granulomatous uveitis at 2 weeks, and (i) resolution of uveitis on steroid therapy at 1 month

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Figure 2: (a) Gram stain of the right eye showing Gram-negative bacilli, (b) white mucoid colonies of Klebsiella pneumoniae on blood agar, and (c) lactose-fermenting colonies of Klebsiella pneumoniae on MacConkey agar

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In view of the recent history of fever with rash and joint pains, pediatric consultation was sought to rule out a systemic cause for corneal ulcer which could be autoimmune or infective. Her blood investigations included a complete blood count with an erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP), random blood sugar along with blood culture, Mantoux test, and chest X-ray. HLA B27, rheumatoid factor, and antinuclear antibody were done to rule out any autoimmune cause. Ultrasound abdomen and pelvis were done to look for any septic foci which came out to be normal. Investigations revealed leukocytosis, raised ESR (102 mm), CRP (26.1 mg/dL), and blood culture with confluent growth of K. pneumonia after 24 h of incubation. Rest investigations were negative. Hence, a probability of endogenous Klebsiella keratitis in the right eye was considered, although the exact source of the primary infection could not be detected. The patient was admitted and started on intravenous antibiotics gentamicin and piperacillin with tazobactam based on the blood culture sensitivity report.

After 1 week of treatment, the patient's systemic symptoms resolved. Ocular examination showed a resolving corneal infiltrate with no epithelial defect and BCVA improved to 20/200 in the right eye [Figure 1]b. The patient was discharged and was kept on follow-up. One week later, she reported to us with redness and watering in her left eye. On examination, her BCVA was 20/40 in both eyes. Slit-lamp examination showed a scarring ring infiltrate in the right eye with mild corneal edema [Figure 1]c. There was a paracentral stromal infiltrate measuring 2 mm × 2 mm with no epithelial defect, quiet AC, and no hypopyon in the left eye [Figure 1]g. Corneal scraping was deferred in view of the small infiltrate size (≤2 mm) and no epithelial defect. The patient was started on the same fortified antibiotics considering the likelihood of Klebsiella keratitis in this eye as well.

One week later, the patient came for a follow-up which revealed scarred infiltrate in the right eye [Figure 1]d and diffuse stromal edema, granulomatous keratic precipitates (KPs), and 3+ cells in AC in the left eye [Figure 1]h. The patient was investigated thoroughly to rule out uveitis due to any other pathology (tuberculosis, sarcoidosis, and syphilis) which came out to be negative. She was started on topical steroids six times a day along with antibiotics and cycloplegic drops keeping a possibility of immune-mediated pathology in the left eye. A diagnosis of healed endogenous Klebsiella keratitis in the right eye with immune-mediated keratouveitis in the left eye was made.

On the 2-week follow-up, her BCVA improved to 20/20 in both eyes with scarred infiltrate and quiet AC bilaterally [Figure 1]d and [Figure 1]i. Her fundus examination was normal on all the visits [Figure 3].
Figure 3: Fundus photograph at 1-week follow-up with normal optical coherence tomography scans of the right eye (a and c) and left eye (b and d)

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The patient was doing well when 4 months later; she again had a similar episode of uveitis in the right eye [Figure 1]e. This was preceded by a fever for 1 day which resolved on symptomatic treatment. There were no other systemic features. Blood investigations were negative. Hence, a possibility of recurrent uveitis secondary to endogenous infection was kept and was successfully treated with a short course of topical steroids [Figure 1]f. The patient is on follow-up for the past 6 months and maintains a BCVA of 20/20 OU with normal intraocular pressure and quiet AC.


  Discussion Top


Klebsiella is a rare ocular pathogen responsible for causing devastating ocular infections in immunocompromised and debilitated individuals. It is reported that females in their reproductive age group and males above 50 years are more prone to Klebsiella infections, especially ESBL producing.[8] Klebsiella strains possess several virulence factors which provide them resistance against the routinely prescribed antibiotics among which Gram-negative lipopolysaccharide, mucoid phenotype, and ability to form the biofilm play a predominant role. Further, the ESBL-producing isolates have resistance to widespread antibiotics creating major therapeutic difficulties. A study by Jayahar Bharathi et al. on 135 Gram-negative clinical ophthalmological isolates reported a 7% ESBL-producer rate.[9]

There are several interesting points with respect to this case. It occurred in a young healthy female with no predisposing factor like trauma or ocular surgery accounting for the Klebsiella infection. However, she gave a history of fever, rash, and burning micturition 2 weeks before ocular complaints with blood culture positive for Klebsiella. This highlights the endogenous source of infection, although we could not localize the primary source of infection. The septic metastasis to the eye can occur from the abscess of the liver, kidney, or genitourinary tract as per literature with a liver abscess as the major endogenous source for K. pneumonia endogenous endophthalmitis.[10]

Endogenous Klebsiella endophthalmitis has been reported in the literature either in immunocompromised or hospitalized patients but, endogenous Klebsiella keratitis has never been described earlier. This might be explained by the early diagnosis and prompt treatment which enabled us to prevent the posterior segment involvement.

Second, a typical ring infiltrate is a classical feature of Acanthamoeba keratitis. It is presumably formed as a result of an immunologic response like antigen–antibody complexes, complement, and polymorphonuclear leukocytes. It can also be seen in patients with Pseudomonas, Serratia, herpetic, or fungal keratitis but has never been described as a corneal infection caused by Klebsiella.

Third, although the Klebsiella isolated from blood as well as corneal scraping were both ESBL producers yet the antibiotic sensitivity profile was slightly different. A higher resistant pattern was noted in the corneal sample with resistance to carbapenem antibiotics in comparison to blood culture. An earlier case report has focused on the use of imipenem/cilastatin for ESBL-producing K. pneumoniae.[4] In our case, we got a good outcome with the use of topical amikacin and tobramycin and intravenous gentamicin with piperacillin and tazobactam.

Another interesting feature was the development of immune-mediated uveitis secondary to healed keratitis in the left eye followed by the right eye after a gap of 4 months which was preceded by fever for 1 day. This could be explained by the fact that initially organism entered the eye through the bloodstream and now the ocular tissue is already sensitized to that antigen. Hence, due to the formation of memory cells, a nonspecific focal reaction might occur in the eyes whenever the inflammatory cells reappear in the bloodstream.[11] This is usually seen in HLA-B27-positive patients or immune-mediated pathologies which was negative in this case.

To our knowledge, this is the first clinical report describing a ring infiltrate in Klebsiella infection in a healthy young girl successfully treated with aminoglycosides. This case highlights the possibility of Klebsiella infection in a ring infiltrate and emphasizes the thorough microbiological and systemic workup to establish the diagnosis and prompt treatment.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the parents have given their consent for the child's images and other clinical information to be reported in the journal. The parents understand that their name 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

The authors declare that there are no conflicts of interests of this paper.



 
  References Top

1.
Abbott S. Klebsiella, Enterobacter, Citrobacter, and Serratia. In: Murray PR, Baron EJ, Jorgensen JH, Landry ML, Pfaller MA. Manual of Clinical Microbiology. 7th ed. Washington DC: ASM Press; 1999. p. 475-82.  Back to cited text no. 1
    
2.
Aung T, Chan TK. Nosocomial Klebsiella pneumoniae conjunctivitis resulting in infectious keratitis and bilateral corneal perforation. Cornea 1998;17:558-61.  Back to cited text no. 2
    
3.
Zarei-Ghanavati S, Sedaghat MR, Ghavami-Shahri A. Acute Klebsiella pneumoniae interface keratitis after deep anterior lamellar keratoplasty. Jpn J Ophthalmol 2011;55:74-6.  Back to cited text no. 3
    
4.
Egrilmez S, Palamar M, Sipahi OR, Yagci A. Extended spectrum beta-lactamase producing Klebsiella pneumoniae-related keratitis. J Chemother 2013;25:123-5.  Back to cited text no. 4
    
5.
Bajracharya L, Sharma B, Gurung R. A case of acute postoperative keratitis after deep anterior lamellar keratoplasty by multidrug resistant Klebsiella. Indian J Ophthalmol 2015;63:344-6.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Cumurcu T, Firat P, Ozsoy E, Cavdar M, Yakupogullari Y. Contact-lens-related corneal ulcer caused by Klebsiella pneumoniae. Clinics (Sao Paulo) 2011;66:1509-10.  Back to cited text no. 6
    
7.
Basak S, Basak SK, Saha S. Acute interface infectious keratitis with multidrug resistant Klebsiella and Escherichia coli following deep anterior lamellar keratoplasty. Indian J Ophthalmol 2020;68:1678-80.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Alipourfard I, Nili NY. Antibiogram of extended spectrum beta-lactamase (ESBL) producing Escherichia coli and Klebsiella pneumoniae isolated from hospital samples. Bangladesh J Med Microbiol 2011;4:32-6.  Back to cited text no. 8
    
9.
Jayahar Bharathi M, Ramakrishnan R, Ramesh S, Murugan N. Extended-spectrum beta-lactamase-mediated resistance among bacterial isolates recovered from ocular infections. Ophthalmic Res 2012;47:52-6.  Back to cited text no. 9
    
10.
Yin W, Zhou H, Li C. Endogenous Klebsiella pneumoniae endophthalmitis. Am J Emerg Med 2014;32:5.e3-5.  Back to cited text no. 10
    
11.
Sihota R, Tandon R. Diseases of uveal tract: Aetiopathogenesis of uveitis. In: Parson's Diseases of the Eye. 22nd ed. Delhi: Elsevier; 2015. p. 233-4.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

 
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