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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 35  |  Issue : 1  |  Page : 66-70

Isolation of unusual bacteria in canaliculitis: A series of four cases


1 Ophthalmic Plastic Surgery Services, LV Prasad Eye Institute, MTC Campus, Bhubaneswar, India
2 Ocular Microbiology Services, LV Prasad Eye Institute, MTC Campus, Bhubaneswar, India

Date of Submission20-Sep-2019
Date of Acceptance06-Dec-2020
Date of Web Publication09-Sep-2021

Correspondence Address:
Dr. Suryasnata Rath
Ophthalmic Plastic Surgery Services, LV Prasad Eye Institute, MTC Campus, Bhubaneswar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1319-4534.325778

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  Abstract 


With increased availability of sophisticated microbiological techniques for isolation, growth and identification of micro-organisms the spectrum of organisms is rapidly. Herein we report four cases of canaliculitis with unusual organisms and highlight their clinical significance. To the best of our knowledge, there are no reports of isolation of Brucella melitensis and Leuconostoc species reported in English literature; and only one report of isolation of Myroides species from canaliculitis exists. Sphingomonas paucimobilis, is an uncommon isolate in canaliculitis. Extremes of age and occupational exposure may be possible risk factors for infection with uncommon organisms. Clinical features at presentation do not vary greatly with uncommon or multi drug resistant organisms' hence sampling and microbiological assessment is warranted. The benefit of curettage in canaliculitis is manifold. Unusual organisms and opportunistic pathogens can be multi-drug resistant and determination of antibiotic susceptibility is important to initiate targeted therapy to ensure disease cure and prevent recurrences.

Keywords: Canaliculitis, pediatric canaliculitis, concretions, Myroides, Leuconostoc, Sphingomona


How to cite this article:
Vempuluru VS, Mitra S, Tripathy D, Mohapatra S, Rath S. Isolation of unusual bacteria in canaliculitis: A series of four cases. Saudi J Ophthalmol 2021;35:66-70

How to cite this URL:
Vempuluru VS, Mitra S, Tripathy D, Mohapatra S, Rath S. Isolation of unusual bacteria in canaliculitis: A series of four cases. Saudi J Ophthalmol [serial online] 2021 [cited 2021 Dec 6];35:66-70. Available from: http://www.sjo.org/text.asp?2021/35/1/67/325778




  Introduction Top


Canaliculitis accounts for 2%–4% of lacrimal diseases and the usual causative organisms include gram-positive organisms such as Actinomycetes, Staphylococcus, and Streptococcus species.[1],[2],[3],[4] We describe four cases of canaliculitis with uncommon microbiological isolates and elaborate on their clinical relevance in terms of susceptibility to antimicrobial agents. There are no reports of isolation of  Brucella More Details melitensis and Leuconostoc reported in the English literature; and we also report the first case of isolation of Myroides species from childhood canaliculitis. Sphingomonas paucimobilis has been an uncommon isolate in primary canaliculitis.[3] Extremes of age and occupational exposure may be possible risk factors for infection with uncommon organisms. As there is significant overlap in the clinical presentation of canaliculitis caused by uncommon organisms sampling and microbiological workup are warranted. The benefit of curettage in canaliculitis is manifold in reducing the organismal load, increasing the potential space of antibiotic reservoir, and allowing microbiological assessment. This case series highlights that unusual isolates from canaliculitis can be opportunistic pathogens showing multi-drug resistance and determination of antibiotic susceptibility helps initiate targeted therapy. Patient consent has been obtained for publishing clinical photographs. The study adhered to the declaration of Helsinki.


  Case Reports Top


Case 1

A 49-year-old male came to us with complaints of watering in his right eye of 3 months duration. On examination, there was erythematous swelling of the medial third of the lower eyelid and an inflamed pouting punctum [Figure 1]a. Debris expressed from the lower canaliculus on the application of pressure with cotton-tipped applicator was sent for microbiological examination. Gram's staining of the canalicular contents revealed numerous gram-positive branching filamentous organisms and groups of gram-positive cocci in pairs and short chains among a large number of gram-negative coccobacilli [Figure 1]b. On aerobic culture, confluent pin-point alpha-hemolytic colonies and confluent translucent gray colonies grew on blood and chocolate agar at the site of inoculation [Figure 1]c. Anaerobic subculture from thioglycolate broth grew discrete whitish colonies with a “molar-tooth” appearance. The organisms were isolated as Streptococcus species, Sphingomonas paucimobilis [Figure 1]d, and Actinomycetes species. Streptococcus and Sphingomonas were sensitive to all antibiotics [Table 1], but Actinomycetes species showed multidrug resistance.
Figure 1: (a) Clinical photograph showing eyelid edema and inflamed canalicular mucosa pouting through the punctum (arrow); (b) Smear showing gram-positive branching filamentous organisms, gram-positive cocci in short chains and pairs and numerous gram-negative bacilli (arrows) throughout (Gram stain, ×100); (c) Confluent alpha hemolytic pin point colonies at the site of inoculation on blood agar; (d) Identification of the organism as  Sphingomonas paucimobilis Scientific Name Search y Vitek 2 Compact identification system

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Table 1: Clinical and microbiological profile of unusual organisms isolated in our series of patients with canaliculitis

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Case 2

A 9-year-old child was brought by his parents with complaints of discharge from his right eye for 3 months. Careful examination of his right eye revealed upper canalicular edema, pouting punctum, and expression of purulent material on application of pressure over the upper canaliculus. Although rare in children, classical features hinted at the diagnosis of canaliculitis which was confirmed by expression of copious pus and multiple concretions on curettage. Gram's staining of smears from the concretion revealed plenty of polymorphonucleocytes with few gram-negative bacilli per oil immersion field [Figure 2]a. On aerobic culture, growth of discrete, pale yellow colored colonies [Figure 2]b and [Figure 2]c at the site of inoculation and the organism was identified as Myroides species by Vitek 2 Compact identification system [Figure 2]d which was multidrug resistant [Table 1] but sensitive to chloramphenicol.
Figure 2: (a) Few gram-negative bacilli (arrows) in a background of plenty polymorphs (Gram stain, ×100); (b and c) Discrete, small, pale yellow colored colonies at the site of inoculated concretions on blood and chocolate agar; (d) Identification of the organism as Myroides species by Vitek 2 compact identification system

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Case 3

A 58-year-old male, a farmer by occupation presented with persistent watering and intermittent discharge for 1 year. He had had multiple consultations earlier and was treated with topical antibiotics. Ocular examination showed edema of the right lower eyelid localized to the medial aspect with a pouting punctum. Canalicular curettage revealed the presence of pus and concretions which were sent for microbiological evaluation. Gram's stain of the smear showed Gram-positive branching filamentous organisms and Gram-positive cocci in clusters.

Nonhemolytic nonpigmented small gray colonies [Figure 3] grew on blood agar. These were identified as Brucella melitensis. Staphylococcus epidermidis and Actinomycetes were also isolated from culture. The organisms were sensitive to chloramphenicol [Table 1].
Figure 3: Nonhemolytic non-pigmented small gray colonies of Brucella melitensis on blood agar

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Case 4

An 80-year-old male presented with watering and discharge from his right eye for 1 month. On examination, mild lower eyelid edema and matting of lashes with the discharge were noted. There was mild edema at the site of the punctum [Figure 4]. A diagnosis of canaliculitis was made and on curettage, debris was expressed from the lower canaliculus. No pus or concretions were present. Gram-positive cocci were seen in pairs and chains admixed with polymorphonucleocytes on the smear and culture on blood agar revealed small smooth round gray colonies which were identified as Leuconostoc species with a favorable sensitivity profile [Table 1].
Figure 4: Subtle lower eyelid edema and discharge in the patient with Leuconostoc infection

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Management

None of our patients were immunocompromised and all were in good systemic health. Among possible factors predisposing for infection were occupational exposure to cattle in case 3 and old age in case 4. All the patients underwent canalicular curettage and treated with topical chloramphenicol. Microbiological sampling, culture, and interpretation were in accordance with institute laboratory norms. Vitek 2 Compact identification system (Biomerieux, NC, l'Etiole, France) was used for identification of microorganisms in the cases 1–3 and Leuconostoc in case 4 was identified with biochemical tests. Kirby Bauer disc diffusion method was used to determine antibiotic susceptibility. All the organisms were susceptible to chloramphenicol and Myroides species was found to be multi-drug resistant. All the patients were free of symptoms on follow-up visits at a median follow-up of 6 weeks.


  Discussion Top


Canaliculitis accounts for about 2% of lacrimal disorders and misdiagnosis of the condition ranges from 33% to 100%.[1],[2],[3],[4],[5],[6] The most common clinical symptoms include tearing, discharge, swelling and less often pain, or none at all.[1],[2],[3],[4],[5] Clinical signs which point at the diagnosis of canaliculitis include canalicular edema, pouting punctum, and expression of discharge on concretions by pressure over the canaliculus.[2],[3]

Canaliculitis has a slight female preponderance with the lower canaliculus being more frequently involved.[1] The female preponderance is presumed to be due to a combination of hormonal and lifestyle factors such as the use of cosmetics.[2],[3] All the four patients in our series were male. While canaliculitis can affects individuals at any age the mean age at diagnosis lies in the sixth decade.[1],[2] Childhood canaliculitis is rare with few reports in English literature.[7] Park et al. reported canaliculitis with isolation of Actinomycetes in a 5-year-old child on treatment with oral steroids for idiopathic thrombocytopenic purpura but there is not enough evidence implicating immunosuppression as a risk factor for canaliculitis.[7] The minimum age reported in two large series reported by Kaliki et al. and Gogandy et al. were 8 and 10 years, respectively.[2],[3]

There appears to be shift in the microbiological spectrum of organisms in primary canaliculitis with recent large series reported by Kaliki et al. and Gogandy et al. showing predominance of Streptococci and Staphylococci species.[2],[3] Gram-negative bacilli such as pseudomonas are known to be more common in punctal plug and canalicular stent-related infections.[4]

Sphingomonas paucimobilis as the name implies is a slowly motile gram-negative bacillus that has been isolated infrequently in primary canaliculitis.[4] Systemic and ocular infections with opportunistic pathogens such as Leuconostoc species and Sphingomonas paucimobilis are known to occur in the setting of immunosuppression.[8],[9],[10] Interestingly, we did not find a history of use of punctal plugs or canalicular stents or immunosuppression in the case 1 with Sphingomonas infection.

Myroides, isolated in childhood canaliculitis in this series, is not a part of human flora and inhabit water or soil.[11] Ali et al. reported an isolated case of Myroides associated canaliculitis showing resistance to chloramphenicol, cephalosporins, and penicillin.[11] Myroides isolated in our patient was resistant to fluoroquinolones, ceftazidime, imipenem, and penicillins.

Ocular  Brucellosis More Details has been reported in chronic systemic brucellosis, in the form of kerato-conjunctivitis episcleritis, uveitis, dacryoadenitis, retinal vasculitis, and optic neuritis.[12] Postulated mechanisms include direct invasion of ocular structures by the pathogen or development of immunological reaction to the bacterium.[12] In either case, the systemic infection is the source and the organism is not known to colonize ocular surface or skin.[12] We herein, report the first case of isolation of B. melitensis in canaliculitis. Our patient (Case 3) was a farmer by occupation and exposure to cattle may have been an antecedent factor. No systemic evidence of brucellosis was present.

Leuconostoc is a gram-positive coccus that is an opportunistic pathogen and is known to cause systemic infections such as septicemia and catheter-related infections in immunosuppressed neutropenic individuals.[9],[10] Apart from our patient being 80 years old, no systemic predisposing factors were noted. Reports of ocular involvement by Leuconostoc are limited to two case reports of endophthalmitis one following uneventful cataract surgery and another following intravitreal ranibizumab injection.[9],[10] Both the strains were resistant to resistant to vancomycin and despite early intervention in both cases, one patient had final vision of hand movements and other patient's eye deteriorated to phthisis.[9],[10] Leuconostoc thus can have a dreadful outcome following intraocular surgery or infection.[9],[10] We herein report the first case of canaliculitis with isolation of Leuconostoc species and emphasize the importance of complete cure and reviewing microbiological culture in canaliculitis, more so before planning any intraocular surgery.

Concretions that were earlier thought to be specific to Actinomycetes have also been reported to occur with, but not limited to Staphylococcus, Streptococcus, Hemophilus, Pseudomonas, Sphingomonas, and Gemella.[3] Infection from concretions. Ali et al. also reported the presence of concretions. Myroides and Brucella in our patients showed the presence of in canaliculitis with Myroides isolation [Table 2].[6]
Table 2: Epidemiology and clinical relevance of unusual organisms isolated in our series of patients with canaliculitis

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Management of canaliculitis has been multi-faceted with reasonable success rates reported for each of the procedures described in different case series.[1],[2],[3],[4],[5] Large series show curettage is effective in primary canaliculitis and prevents further recurrences.[2],[3] The advantages of curettage include decrease in organismal load, improved penetration of antibiotics, and the availability of specimens for microbiological evaluation to identify and determine antibiotic susceptibility. Punctal dilatation and expression as described by Kaliki et al. is less invasive procedure but had higher (40%) episodes of recurrent canaliculitis.[2] Conservative management by irrigation with antibiotics, described by Mohan et al. was reported to be effective.[13] Thus, conservative and/or punctal dilation and expression may be useful as the first line of therapy in most primary canaliculitis, canalicular curettage is the preferred management modality for recurrent disease.

Culture positivity in canaliculitis has improved over time with recent large case series on canaliculitis showing culture positivity range from 78% to 91%. Freedman et al. reported no organisms or non-specific organisms in 30% of the canaliculitis.[1],[2],[3] This is likely to be a consequence of advances in microbiological techniques over time.

In summary, this short case series documents for the first time isolation of Brucella melitensis and Leuconostoc in primary canaliculitis. We also report the first case of Myroides species from childhood canaliculitis. Sphingomonas paucimobilis remain an uncommon isolate in primary canaliculitis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patients have given their consent for 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.
Freedman JR, Markert MS, Cohen AJ. Primary and secondary lacrimal canaliculitis: A review of literature. Surv Ophthalmol 2011;56:336-47.  Back to cited text no. 1
    
2.
Kaliki S, Ali MJ, Honavar SG, Chandrasekhar G, Naik MN. Primary canaliculitis: Clinical features, microbiological profile, and management outcome. Ophthalmic Plast Reconstr Surg 2012;28:355-60.  Back to cited text no. 2
    
3.
Gogandy M, Al-Sheikh O, Chaudhry I. Clinical features and bacteriology of lacrimal canaliculitis in patients presenting to a tertiary eye care center in the Middle East. Saudi J Ophthalmol 2014;28:31-5.  Back to cited text no. 3
    
4.
Huang YY, Yu WK, Tsai CC, Kao SC, Kau HC, Liu CJ. Clinical features, microbiological profiles and treatment outcome of lacrimal plug-related canaliculitis compared with those of primary canaliculitis. Br J Ophthalmol 2016;100:1285-9.  Back to cited text no. 4
    
5.
Feroze KB, Patel BC. Canaliculitis. In: StatPearls. Treasure Island (FL): StatPearls Publishing; 2018. https://pubmed.ncbi.nlm.nih.gov/28722951/ [Last updated on 2019 Jan 13].  Back to cited text no. 5
    
6.
Singh M, Gautam N, Agarwal A, Kaur M. Primary lacrimal canaliculitis – A clinical entity often misdiagnosed. J Curr Ophthalmol 2018;30:87-90.  Back to cited text no. 6
    
7.
Park A, Morgenstern KE, Kahwash SB, Foster JA. Pediatric canaliculitis and stone formation. Ophthalmic Plast Reconstr Surg 2004;20:243-6.  Back to cited text no. 7
    
8.
Roca M, García A, Peñas-Pardo L, Bosch-Aparicio N, Agustí J. Sphingomonas paucimobilis keratitis in a patient with neurotrophic keratopathy and severe neurosensory hypoacusis: Treatment with penetrating keratoplasty and amniotic membrane grafting. Oman J Ophthalmol 2018;11:291-3.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Kumudhan D, Mars S. Leuconostoc mesenteroids as a cause of post-operative endophthalmitis – A case report. Eye (Lond) 2004;18:1023-4.  Back to cited text no. 9
    
10.
Damasceno NP, Horowitz SA, Damasceno EF. Leuconostoc as a cause of endophthalmitis post-intravitreal injection of ranibizumab. Ocul Immunol Inflamm 2016;24:118-9.  Back to cited text no. 10
    
11.
Ali MJ, Joseph J, Sharma S, Naik MN. Canaliculitis with isolation of Myroides species. Ophthalmic Plast Reconstr Surg 2017;33:S24-5.  Back to cited text no. 11
    
12.
Lashay AR, Manaviat MR, Azimi R, Riazi M. Ocular complications of brucellosis. Bina J Ophthalm 2003;4:345-8.  Back to cited text no. 12
    
13.
Mohan ER, Kabra S, Udhay P, Madhavan HN. Intracanalicular antibiotics may obviate the need for surgical management of chronic Suppurative canaliculitis. Indian J Ophthalmol 2008;56:338-40.  Back to cited text no. 13
[PUBMED]  [Full text]  


    Figures

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