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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 543-545

Fungal orbital cellulitis presenting as a discharging fistula


Department of Ophthalmology, B.R.D. Medical College, Gorakhpur, Uttar Pradesh, India

Date of Submission31-Aug-2021
Date of Acceptance22-Nov-2021
Date of Web Publication13-Apr-2022

Correspondence Address:
Aditi Jhunjhunwala
Department of Ophthalmology, B.R.D. Medical College, Gorakhpur, Uttar Pradesh - 273 013
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_2257_21

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  Abstract 


We present a very rare case of fungal orbital cellulitis which presented to us as a pus-discharging fistula below the medial canthus. The patient presented with signs of orbital cellulitis along with a pus-discharging fistula below the medial canthus with positive mucopurulent discharge from the puncta and fistula on syringing. It started with swelling near the medial canthus. The patient responded well on broad-spectrum antibiotics but developed a necrotic patch on the hard palate. The Magnetic resonance imaging (MRI) showed intraconal and extraconal involvement. Diagnostic nasal endoscopy (DNE) was done and the tissue was sent for Potassium hydroxide (KOH) mounting, culture, and histopathology. After the KOH mount showed budding-like yeast cells, the patient was started on antifungals and discharged when the repeat culture report showed no growth. Therefore, a high level of suspicion should be maintained for a fungal cause even in the presence of the pus-discharging fistula.

Keywords: Candida, fistula, fungal, orbital cellulitis, pus


How to cite this article:
Yadav RY, Rai C, Jhunjhunwala A. Fungal orbital cellulitis presenting as a discharging fistula. Indian J Ophthalmol Case Rep 2022;2:543-5

How to cite this URL:
Yadav RY, Rai C, Jhunjhunwala A. Fungal orbital cellulitis presenting as a discharging fistula. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 18];2:543-5. Available from: https://www.ijoreports.in/text.asp?2022/2/2/543/342939



As we all are hiding behind masks in this time of COVID- 19 Virus (COVID-19), we present a very rare case of fungal orbital cellulitis hiding behind a pus-discharging fistula below the medial canthus, its clinical course, and its successful management. Due to the high mortality and morbidity associated with fungal infections in the eye and with rising cases of fungal orbital cellulitis during the second wave of COVID-19 in our country, it becomes very important to understand the various presentations and their management.


  Case Report Top


A 41-year-old male, a known diabetic, presented to us at our post-COVID-19 fungal Out Patient Clinic (OPD) on May 29 with complaints of pain and swelling over the left cheek and periorbital area, diminution of vision, and drooping of the left eyelid for 20 days, and pus discharge near the medial canthus for 1 week, which was sudden in onset and progressive. The patient had a fever and severe myalgia in mid-April for which he took over-the-counter drugs. He was never tested for COVID-19. Fifteen days after recovering from fever, he started having pain and swelling over the left cheek which progressed to the periorbital area along with the bulging of the eyeball, drooping of the eyelid, and diminution of vision in the next 10 days. The patient also developed a pus-discharging fistula below his medial canthus. The Computed Tomography (CT) scan was done elsewhere [Figure 1]b and [Figure 1]c and the patient was started on oral antibiotics, and then, referred to us [Figure 1]a.
Figure 1: (a) Patient at the time of presentation showing fistula with pus discharge after broad-spectrum antibiotics given elsewhere. (b) CT scan of the patient showing soft-tissue density involving the left ethmoid sinus. The lesion is causing the osteoerosion of the nasal bone causing enhanced thickening along with the left intraorbital extension. (c) There is a hyperdensity with fat stranding noted involving the intraconal compartment of the left orbit. The lesion is also seen encasing the optic nerve and extending up to the orbital apex. (d) KOH mount of nasal swab showing budding yeast-like cells

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On examination, the Glasgow Chroma Scale of the patient was 15/15, with no meningeal signs, and normal higher mental, motor, and sensory functions. On examining the left eye, there was complete ptosis, proptosis with congested and chemosed conjunctiva, hazy cornea, and normal-shaped and sluggish reacting pupil with extraocular movement restricted in all gazes. The patient only had a perception of light. A pus-discharging fistula was present just below the medial canthus. Syringing was done which showed mucopurulent discharge coming from the fistula as well as both puncta. The fundus was normal on the right side and hard exudates at and around the macula were present on the left. Examination of the right eye was normal. The patient was advised admission but he refused. He was started on broad-spectrum IV antibiotics (vancomycin, metrogyl, and meropenem) and topical antibiotics. A routine investigation was done and the Random Blood Sugar (RBS) of the patient was 324 mg/dL. After 1 week, the patient developed a white necrotic patch on the left side of the hard palate [Figure 2]a. Again, he was advised admission and he denied it. Ear, Nose and Throat (ENT) consultation was done and the patient was started on an oral antifungal along with an antibiotic. He came back with pus-discharging necrotic area over the hard palate and was admitted on July 8. The MRI [Figure 2]b and [Figure 2]c and all the routine investigations were done. At the time of admission, the RBS was 155 mg/dL. He was started on IV vancomycin, meropenem, metrogyl, and liposomal amphotericin B (5 mg/kg). The nasal swab was sent for KOH mount which showed budding-like yeast cells [Figure 1]d. DNE was done the next day. Necrotic tissue was seen on the inferior and middle turbinate; scrapping was done and sent for KOH and culture. Three retrobulbar amphotericin B injections on alternate days were given. On July 13, S. creatinine was 1.7 mg/dL, so IV liposomal amphotericin B was stopped and the patient was shifted to IV posaconazole. Functional Endoscopic Sinus Surgery (FESS) was done the next day. The maxillary, ethmoid, and sphenoid sinuses were cleared. The posterior wall of the maxillary sinus with the floor of orbit was found eroded and removed. The culture showed the growth of candida species [Figure 3]a and [Figure 3]b. The patient was continued on an antifungal (voriconazole) and antibiotic and discharged when the repeat culture report showed no growth. At the time of discharge, the patient had a visual acuity of 2/60. At 1-month post-op follow-up, the patient showed healing of the fistula site and improvement in ptosis and proptosis [Figure 3]c.
Figure 2: (a) Picture showing white necrotic lesion on the left side of the hard palate. For further characterization of the lesion, contrast-enhanced MRI was performed in our institute which shows (b) soft-tissue lesion with signal intensity alteration involving the left ethmoid sinus with extension into the preseptal region and causing enhancing thickness. The lesion also shows infiltration of the optic nerve. (c) The lesion is showing avid enhancement on post-contrast imaging

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Figure 3: (a) Culture cells showing yeast-like cells. (b) Culture showing white colonies characteristic of candida. (c) One month post-op

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  Discussion Top


Fungal spores are present everywhere which act as an opportunistic infection in immunocompromised individuals and are associated with a high mortality rate.[1] These are most commonly by fungus of order mucora le (rhizopus) which is angioinvasive[2],[3] and order Eurotiales (aspergillosis).[3] Orbital cellulitis due to candida is very rare. Not a lot of information is available currently about candidal orbital cellulitis but immunocompromised and antibiotic overuse are common risk factors along with COVID-19, which in itself is an immunocompromised state.[4] In 2009, Kwak et al.[5] reported a rare case of facial cellulitis caused by candida in a patient with uncontrolled diabetes whereas in 2013, Mukherjee et al.[6] reported a case of reverse masquerade syndrome where a fungal adnexal infection caused by candida was mimicking carcinoma in an Human Immunodeficiency virus (HIV)-positive patient. In 2015, Motukupally et al.,[7] and in 2017, Ranjith et al.,[8] each reported one case of candida orbital cellulitis as part of their broad study to determine the most common candida species in an eye infection. The Michigan hospital guideline suggests voriconazole and amphotericin B for the treatment of orbital candidiasis.[9] We searched the electronic database of PubMed and Google Scholar using keywords but did not find any reported case of candidal orbital cellulitis which presented with pus-discharging fistula.


  Conclusion Top


A high level of suspicion should be maintained for fungal causes even in the presence of the pus-discharging fistula showing mucopurulent discharge on syringing.

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.
Farooq AV, Patel RM, Lin AY, Setabutr P, Sartori J, Aakalu VK. Fungal orbital cellulitis: Presenting features, management and outcomes at a referral center. Orbit (Amsterdam, Netherlands) 2015;34:152-9.  Back to cited text no. 1
    
2.
Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human disease. Clin Microbiol Rev 2000;13:236-301.  Back to cited text no. 2
    
3.
Mukherjee B, Raichura ND, Alam MS. Fungal infections of the orbit. Indian J Ophthalmol 2016;64:337-45.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Song G, Liang G, Liu W. Fungal Co-infections associated with global COVID-19 pandemic: A clinical and diagnostic perspective from China. Mycopathologia 2020;185:599-606.  Back to cited text no. 4
    
5.
Kwak OS, Kang MI, Kim JB, Kim MW, Kim YK. A rare case of facial Candida albicans cellulitis in an uncontrolled diabetic patient. Mycoses 2009;52:379-81.  Back to cited text no. 5
    
6.
Mukherjee B, Chatterjee R, Biswas J. Reverse masquerade syndrome: Fungal adnexal infection mimicking carcinoma in a HIV-positive patient. Indian J Ophthalmol 2013;61:521-3.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Motukupally SR, Nanapur VR, Chathoth KN, Murthy SI, Pappuru RR, Mallick A, et al. Ocular infections caused by Candida species: Type of species, in vitro susceptibility and treatment outcome. Indian J Med Microbiol 2015;33:538-46.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Ranjith K, Sontam B, Sharma S, Joseph J, Chathoth KN, Sama KC, et al. Candida species from eye infections: Drug susceptibility, virulence factors, and molecular characterization. Invest Ophthalmol Vis Sci 2017;58:4201-9.  Back to cited text no. 8
    
9.
Available from: med.umich.edu/asp/pdf/adult_guidelines/Ocular_ADULT.  Back to cited text no. 9
    


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