Late onset endophthalmitis with rare fungus Exophiala dermatitidis

Shreya J Shah; Chinmay Nakhwa; Shraddha Shah; Madhu Rai

doi:10.4103/ijo.IJO_2280_21

CASE REPORT

Year : 2022 | Volume : 2 | Issue : 2 | Page : 480-482

Late onset endophthalmitis with rare fungus Exophiala dermatitidis

Shreya J Shah¹, Chinmay Nakhwa², Shraddha Shah³, Madhu Rai⁴
¹ Phaco-Refractive Services, Rushabh Eye Hospital and Laser Centre, Mumbai, Maharashtra, India
² Vitreo-Retina Services, Rushabh Eye Hospital and Laser Centre, Mumbai, Maharashtra, India
³ Comprehensive Ophthalmology Services, Rushabh Eye Hospital and Laser Centre, Mumbai, Maharashtra, India
⁴ P.D. Hinduja Hospital and Medical Research Centre, Mumbai, Maharashtra, India

Date of Submission	01-Sep-2021
Date of Acceptance	17-Dec-2021
Date of Web Publication	13-Apr-2022

Correspondence Address:
Shreya J Shah
Rushabh Eye Hospital and Laser Centre, Mumbai - 400 071, Maharashtra
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_2280_21

Abstract

The authors report a case of a 64-year-old diabetic male patient with late-onset fulminant fungal endophthalmitis occurring 6 weeks following complicated cataract surgery. A pigmented, dimorphic fungus known as Exophiala dermatitidis was isolated from aqueous and vitreous samples of the patient. The patient underwent pars plana vitrectomy, followed by daily injections of intravitreal voriconazole, and eventually had marked improvement in the best-corrected visual acuity to 6/9. So far, eight cases of Exophiala endophthalmitis have been reported in the literature, of which six have had a poor visual outcome. This case report demonstrates a successful outcome in a case of E. dermatitidis endophthalmitis, tackled with an aggressive medical and surgical approach.

Keywords: Dematiaceous, Exophiala dermatitidis, fungal endophthalmitis, Wangiella

How to cite this article:
Shah SJ, Nakhwa C, Shah S, Rai M. Late onset endophthalmitis with rare fungus Exophiala dermatitidis. Indian J Ophthalmol Case Rep 2022;2:480-2

How to cite this URL:
Shah SJ, Nakhwa C, Shah S, Rai M. Late onset endophthalmitis with rare fungus Exophiala dermatitidis. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2023 Jun 9];2:480-2. Available from: https://www.ijoreports.in/text.asp?2022/2/2/480/342942

Late-onset endophthalmitis after cataract surgery is less common than early-onset endophthalmitis. The main etiologic agents identified in postoperative endophthalmitis are bacteria such as Staphylococcus aureus, coagulase-negative Staphylococcus, Propionibacterium acnes, and some filamentous fungi.^[1] Fungal endophthalmitis is an uncommon entity but a visually debilitating infection affecting tropical and subtropical countries such as India. It is most commonly due to hyaline fungal species; however, dematiaceous fungi are also associated with new emerging cases. Authors here report a case of a late-onset, exogenous, fulminant endophthalmitis after cataract surgery due to a rare fungus Exophiala dermatitidis, with a successful treatment outcome.

Case Report

A 64-year-old male patient presented with a 1-day history of sudden onset of pain, redness, and diminution of vision in his left eye (OS) 6 weeks after cataract surgery. The patient had undergone phacoemulsification with intraocular lens (IOL) implantation in the left eye. A central posterior capsular rent occurred during surgery that was managed by anterior vitrectomy and subsequent placement of a hydrophobic acrylic IOL in the sulcus. The patient had underlying type II diabetes mellitus since 10 years, which was well controlled with oral hypoglycemic drugs. The presenting best-corrected visual acuity (BCVA) was hand movements in the OS and 6/6 in the right eye (OD). Intraocular pressure was 14 mmHg in OD and 22 mmHg in OS. Slit-lamp examination of the OS revealed findings of circumcorneal congestion, corneal edema, significant anterior chamber reaction with a dense fluffy fibrinous exudate extending up to the pupil [Figure 1]a. The posterior chamber IOL was in situ. The posterior segment view of the OS was hazy with the presence of dense vitritis, confirmed on B-scan ultrasonography [Figure 3]. The anterior and posterior segment findings of the OD were unremarkable. The patient tested negative for COVID-19 antigen confirmed by real-time-polymerase chain reaction (RT-PCR). The patient was started on topical treatment of moxifloxacin 0.5% (Vigamox, Alcon, India) eye drops hourly, homatropine 2% (Homide, Warren, India) eye drops twice a day, prednisolone acetate 1% (Predforte, Allergan, India) eye drops hourly, brimonidine tartrate 0.2%/timolol maleate 0.5% (Combigan, Allergan, India) twice a day. An anterior chamber tap of 0.1 cm³ was done, and the patient was empirically given intravitreal injection of vancomycin (1 mg/0.1 mL) and ceftazidime (2.25 mg/0.1 mL). Gram's stain and 10% potassium hydroxide mount of aqueous tap showed the presence of fungal hyphae, which was further subjected to lactophenol blue staining [Figure 2]a. The presence of septate hyphae with plenty of conidiogenous cells was seen around it. Keeping in mind a diagnosis of fungal endophthalmitis, topical steroids were stopped and the patient was taken up for a 23-gauge trans pars plana vitrectomy with intravitreal injection of amphotericin B (5 μg/0.1 mL) on day three. Vitreous biopsies obtained during the procedure were sent for microbiological testing and they revealed similar results. Topical treatment with fortified Voriconazole 1% (Vozole, Aurolab, India) six times a day, homatropine 2% (Homide) two times a day, brimonidine tartrate 0.2%/timolol maleate 0.5% (Combigan, Allergan, India) twice a day was continued and intravitreal amphotericin B was repeated the next day. With further deterioration in the condition, a prompt decision to change the anti-fungal was taken. The patient was given an intravitreal injection of 100 μg/0.1 mL of voriconazole. On the fifth day of incubation, culture on Sabouraud dextrose agar showed, large-sized brown to black round colonies, suggestive of a rare fungal organism known as Exophiala dermatitidis [Figure 2]b. The patient was started on oral fluconazole 150 mg (Zocon, FDC Limited, India) twice a day and continued on daily intravitreal injections of voriconazole for 6 days [Figure 1]b, followed by alternate-day injections for 2 weeks for a total of 13 injections. Topical steroids (difluprednate 0.05%, Diflupred, General, India) three times a day were started after 3 weeks, once visible clinical resolution was observed [Figure 1]c and were tapered very slowly over the next 4 months. While topical voriconazole drops were continued and slowly tapered over two months, systemic fluconazole (150 mg twice daily) was stopped after 6 weeks. By the end of 6 weeks, the patient had a dramatic improvement in the best-corrected Snellen's visual acuity to 6/9. Clear cornea, consolidation of the anterior chamber fibrinous exudate lying inferiorly with a clear view of the fundus was the result seen [Figure 1]d and [Figure 4]. A quiet anterior chamber was eventually noted by the end of 6 months [Figure 5].

Figure 1: Anterior segment photographs showing (a) the presence of circumcorneal congestion, corneal edema, and dense fibrinous exudate with improvement seen at day 10 (b), at 3 weeks (c) and at 6 weeks with a (d) quiet eye, clear cornea, and consolidated exudate lying inferiorly

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Figure 2: (a) Photomicrograph showing the presence of septate hyphae with coniodiogenous cells (lactophenol blue, 40×) and (b) brown–lack colonies of Exophiala dermatitidis on Sabouraud dextrose agar

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Figure 3: Bscan Image of the left eye showing dense vitritis

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Figure 4: Fundus photograph of left eye, post TPPV at six weeks duration showing clear media with a normal disc and vessels with dull foveal reflex

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Figure 5: Anterior segment photograph showing clear cornea with quiet anterior chamber and posterior chamber intraocular lens insitu at six months post-operatively

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Discussion

Exophiala species are widely distributed dematiaceous fungi in the environment and have been described as pathogens in cutaneous and subcutaneous infections. Melanin present in the cell wall is a major factor contributing to the pathogenicity of E. dermatitidis and resistance to host defenses and anti-fungal treatments along with other factors such as biofilm formation and adhesion.^[2] Intra-ocular infections are extremely rare and comprise only 6.8% of the infections caused by Exophiala.^[3] Until now, eight cases of Exophiala endophthalmitis have been reported worldwide,^[4],[5] of which two included E. jaenselmei species^[1] and one developed endogenous endophthalmitis.^[5] Three cases of postoperative delayed-onset E. dermatitidis endophthalmitis have been documented with two cases showing a successful outcome.^[4],[6] Quintero-Estades et al.,^[6] reported a BCVA of 6/24 by the end of 7 weeks, but the patient had to undergo two penetrating keratoplasties and was left aphakic. Homa et al.^[4] showed similar success wherein the BCVA with aphakic correction was 6/9. Amphotericin B injections were the mainstay treatment for Exophiala infections; however, other drugs such as voriconazole, posaconazole, fluconazole, flucytosine, terbinafine, and echinocandins have also been found to be effective against E. dematitidis even though drug susceptibility data are limited.^[2] In comparison to previous reports, our patient had a rapidly progressing presentation of late-onset fungal endophthalmitis. After prompt pars plana vitrectomy while preserving the IOL, he was successfully treated with multiple intravitreal voriconazole injections, voriconazole eye drops, and oral fluconazole to achieve visual acuity of 6/9. Intravitreal steroids have a controversial role in the treatment of fungal endophthalmitis.^[7],[8] Although multiple studies have shown improvement in visual outcomes after administering intravitreal steroids along with antifungal drugs, the results have been conflicting. In this case, because drug sensitivity patterns were not established owing to the paucity of data on the organism in question, we did not initiate intravitreal steroid treatment; instead, topical steroids were started after performing vitrectomy.

Conclusion

In conclusion, this case report describes a successful outcome in a case of late-onset E. dermatitidis fungal endophthalmitis using an aggressive medical and surgical approach. In accordance with previous case reports,^[4],[6] we also concur that voriconazole appears to be an effective drug against this class of dematiaceous fungi.

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

1.	Hofling-Lima AL, Freitas D, Fischman O, Yu MC, Roizenblatt R, Belfort R. Exophiala jeanselmei causing late endophthalmitis after cataract surgery. Am J Ophthalmol 1999;128:512-4.
2.	Kirchhoff L, Olsowski M, Rath PM, Steinmann J. Exophiala dermatitidis: Key issues of an opportunistic fungal pathogen. Virulence 2019;10:984-98.
3.	Zeng JS, Sutton DA, Fothergill AW, Rinaldi MG, Harrak MJ, De Hoog GS. Spectrum of clinically relevant Exophiala species in the United States. J Clin Microbiol 2007;45:3713-20.
4.	Homa M, Manikandan P, Saravanan V, Revathi R, Anita R, Narendran V, et al. Exophiala dermatitidis endophthalmitis: Case report and literature review. Mycopathologia. 2018;183:603-9.
5.	Gutierrez-Velez M, Hoang A, Weinstein J, Harkins K, Shah R. Endogenous Exophiala dermatitidis endophthalmitis. Am J Ophthalmol Case Rep 2020;19:100774.
6.	Quintero-Estades JA, Walter S, Valenzuela F, Amescua G. Delayed-onset postoperative endophthalmitis secondary to Exophiala. BMJ Case Rep 2015;2015:bcr2014208680.
7.	Majji AB, Jalali S, Das T, Gopinathan U. Role of intravitreal dexamethasone in exogenous fungal endophthalmitis. Eye 1999;13:660-5.
8.	Ching Wen Ho D, Agarwal A, Lee CS, Chhablani J, Gupta V, Khatri M, et al. A review of the role of intravitreal corticosteroids as an adjuvant to antibiotics in infectious endophthalmitis. Ocul Immunol Inflamm 2018;26:461-8.