Case Report

Fungal endophthalmitis caused by Zygomycetes after phacoemulsification

Ferreira, Flávio C. MD*; Ishii, César K. MD; Kusabara, Alessandra A. MD; Godinho, João Victor V. MD; Hida, Richard Y. MD

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Journal of Cataract and Refractive Surgery Online Case Reports: July 2018 - Volume 6 - Issue 3 - p 43-46
doi: 10.1016/j.jcro.2018.02.001
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Abstract

Endophthalmitis is an intraocular inflammation that usually results from infection. It is a rare, potentially devastating and sight-threatening condition associated with a poor visual prognosis.1–3 Exogenous endophthalmitis is more common than endogenous endophthalmitis and is usually related to ocular surgery or trauma. Most infections are caused by bacteria, in particular after lens extraction because it is the most frequently performed surgery worldwide. Fungal endophthalmitis is less common and can also occur secondary to exogenous infections after surgery, trauma, or endogenous infections, especially in patients with risk factors.1,3–6 Most common fungal exogenous endophthalmitis are Candida species, Fusarium species, Paecilomyces species, Aspergillus species, and Acremonium species.1,4 Based on an extensive literature search in PubMed using the search terms “Zygomycetes” and “cataract or endophthalmitis,” only 1 case of Zygomycetes endophthalmitis after ocular surgery was reported in 1989.7

The purpose of this report is to describe a case of endophthalmitis after cataract surgery caused by Zygomycetes. To our knowledge, this is the second case of Zygomycetes endophthalmitis after ocular surgery described in the literature.

CASE REPORT

A 56-year-old man, a tailor from São Paulo with no significant medical history, presented to Santa Casa de Misericórdia de São Paulo General Hospital for cataract surgery in both eyes. On March 2016, he had routine phacoemulsification and intraocular lens (IOL) implantation in the right eye. During the procedure, anterior vitrectomy was performed, the IOL was placed in the sulcus, and 1 nylon 10-0 suture was placed because of a posterior capsule rupture.

During the postoperative follow-up, the patient's corrected distance visual acuity (CDVA) was 20/20. Thirty-eight days postoperatively, the patient reported slightly blurred vision, ocular pain, and red eye. Slitlamp examination showed moderate ocular hyperemia, anterior chamber reaction, flare, and superior corectopia. An inferior (at 6 o'clock), round, white, well-delimited mass on the iris was observed in the operative eye (Figure 1). The right eye fundus examination showed a mild vitreous opacity only. A presumptive diagnosis of inflammation caused by residual lens cortex was made, and oral prednisone (60 mg) with a gradual taper was prescribed.

Figure 1.
Figure 1.:
Round white mass in anterior chamber.

There was no significant improvement during the next 7 days; therefore, anterior chamber washout with a balanced salt solution was performed. The round mass that was adhered to the iris was sent for microbiology and histopathology study. The patient reported partial improvement of the symptoms on the next day, although the blurred vision remained.

Microbiology analysis did not show any growth. Histopathology showed multinucleated fungi and coenocytic hyphae, rarely septate, suggesting Mucorales from the Zygomycetes class (Figures 2, 3, and 4).

Figure 2.
Figure 2.:
Biopsy of mass in the anterior chamber (hematoxylin–eosin stain; original magnification ×40).
Figure 3.
Figure 3.:
Biopsy of mass in the anterior chamber at highest magnification (original magnification ×100) (hematoxylin–eosin stain).
Figure 4.
Figure 4.:
Magnification of the fragment showing Zygomycetes, with coenocytic hyphae, predominantly aseptate (Grocott methenamine silver; original magnification ×400).

On the 60th postoperative day, the patient presented with a CDVA of 20/30, ocular pain, moderate hyperemia, moderate anterior chamber reaction with recurrent fibrinous, and a similar elevated mass at 6 o'clock. Anterior chamber washout was performed with liposomal amphotericin B (0.5 mg/mL), and an aqueous humor sample was collected. Systemic intravenous liposomal amphotericin B (3 mg/kg/day) diluted in glucose solution 5.0% was performed. After 5 days of systemic treatment, the vitreous opacity remained and the CDVA declined to 20/200 as a result of cystoid macular edema. Therefore, a pars plana vitrectomy (PPV) was performed and a sub-Tenon injection of triamcinolone given. Twelve days after systemic amphotericin B treatment, the patient was discharged from the hospital because of the favorable clinical improvement. There were no major findings in anterior segment slitlamp examination. Three weeks after the last procedure, the CDVA was 20/20.

DISCUSSION

The incidence of endophthalmitis after cataract surgery varies from 0.072% to 0.13%.1,3,4,8,9 According to the literature, most cases of infectious endophthalmitis after surgery (83%) are caused by a variety of bacteria, with few reports of fungal agents.9,10 These infections can be caused by exogenous or endogenous microorganisms. In fungal cases, only 5.0% of infections are caused by exogenous agents.11 Treatment options are limited for this uncommon disorder, and outcomes are often poor, with 50% of patients having serious visual impairment and permanent vision loss.4,6,12

Fungal infections are more common in patients with risk factors, such as the chronic use of steroids or antibiotics during the postoperative period, previous organ transplantation, intravenous drug abuse, and malignant or debilitating disease.8 The epidemiology and etiology vary according to the anatomic site and the country's geography and climate. Moreover, they vary based on predisposition, and risk factors.4,11

Fungal endophthalmitis makes up between 8.6% and 18.6% of all intraocular infections.3Candida albicans and Aspergillus species are the most frequent agents isolated in cultures.1,10 In areas with tropical climates, Aspergillus and Fusarium species are relatively common.2,11 When endophthalmitis is exogenous, it can be related to other rare species. Postoperative fungal endophthalmitis agents are mainly caused by Candida species (C glabrata, C famata, and C parapsilosis).1,6 Furthermore, the rare species of Trichophyton, Trichosporon asahii, and Aspergillus ustus have also been reported in endophthalmitis after cataract surgery.5,12,13 We found only 1 other report in the literature of postoperative endophthalmitis caused by Zygomycetes.7 In ophthalmology, according to our search protocol, 2 other cases were reported; however, they were not related to a history of ocular surgery.14,15 Zygomycosis, an opportunistic pathogen that belongs to class Zygomycetes and order Mucorales, rarely occurs in immunocompetent hosts. The most common sites are pulmonary and rhinocerebral.16,17

Rhizopus species is the genera most commonly associated with Mucormycosis (order Mucorales). However, its identification was impossible in all 3 case reports (2 endogenous) of ocular infections related to Zygomycosis. Other pathogens such as Mucor, Rhizomucor, Absidia, Apophysomyces, Saksenaea, Cunninghamella, Cokeromyces, and Syncephalastrum can also cause human disease.8,16,17 In the literature, there are many reports of negative culture growth for Zygomycetes isolates on autopsy resulting from manipulation of the specimen, especially because the hyphae are aseptate. When compressed, cytoplasm is usually lost, preventing the fungus from growing.16,17 In contrast to our case, most fungal ophthalmologic diseases, such as keratitis or endophthalmitis, are caused by nonpigmented and septate filamentous fungi.1,8,10

A previous study11 described 20 cases of fungal endophthalmitis caused by Fusarium oxysporum. The endophthalmitis occurred within 16 days and 79 days (mean 31.3 days) after cataract surgery, causing a decrease in visual acuity, iritis, an anterior chamber reaction, and hypopyon. Contamination of materials, the ophthalmic viscosurgical device, IOL, or fluids for irrigation might increase the risk for fungal infection after any surgical procedure. Another possibility is that the injection of the IOL damages the fungal colonies surrounding the incision, allowing the fungi to enter the eye during IOL implantation.5 Tarkkanen et al.18 report that fungal endophthalmitis caused by Paecilomyces variotii can be related to the air-conditioning system.

Treatment of fungal infections can be difficult and depends on the site of infection, the causative organism, and the drug used for treatment and its mode of administration. Treatment can include systemic therapy or intravitreous injection, resolution of the patient's underlying condition, and surgical intervention. Amphotericin B is considered the first choice for an intravenous injection.

Recently, caspofungin and voriconazole have been considered effective alternatives to amphotericin B for fungal intraocular infection.10 Gonul et al.13 reported successful treatment of fungal endophthalmitis with intravitreous/systemic voriconazole, PPV, and removal of the IOL with entire lens capsule when amphotericin B failed.

The improvement of antifungal agents for ophthalmic infection is a challenge because most of these drugs are not water soluble, which can affect tissue penetration. Moreover, amphotericin B and its derivatives are toxic with considerable side effects.8

In conclusion, fungal infections are rare after intraocular surgery and an unusual clinical finding can be an important hint in the diagnosis of rare microorganisms. Unfortunately, it can take a relatively long time to diagnose rare cases, leading to unsuccessful treatment in some cases.

Disclosures:

None of the authors has a financial or proprietary interest in any material or method mentioned.

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