Exophiala Dermatitidis Endophthalmitis: Case Report and Literature Review
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View metadata, citation and similar papers at core.ac.uk brought to you by CORE provided by SZTE Publicatio Repozitórium - SZTE - Repository of Publications Mycopathologia https://doi.org/10.1007/s11046-017-0235-4 ORIGINAL PAPER Exophiala dermatitidis Endophthalmitis: Case Report and Literature Review Mo´nika Homa . Palanisamy Manikandan . Veerappan Saravanan . Rajaraman Revathi . Raghavan Anita . Venkatapathy Narendran . Kanesan Panneerselvam . Coimbatore Subramanian Shobana . Mohammed Al Aidarous . La´szlo´ Galgo´czy . Csaba Va´gvo¨lgyi . Tama´s Papp . La´szlo´ Kredics Received: 18 May 2017 / Accepted: 16 December 2017 Ó Springer Science+Business Media B.V., part of Springer Nature 2018 Abstract We report a case of a 59-year-old male and aggressive antifungal therapy combined with patient with a postoperative fungal infection of the left surgical intervention is necessary to prevent vision eye. A dark-pigmented yeast, Exophiala dermatitidis loss in cases of endophthalmitis due to Exophiala (previously known as Wangiella dermatitidis), was species. Beside the case description, we also aim to identified from the culture of the biopsy taken from the provide a literature review of previously reported eye posterior capsule. The infection was successfully infections caused by Exophiala species in order to help eradicated by a combination of surgical and medical the future diagnosis and management of the disease. (i.e., voriconazole and fluconazole) treatment. This is the first report of successfully treated E. dermatitidis Keywords Dematiaceous fungi Á Exophiala Á endophthalmitis, which demonstrates that a prompt Keratitis Á Endophthalmitis Á Antifungal susceptibility Mo´nika Homa and Palanisamy Manikandan have been contributed equally to this work. M. Homa Á T. Papp P. Manikandan MTA-SZTE ‘‘Lendu¨let’’ Fungal Pathogenicity Greenlink Analytical and Research Laboratory India Mechanisms Research Group, Ko¨ze´p Fasor 52, Private Limited, Coimbatore, Tamilnadu 641 014, India Szeged 6726, Hungary K. Panneerselvam M. Homa Á L. Galgo´czy Á C. Va´gvo¨lgyi Á Research Department of Microbiology, M. T. Papp (&) Á L. Kredics R. Government Arts College, Department of Microbiology, Faculty of Science and Mannargudi, Tiruvarur District, Tamilnadu 614 001, India Informatics, University of Szeged, Ko¨ze´p Fasor 52, Szeged 6726, Hungary C. S. Shobana e-mail: [email protected] Department of Microbiology, PSG College of Arts and Science, Coimbatore, Tamilnadu 641 014, India P. Manikandan Á V. Saravanan Á R. Revathi Á R. Anita Á V. Narendran Aravind Eye Hospital and Postgraduate Institute of Ophthalmology, Coimbatore, Tamilnadu 641 014, India P. Manikandan Á M. A. Aidarous Department of Medical Laboratory Sciences, College of Applied Medical Sciences, Majmaah University, Al Majma’ah 11952, Saudi Arabia 123 Mycopathologia Table 1 A retrospective summary of Exophiala eye infections reported since 1990 Aetiological agent Age/ Country Therapyb Underlying conditionc Outcome References Gendera Keratitis E. dermatitidis 35/M Czechoslovakia AMB NF1, Leukoma [7] immunodeficiency E. jeanselmei 42/M Saudi Arabia NTM, MCN, AMB – Improved [8] E. jeanselmei 58/F Israel NTM, AMB – Improved [6] E. dermatitidis 52/M USA AMB – Improved [9] E. jeanselmei 39/F USA AMB, ITC – Improved [10] E. jeanselmei 41/M USA VRC, NTM, KTC, – Improved [11] SI E. phaeomuriformis 84/F USA VRC, SI – Improved [12] Subconjunctival mycetoma E. dermatitidis 44/F China SI – Improved [13] Endophthalmitis E. dermatitidis 75/F USA AMB, SI Diabetes Enucleation [14] E. dermatitidis 31/M France ITC, AMB, SI – Failed [15] E. jeanselmei 67/F Brazil AMB Purpura Atrophy [16] E. jeanselmei 52/F Brazil AMB Diabetes Atrophy [16] E. dermatitidis 60/M USA AMB, SI Crohn’s disease, Enucleation [17] Herpes zoster keratitis Exophiala sp. 65/F Puerto Rico VRC, FLC, SI Diabetes Improved [18] E. dermatitidis 59/M India VRC, FLC, SI – Improved Present case aF female; M male bAMB amphotericin B; FLC fluconazole; ITC itraconazole; KTC ketoconazole; MCN miconazole; NTM natamycin; PSC posaconazole; SI surgical intervention; VRC voriconazole cNF1 neurofibromatosis type I Background Exophiala species are extremely rare. Most cases occur after a penetrating injury or post-eye surgery [6]. Fungal eye infections are among the main causes of Though the presented case is the fourth documented visual disability in tropical and subtropical countries, case of endophthalmitis caused by E. dermatitidis such as India. Besides hyaline fungal species, which across the globe according to a deep search of the are responsible for the majority of keratomycoses and available literature (Table 1)[7–18], it is unique, as it fungal endophthalmitis in the country, dark-pig- is the first report with a successful treatment outcome. mented fungi are also associated with emerging cases Human ocular infection due to E. dermatitidis has not [1, 2]. The cosmopolitan genus Exophiala comprises been documented in India before. dimorphic dematiaceous fungi commonly isolated from soil and plant debris [3, 4]. These fungi are the clinically most relevant black yeasts causing various Case Presentation uncommon forms of cutaneous, subcutaneous and disseminated (e.g., pneumonia and brain abscess) A 59-year-old male patient presented at the Cornea human infections [5], but eye infections due to Department of Aravind Eye Hospital and Postgraduate 123 Mycopathologia Institute of Ophthalmology (Coimbatore, Tamilnadu, Laboratories Ltd., India) eight times a day, plus India) in December 2012 with complaints of decreased voriconazole (1% (w/v); Vozole, Aurolab, India) eye vision, pain and redness in his left eye for the past drops six times a day and homatropine (2% (w/v); 15 days. The patient gave a history of an uneventful Homide, Warren, India) eye drops twice a day. The cataract surgery with intraocular lens implantation in topical treatment continued till the time of the his left eye performed 4 months earlier. He had good intraocular lens removal (1 month from the first vision in the first month following surgery, that is, vitreous tap). visual acuity (VA) was 6/6. Three months later, he Vitreous biopsies obtained during the procedure presented with gradual deterioration of vision (VA were plated on 5% sheep blood agar (SBA; HiMedia 6/60). The patient was otherwise in good health and Laboratories, India), potato dextrose agar (PDA; had no known allergies. HiMedia Laboratories, India) and thioglycolate broth On examination, the best corrected visual acuities (HiMedia Laboratories, India). A part of the sample were 6/6 in the right eye and 6/60 in the left eye. was sent for molecular identification (Xcyton Diag- Intraocular pressure as measured by Goldmann appla- nostics Limited, Bengaluru, India). Gram stained nation tonometry was 15 mm in the right eye and smears and 10% potassium hydroxide wet mount 13 mm in the left eye. Slit lamp examination of the were also investigated. The smears as well as the right eye was within normal limits except for an early cultures proved to be negative for fungal filaments. nuclear sclerotic cataract of grade 1. Examination of Molecular analyses were negative for all the tested the left eye revealed circumcorneal congestion, a clear bacteria and fungi. Postoperatively the same medica- cornea and 3? cells and 1? flare in the anterior tions were continued. Prednisolone acetate (1% (w/v); chamber. Texture and color of the iris were normal. Predforte, Allergan, India) was added at a frequency of The pupil was pharmacologically dilated and unre- six times a day. Dexamethasone (8 mg in 2 ml; sponsive to light. A posterior chamber intraocular lens Dexadran, Searle Labs Pvt. Ltd. India) was adminis- was present, and a fluffy grayish white growth was tered intravenously twice a day for 2 days and was noted on the posterior capsule (Fig. 1a, b). subsequently replaced by oral prednisolone Indirect ophthalmoscopy of the right eye was (40 mg/day; Omnacortil, MacLeods Pharmaceuticals within normal limits, but in the left eye moderate Ltd., India), tapered over a period of 15 days. vitritis was observed. The patient underwent vitreous On subsequent follow-ups, the visual acuity was tap with a 24-gauge needle inserted into the central gradually improving (i.e., VA 6/9 after 1 week post- vitreous cavity along with intravitreal injections of vitreous tap), but the anterior chamber inflammation moxifloxacin (500 lg/0.1 ml; Vigamox, Alcon Labo- persisted, and fluffy deposits on posterior surface of ratories, USA) and voriconazole (100 lg/0.1 ml; the intraocular lens persisted. Oral steroids were Aurolab, India). He was placed on topical moxi- discontinued, and the topical steroid dose (6 times floxacin (0.5% (w/v); Vigamox, Alcon, India) and per day; Predforte, Allergan, India) was tapered to four fortified tobramycin (14 mg/ml; Toba, Sun Pharma times. A repeated vitreous biopsy was performed on Fig. 1 Slit lamp biomicroscopy (a) with retro illumination (b) showing the fungal colonies on the posterior surface of the intraocular lens 123 Mycopathologia the 9th day following the first vitreous tap under local month, topical steroids (Predforte, Allergan, India) anesthesia using a 25-gauge vitreous cutter through were started with a very low dose twice a day, and the pars plana route. Care was taken to take the sample tapered very slowly over the next 4 months. The best at the location of the fluffy growth with localized corrected Snellen’s visual acuity with aphakic correc- posterior capsulectomy and anterior vitrectomy. Spec- tion was 6/9. imens thus taken from the posterior capsule and For the molecular identification, genomic DNA was anterior vitreous were plated on SBA, chocolate agar extracted using the MasterPure Yeast DNA Purifica- (CA; HiMedia Laboratories, India), PDA and Lowen- tion Kit (Epicentre Biotechnologies, USA) following stein Jensen medium (HiMedia Laboratories, India). A the instructions of the manufacturer. PCR amplifica- direct microscopic examination of Gram’s smears tion was carried out by targeting the internal tran- clearly revealed fungal filaments. The topical steroid scribed spacer (ITS) region using ITS1 and ITS4 therapy was stopped, and the frequency of topically primers [19].