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Exophiala phaeomuriformis Fungal Keratitis: Case Report and In Vivo Confocal Microscopy Findings

Aggarwal, Shruti M.D.; Yamaguchi, Takefumi M.D.; Dana, Reza M.D., M.P.H., M.Sc.; Hamrah, Pedram M.D.

Eye & Contact Lens: Science & Clinical Practice: March 2017 - Volume 43 - Issue 2 - p e4–e6
doi: 10.1097/ICL.0000000000000193
Case Report

Purpose: Corneal infections, particularly fungal keratitis due to rare fungal species, pose a diagnostic and therapeutic challenge because of difficulty in identification and varying susceptibility profiles. In this study, we report the first case of fungal keratitis because of Exophiala phaeomuriformis.

Methods: We report the clinical findings and microbial identification techniques of a case of fungal keratitis due to E. phaeomuriformis. An 84-year-old woman presented with redness, pain, and itching in the left eye for 2 weeks. Slit-lamp biomicroscopy revealed one broken suture from previous penetrating keratoplasty (PKP), black infiltrates at the 4-o'clock position, without an overlying epithelial defect and hypopyon. Microbial identification was based cultures on Sabouraud dextrose agar and DNA sequencing and correlations to laser in vivo confocal microscopy (IVCM; Heidelberg Retinal Tomograph 3/Rostock Cornea Module, Heidelberg Engineering) and multiphoton microscopy (Ultima Microscope; Prairie Technologies) images.

Results: Slit-lamp biomicroscopy revealed one broken suture from previous PKP, black infiltrates at the 4-o'clock position, without an overlying epithelial defect and hypopyon. Based on a clinical suspicion of fungal keratitis, antifungals and fortified antibiotics were started. However, the patient did not respond to therapy and required urgent PKP. After surgery, the patient was maintained on topical and systemic voriconazole and also topical 2% cyclosporine for 5 months because of possibility of scleral involvement noticed during surgery. At the end of the treatment period, her vision improved from hand motion to 20/40, with no recurrence observed in a follow-up period of 1 year. Results of diagnostic tests were supported by fungal elements in stroma on IVCM. Culture from the infiltrate grew black yeast. DNA sequencing led to the diagnosis of E. phaeomuriformis keratitis. Antifungal susceptibility testing revealed sensitivity to voriconazole.

Conclusion: This is, to our knowledge, the first reported case of E. phaeomuriformis fungal keratitis. Diagnostic testing included slit-lamp biomicroscopy, which revealed pigmented infiltrates, culture plates grew black yeast, microscopy showed branched fungal hyphae with budding conidia, and physiological features showed tolerance to high temperatures, nitrate assimilation, and ribosomal DNA sequencing. Collectively, these tests demonstrate unique features seen for this microorganism. High suspicion should be kept with pigmented infiltrates and with dark yeast on culture plates. Prompt and aggressive medical management with voriconazole or therapeutic PKP in nonresponsive cases is essential to prevent irreversible loss of vision.

Ocular Surface Imaging Center (S.A., P.H.), Department of Ophthalmology, Harvard Medical School, Massachusetts Eye and Ear Infirmary, Boston, MA; Cornea and Refractive Surgery Service (S.A., T.Y., R.D., P.H.), Department of Ophthalmology, Harvard Medical School, Massachusetts Eye and Ear Infirmary, Boston, MA; and Boston Image Reading Center and Cornea Service (P.H.), New England Eye Center/Tufts Medical Center, Tufts University School of Medicine, Boston, MA.

Address correspondence to Pedram Hamrah, M.D., Boston Image Reading Center and Cornea Service, New England Eye Center/Tufts Medical Center, Tufts University School of Medicine, 800 Washington St, Boston, MA, 02111; e-mail: phamrah@tuftsmedicalcenter.org

The authors have no conflicts of interest to disclose.

Supported by NIH K08-EY020575 (P.H.), Falk Medical Research Foundation (P.H.), Research to Prevent Blindness Career Development Award (P.H.), and MEEI Foundation (P.H.).

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The funding organizations had no role in the design or conduct of this research.

Accepted July 27, 2015

© 2017 Contact Lens Association of Ophthalmologists, Inc.