Recurrent Lecythophora mutabilis Keratitis and Endophthalmitis After Deep Anterior Lamellar Keratoplasty FREE

Lecythophora mutabilis is a mold that has been rarely reported as the cause of ocular infection: 1 case of endophthalmitis¹ and 1 case of blebitis.² We report a case of L mutabilis keratitis with intraocular invasion that required aggressive treatment.

A 53-year-old woman was treated for a contact lens–associated Pseudomonas aeruginosa corneal ulcer in the right eye. Fortified antibiotic eyedrops were used, but corticosteroids were not. On resolution of this infection, she developed a corneal infiltrate caused by a species of fungus that was not identifiable by a local laboratory. She began hourly treatment with voriconazole, 1%, eyedrops; because of poor clinical response, treatment with natamycin, 5%, eyedrops every 2 hours was later added. Because of the progressing infiltrate, a therapeutic deep anterior lamellar keratoplasty was performed. Postoperatively, treatment with prednisolone acetate, 1%, eyedrops every 2 hours and gatifloxacin, 0.3%, eyedrops 4 times daily was prescribed. One week postoperatively, a new infiltrate at the graft-host interface was discovered.

The patient was referred to us with visual acuity of hand motions OD. In addition to the stromal infiltrate, white clumps of presumed fungal balls were seen in the anterior chamber adherent to the cornea and iris (Figure 1). After an anterior chamber tap and injection of 0.1 mL of voriconazole (1 mg/mL), treatment with hourly voriconazole, 1%, eyedrops was started. Tap and injection of voriconazole were repeated daily for the next 2 days. No anterior chamber samples demonstrated microbiological growth. Because of the progressing infiltrate, a therapeutic penetrating keratoplasty was performed. Intraoperatively, the presumed fungal collections were peeled from the iris and the anterior chamber was irrigated with voriconazole (1 mg/mL). On histopathologic analysis, fungal filaments were seen penetrating through the cornea into the anterior chamber (Figure 2).

Postoperatively, the patient was treated with voriconazole, 200 mg orally twice daily, gatifloxacin, 0.3%, eyedrops 4 times daily, and prednisolone acetate, 1%, eyedrops 4 times daily. Six days after surgery, recurrence of the fungal infection in the host at the wound edge was noted. Tap and injection with voriconazole were performed and continued daily. Amphotericin B, 0.1 mL (100 μg/mL), was injected on the third day, and hourly amphotericin B, 0.15%, eyedrops were added owing to continued progression of the infiltrate. Five days later, the patient underwent a limbus-to-limbus penetrating keratoplasty because of worsening infection. Intraoperatively, the areas of the host cornea that clinically appeared to be infiltrated (superiorly from the 9-o’clock to 3-o’clock positions) were treated with double freeze-thaw cryotherapy before the cornea was excised, and the anterior chamber was irrigated with amphotericin B (100 μg/mL) and voriconazole (1 mg/mL).

Daily anterior chamber tap and injection of amphotericin B were continued for 4 days. Treatment with topical amphotericin B, voriconazole eyedrops, and oral voriconazole was continued for 6 weeks. At the last follow-up visit 6 months after the last procedure, although the graft had failed, there was no evidence of active infection. Cultures from the corneal tissue from the first penetrating keratoplasty had been sent to the Fungal Testing Laboratory, San Antonio, Texas, and ultimately grew L mutabilis sensitive to both amphotericin B and voriconazole by broth dilution sensitivity testing.

L mutabilis ocular infections are extremely rare and potentially very aggressive. Fortunately, our case responded to amphotericin B in contrast to the 2 reported cases in the literature.^1- 2 Although deep anterior lamellar keratoplasty has been reported by some investigators to be an alternative to penetrating keratoplasty in the setting of active corneal infections,³ in this instance a full-thickness graft may have been a better choice given that the infection recurred in the host bed and that fungus was found penetrating into the anterior chamber.

Correspondence: Dr Jeng, Department of Ophthalmology, University of California, San Francisco, 10 Koret Way, K-304, San Francisco, CA 94143-0730 (jengb@vision.ucsf.edu).

Financial Disclosure: None reported.

Funding/Support: This work was supported in part by a grant from That Man May See and Research to Prevent Blindness to the Department of Ophthalmology, University of California, San Francisco.