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Oral Miltefosine as Adjunctive Treatment for Recalcitrant Acanthamoeba Keratitis

Dewan, Nikhil, BHSc; Ming, Wendy, MD; Holland, Simon P., MD, FRCSC; Yeung, Sonia N., MD, PhD, FRCSC; Iovieno, Alfonso, MD, PhD, FRCSC

doi: 10.1097/ICO.0000000000001968
Case Report

Purpose: Acanthamoeba keratitis is a rare, vision-threatening disease. Commercially available antiamoebics are poorly cysticidal and highly toxic, and therapeutic keratoplasties can be complicated by recurrence or graft failure. We aimed to discuss the use of oral miltefosine for treatment of recalcitrant Acanthamoeba keratitis.

Methods: A 44-year-old contact lens wearer presented with a 2-week history of red painful eye and decreasing vision. After poorly responding to topical corticosteroid on the presumptive diagnosis of anterior uveitis, she developed radial keratoneuritis. Corneal scraping was positive for Acanthamoeba. No clinical response to treatment was observed with topical chlorhexidine 0.02%, polyhexamethylene biguanide 0.02%, and oral voriconazole. She then underwent 2 therapeutic keratoplasties with prompt recurrence of the disease in the keratoplasty graft.

Results: Oral miltefosine was added to the treatment. She underwent a third penetrating keratoplasty 8 months later. The excised button was negative for amoeba. She continued miltefosine for 3 more months. No recurrence was observed after 30 months.

Conclusions: This case shows resolution of recalcitrant Acanthamoeba keratitis with oral miltefosine in an immunocompetent patient. Further clinical evidence would be needed to possibly incorporate this medication in the antiamoebic armamentarium.

Department of Ophthalmology and Visual Sciences, University of British Columbia, Vancouver, BC, Canada.

Correspondence: Alfonso Iovieno, MD, PhD, FRCSC, Department of Ophthalmology, University of British Columbia, 2550 Willow St, Vancouver, BC V5Z3N9, Canada (email:

The authors have no funding or conflicts of interest to disclose.

Presented at the Ocular Microbiology and Immunology Group 2018 Meeting; October 26, 2018; Westin Michigan Avenue, Chicago, IL.

Received November 20, 2018

Received in revised form March 09, 2019

Accepted March 12, 2019

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