We describe a case of Candida parapsilosis endophthalmitis with a cons
ecutive keratitis after phacoemulsification and posterior chamber intr
aocular lens implantation in an otherwise healthy eye. Despite aggress
ive medical and surgical management during a 2-year period, multiple e
pisodes recurred, with the development of an intracapsular plaque and
an infectious nidus on the corneal endothelium 6 months after the init
ial presentation. After subtotal removal of the culture-positive capsu
le, intravitreal and topical amphotericin B, and oral fluconazole, the
inflammation improved. However, the corneal endothelial plaque persis
ted with recurrent inflammation 2 months later, prompting debridement
of the culture-positive plaque and further removal of the culture-nega
tive capsular remnants and lens implant. The infection was quiescent f
or the subsequent 12-month period until recurrent intraocular inflamma
tion developed with enlargement of the endothelial plaque. Culture of
this plaque was again positive for C. parapsilosis. After debridement
and intraocular and topical amphotericin B, the eye has now been quies
cent for 13 months. This case demonstrates the development of a second
ary keratitis in an eye affected by pseudophakic C. parapsilosis endop
hthalmitis, with the posterior cornea serving as a sanctuary site for
the fungus despite aggressive management leading to recurrent infectio
n 1 year after the clinical disease appeared to be quiescent.