Purpose. To report a case of Lyme disease that presented with a single numm
ular unilateral interstitial keratitis. Methods, Case report and review of
the literature. Results. A 57-year-old black man who had contact with fresh
ly killed deer had a chief complaint of foreign-body sensation in his right
eye (OD) that had been diagnosed and treated for herpes simplex stromal ke
ratitis. The patient underwent a systemic workup for interstitial keratitis
. All results including RPR and MHA-TP were negative except for Lyme antibo
dy titer (enzyme-linked immunosorbent assay [ELISA]) 178 U/ml (normal, <159
U/ml). Conclusion. Interstitial keratitis from Lyme disease has been regar
ded as a bilateral disease in the literature. We present this infrequent oc
ular manifestation of Lyme disease as a rare single nummular unilateral pre
sentation.