Purpose Present two patients on two different novel FGFR inhibitors who developed trichiasis with dry eye syndrome. Observations Two patients developed trichiasis and dry eye following FGFR inhibitor therapy. Treatments included artificial tears, lifitegrast or cyclosporine, and epilation as needed. One patient discontinued treatment with AZD4547 due to the severity of the ocular adverse effects. Conclusions and Importance Trichiasis has not yet been reported in patients receiving AZD4547 or INCB054828 treatments and may represent a rare adverse effect of these drugs. Continued research is necessary to determine whether there is a definite link between these FGFR inhibitors and the development of trichiasis.