Fluconazole has been associated with various teratisms in animals, inc
luding craniofacial ossification defects, thin, wavy ribs, and renal p
elvis defects. We describe three infants born to women who were receiv
ing fluconazole through or beyond the first trimester of pregnancy. Al
l of the infants had congenital anomalies; no other drug was implicate
d. Only one of the three infants survived. Their anomalies, similar to
those observed in animal studies, were largely craniofacial, skeletal
(i.e., thin, wavy ribs and ossification defects), and cardiac. One of
these infants was previously reported as having Antley-Bixler syndrom
e; however, given the chronology described herein and the similarity o
f this infant to the others, we conclude that her deformities also rep
resent the potent teratogenic effect of fluconazole.