Misoprostol exposure in the first trimester of pregnancy has been related to congenital malformations, particularly the mobius sequence and terminal transverse limb defects. An unusual case of a 25-years old primigravida who was exposed to misoprostol at 8-weeks gestation is presented here. Failing to medical termination, the pregnancy continued and a live baby girl was delivered preterm at 35-weeks gestation with multiple anomalies like fronto-nasal bone, scalp defects and protrusion of duramater. Microcephaly and equinovarus deformities were also present. Hence, clinicians must be aware of the teratogenic risk of misoprostol with especial context to its unsuccessful use in the first trimester of pregnancy. For the ongoing pregnancies, patient must also be informed in detail.KEY WORDS: Misoprostol. First trimester exposure. Teratogenicity. Cranial bone and scalp defect.