We present a case of congenital toxoplasmosis (TXP) in a woman with Toxoplasma gondii infection more than 6 months before conception. The woman has been treated with adalimumab for ankylosing spondylitis for 4 years until 5 months before conception. TXP serology at the first trimester was compatible with infection prior pregnancy. An ultrasound performed at 26 weeks gestation (WG) showed cerebral echogenic lesions compatible with intrauterine infection. Amniocentesis was performed which confirmed TXP fetal infection. Termination of the pregnancy was performed upon parent's requests and the fetal autopsy confirmed the diagnosis. Here, we discuss the potential role of immunosuppressive treatments, such as adalimumab, in the risk of congenital toxoplasmosis and the importance of counseling before pregnancy.