It has been established that maternal immunity developing before conce
ption protects the fetus from congenital toxoplasmosis. We observed a
case of congenital toxoplasmosis consecutive to a maternal toxoplasma
infection that had preceded pregnancy. A woman with normal immune syst
em developed toxoplasmosis 2 months before conceiving. No treatment wa
s given to this prepregnancy seroconverted patient. At 25 weeks of ame
norrhoea, the ultrasound examination showed a fetal cerebral ventricul
ar dilatation. Amniocentesis and cordocentesis showed fetal toxoplasmo
sis infection. Fetopathologic examination confirmed the diagnosis of c
erebral toxoplasmosis. The pathophysiology of maternal-fetal toxoplasm
a transmission and the role played by maternal immunodeficiency are di
scussed. This exceptional case-report showed the difficulties of the m
anagement in patients with 3-month pre-pregnancy toxoplasmosis and its
practical implications.