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Dandy walkar malformation is characterised by cystic dilation of the fourth ventricle and an enlarged posterior cranial fossa with upward displacement of the tentorium, lateral sinuses, with agenesis or hypoplasia of vermis. Joubert syndrome is associated with distinctive brainstem and cerebellar malformation. It is rare to find mixed features of both the diseases in a baby. Case discussion A 18 year old female named X who is a primigravida with 17 weeks 1 day period of gestation has come for regular antenatal care. An anomaly scan was done, which showed bilateral enlarged ventricles, evidenced by floating or dangling choroid occupying less than 2/3rd of the ventricular cavities. Thalamopeduncular elongation is noted with an obtuse thalamopeduncular angle on axial images, saggital images showed a 'tectal breaking' involving tectum of midbrain .There was a dilated 4th ventricle which took an unusual posterior turn and was found to be communicating with a cystic structure measuring 6.3x3.6mm lying in midline caudal to the occcipital bone,cisterna magna was found to be obliterated with flattened cerebellum.These anomalous features were informed to the couple was counselled regarding the prognosis of the baby and need for termination of pregnancy. Conclusion Early diagnosis of such genetic defect and further evaluation of the parents for such genetic traits help the parents in decision making regarding the continuation or termination of the pregnancy and in educating them regarding the chances of bearing similar babies in future pregnancies.