Cerebral sinovenous thrombosis occurring in utero has rarely reported and its true prevalence or underlying cause remains uncertain. Antenatal counseling of couples diagnosed with such entity is still very challenging having very few case reports to rely on. Unfortunately, natural history in utero or long‐term postnatal neurodevelopment outcome vary widely between cases. We experienced a case of cerebral sinovenous thrombosis that was prenatally diagnosed, thus hereby report the case with literature review.