Cortical dysplasia is a neuronal migration disorder occasionally associated with anomalous draining veins. However, to our knowledge, no intracranial haemorrhage in conjunction with this association has been reported in the literature. We herein report a 7-month-old baby girl with cortical dysplasia associated with an ipsilateral cortical draining vein and complicated with subdural haemorrhage, with the diagnosis made by computed tomography and magnetic resonance imaging. This case demonstrated that patients with this condition could have an excellent prognosis even when the anomalous veins coexist with intracranial haemorrhage. We postulate that venous rupture may be the cause of this condition and further discuss the possible pathophysiology.