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Subpial haemorrhage is a rare cause of seizures in term neonates. A 3-day-old male infant, born at term with no history of perinatal hypoxia, presented with seizures and unremarkable physical examination in the interictal state. Imaging demonstrated left temporal subpial haemorrhage with the classic ‘yin-yang sign’ on MRI. The patient was subsequently diagnosed with factor XIII deficiency. Follow-up at 6 months and 12 months revealed encephalomalacia in the previous haemorrhagic areas with normal developmental milestones.