Background: The incidence of intracranial aneurysm in childhood is rare, especially in infancy. In the literature, most of childhood intracranial aneurysms occur in the posterior and middle cerebral circulation. We report a case of a ruptured giant left distal anterior cerebral artery aneurysm in a 2-month-old baby. This report will discuss the rarity of this case as well as the diagnostic and surgical challenges in treating this condition. Case Report: A 2-month-old baby girl presented to our centre with a 1-day history of multiple afebrile seizures. A CT scan of her brain showed a large frontal interhemispheric acute bleed with intraventricular extension and acute hydrocephalus. An external ventricular drain was inserted and she was nursed in the Paediatric Intensive Care Unit. Subsequent CT angiogram and 4-vessel angiogram showed a giant aneurysm originating from the A3 segment of the left anterior cerebral artery. She underwent craniotomy and clipping and excision of the giant aneurysm. Discussion: Giant distal anterior artery aneurysms are very rare in infants. They are more commonly associated with the posterior and middle cerebral arteries. The aneurysms in infants tend to be larger compared to adults and they commonly present with subarachnoid haemorrhage and seizures. CT angiogram and 4-vessel cerebral angiogram are important diagnostic tools in this case. The angiogram demonstrated no distal runoff from the aneurysm and this assisted in our planning for surgical treatment of the aneurysm. A bifrontal craniotomy was done and the giant aneurysm was approached interhemispherically. The proximal and distal parent vessel was clipped and the aneurysm wall was excised. The baby recovered from surgery and did not require any cerebrospinal fluid diversion. Conclusion: Giant distal anterior cerebral artery aneurysms are rare in infants. Detailed angiographic investigation is important. Surgery is the treatment of choice, although there may be a role for endovascular intervention.

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