An accessory middle cerebral artery is one variation of the intracranial vasculature that may be a source of misinterpretation by clinicians dealing with cerebrovascular diseases. We report a case of an elderly female found to have bilateral accessory middle cerebral arteries, who presented with the rupture of an aneurysm of the anterior part of the circle of Willis. Accessoty middle cerebral arteries are rare anatomical findings and the bilateral occurrence is exceedingly rare. We believe this to be the first report of bilateral accessory middle cerebral arteries associated with an aneurysm of the anterior cerebral-anterior communicating arteries. The anatomical and clinical relevance of this variation is described. Copyright © 2006 Via Medica.