Unlike fenestration of the posterior cerebral arterial circulation, fenestration of the anterior cerebral arterial circulation has not been well described. We investigated the location and configuration of fenestration of the middle cerebral artery (MCA) detected by magnetic resonance (MR) angiography. We found 6 fenestrations of the MCA among cranial MR angiography images obtained from about 2,000 patients during the past 9 years at our institution using either of two 1.5T imagers. All images were obtained by the three-dimensional time-of-flight technique. Maximum-intensity projection images in the horizontal rotation view were displayed stereoscopically. All 6 fenestrations had small slit-like configurations, five located at the proximal M1 segment, the other, at the middle M1 segment. No associated aneurysm was found. Although MCA fenestration is extremely rare and cerebral artery fenestration usually has no clinical significance, an aneurysm can arise at the proximal end of the fenestration. Thus, recognizing MCA fenestration is important when interpreting cranial MR angiograms.