Celio-mesenteric trunk (CMT) is a rare vascular variation of the ventral branches of the abdominal aorta that supply blood to the mesentery and the gut. This rare variation is seen in 2/100.000 population. The presence of this anomaly is associated with an increased risk of mesenteric ischemia in the case of proximal occlusion. Median arcuate ligament (MAL) syndrome is a controversial entity characterized by compression of the celiac axis by MAL causing post-prandial pain. We report the fourth case of MAL compression syndrome involving a CMT in the world