Background: Asymptomatic compression of celiac artery (CA) by median arcuate ligament (MAL) is quite common, but simultaneous compression of other arteries by MAL is very rare. Case details: A 20-year-old man presented with post-prandial epigastric pain for the past 1 year and a 5 kg weight loss. On examination, he had a body mass index of 17.2 kg/m2 and was normotensive. Blood tests were unremarkable except for a mildly raised serum creatinine. A CT angiography (CTA) showed MAL-related 60% proximal CA narrowing and 40% proximal right renal artery (RRA) narrowing with a small right kidney. After multi-disciplinary team discussion, surgical MAL release at both sites was planned. The surgery was started laparoscopically to identify CA origin by a ‘top to down’ approach. The CA origin could not be visualised as it was looping behind the pancreatic neck caudally; hence the procedure was converted to open. CA origin was identified and overlying MAL divided. After kocherisation, RRA origin was found compressed by right limb of MAL and was divided. The operative duration was 200 mins with 50 mL blood loss. Results: The patient had an uneventful recovery. At 12-month, he is asymptomatic with serum creatinine normalised and gained 8 kg weight. A follow-up CTA at 2 months showed a normal CA and RRA. In this report, we discuss briefly about the current presentation, diagnosis and treatment of MAL compression of CA and other upper abdominal arteries. Conclusions: The learning point from this case is that one should carefully inspect all upper abdominal arteries in CTA for MAL-related compression as they may be asymptomatic, and this enables simultaneous treatment at these sites too in a single surgery.