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A mesenteric cyst is defined as a cyst that is located in the mesentery of the gastrointestinal tract and may extend from the base of the mesentery into the retroperitoneum. A case report of a patient with mesenteric cyst is presented. In addition, a systematic review was performed of English language literature on chylous mesenteric cysts in adult humans. Of the 18 articles included in the review, there were 19 cases of chylous mesenteric cysts reported. Male to female ratio was 1.4:1 with a median age of 46 years. A preoperative diagnosis of mesenteric cyst was made in four patients based on computed tomography. All patients underwent surgery and there were no reports of recurrence on follow up. Chylous mesenteric cyst is a rare entity that needs to be recognized whenever a preliminary diagnosis of intra-abdominal cystic mass is made.