Background: Moellerella wisconsensis, a member of the family of Enterobacteriaceae, although isolated widely in nature, rarely causes infections in humans. Herein, we report a case of isolation of M. wisconsensis from pigtail end culture, urine culture and blood culture in a 76-year-old patient. Objective: To systematically address all the relevant information regarding M. wisconsensis through literature. Methods: We searched PubMed and Scopus databases up to January 2022 and performed a qualitative synthesis of published articles reporting infection from M. wisconsensis in humans. Results: We identified 25 records on PubMed and 43 additional records on Scopus. After removing duplicates, we examined in detail 15 articles. Ten studies with a total of 17 cases were included in our systematic review. Nine studies described isolated case reports, while 1 study described 8 cases. The origin of the infection was the alimentary tract in 9 cases, gallbladder in 4 cases, peritoneal cavity in 2 cases, respiratory tract in 1 case and hemodialysis catheter insertion site in 1 case. In 3 of the aforementioned cases M. wisconsensis was also isolated in blood cultures. Conclusion: Physicians should be aware that M. wisconsensis can be present in multiple clinical specimens and that the antibiotic resistance profile of the isolates may pose significant challenges.