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Background: Fetal intra-abdominal cysts have an incidence of 1/500–1/1000 live births. Cysts can be physiologic or pathologic and can either spontaneously regress or require intervention and treatment. Cytomegalovirus (CMV) is the most common cause of congenital infection in the USA with an incidence of 0.2–2%. The risk of transmission is greatest with a primary maternal infection and the severity of fetal injury increases when transmission occurs in the first half of the pregnancy. An infectious etiology for a fetal intra-abdominal cyst has not been reported to the best of our knowledge. Case presentation: A 31-year-old multigravida presented at 19 weeks' gestation for an anatomical survey. The female fetus was noted to have a 2.4 × 2.0 × 3.1 cm echolucent cyst in the right side of the abdomen. Three weeks later, the cyst was not seen; however, there was free fluid and a few echogenic areas within the fetal abdomen. Maternal serum tested positive for CMV IgM and IgG titers, and the CMV IgG avidity test was 0.75, consistent with high avidity. At 27 weeks' gestation, ascites remained and a pericardial effusion was noted. Amniocentesis resulted in >2,000,000 copies of CMV DNA by polymerase chain reaction (PCR) in the amniotic fluid. The patient underwent termination of the pregnancy at 29 weeks of gestation. Conclusion: It would be important to consider an infectious etiology in the differential diagnosis of fetal intra-abdominal cysts as the outcome in the fetus with congenital CMV infection could be much different. Amniocentesis is considered the best option for the diagnosis of fetal congenital CMV infection if performed after 21 weeks' gestation and more than 6 weeks from maternal infection. [ABSTRACT FROM AUTHOR]