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BACKGROUND Lymphocytic choriomeningitis virus（LCMV） is a neglected rodent-borne arenavirus associated with transplacental transmission and fetal infection.AIM To summarize the epidemiological, clinical, and diagnostic features of reported patients with congenital LCMV infection.METHODS A literature search was conducted in PubMed, Medline, Google Scholar, and ResearchGate. The keywords used were ‘congenital lymphocytic choriomeningitis virus,’ and 48 studies were included. In addition, we conducted a relevant search by Reference Citation Analysis（RCA）（https://www.referencecitationan alysis.com）.RESULTS The results have shown 27 reports of congenital LCMV infection in 86 patients, with 52.73% of them being males. Patients presented with chorioretinitis（83.53%）, hydrocephalus（54.12%）, and psychomotor retardation or developmental delay（54.12%）. Computed tomography and/or magnetic resonance imaging most often demonstrated ventriculomegaly（74.07%）, periventricular calcifications（66.67%）,and microcephaly（40%）. Most mothers of congenitally infected infants were exposed to rodents during pregnancy, predominantly mice, with flu-like symptoms mainly occurring during the first two trimesters of gestation. Mortality in congenitally infected children was 16.47%. The diagnosis of congenital LCMV infection was confirmed serologically in most patients（86.67%）.CONCLUSION LCMV is still an insufficiently recognized fetal teratogen that often leads to long-term neurologic sequelae. Clinicians need to be familiar with LCMV and its potential teratogenic effect and as well as to effectively differentiate LCMV from other TORCH（T: Toxoplasma gondii, O: Other pathogens, R: Rubella virus, C: Cytomegalovirus, H: Herpes simplex virus） pathogens.