Objective: The aim of this study was to identify the clinical significance associated with ptyalism gravidarum in Japanese singleton pregnancies. Methods: Data were collected from 22 patients complicated by ptyalism gravidarum whose symptoms continued until delivery and from 7,743 unaffected controls. Results: The incidence of ptyalism gravidarum in the Japanese pregnant women was about 0.3 %. Using multiple logistic regressions, ptyalism gravidarum was associated with history of hyperemesis gravidarum ( p < 0.01), neonatal male sex ( p = 0.02) and small for gestational age infants ( p = 0.04). Conclusions: Ptyalism gravidarum may be a distinctive condition leading to adverse perinatal outcomes.