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We report a case of Chilaiditi sign associated with thecongenital anomaly.A 45-year-old male patient with a 15-year history of hepatitis Bvirus(HBV)infectionwasadmittedforfatigue.Hehadnoabdominalsurgery or trauma history, nor any medications history.The physicalexamination was unremarkable. Apart from positive serum HBVmarkers, the laboratory values were normal. The chest radiographshowed a distended loop of large intestine below an elevated righthemidiaphragm(Fig. 1a).Abdominalcomputedtomographyshowedhepatodiaphragmaticcolonicinterposition,segmentalagenesisoftheright lobe of the liver with enlarged left hepatic lobe, isolated portalvein(Fig. 1b,arrow),andtheinferiorvenacavapassthroughcaudatelobe and the remains right hepatic lobe (Fig. 1c, arrow).Agenesis or hypogenesis of the right liver lobe is considered to becausedbyafailureoftherightportalveintodevelop,oranerrorofmutualinduction between the primitive diaphragm and the endodermal diver-ticulum representing the primitive liver.