International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties.Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations. Agenesis of gallbladder is a rare (13-65 cases/100,000) anomaly, in which about 23% patient presents with symptoms of biliary disease. In these patients, ultrasonography (USG) abdomen frequently falsely reveals shrunken or contracted gallbladder and sometimes non-visualization of gallbladder (GB) in GB fossa. Basis of these misinterpreted reports these patients undergone unnecessary surgery and may encounter iatrogenic biliary tract injuries and portal injuries because of excessive dissection to find out the absent gallbladder and ectopic gallbladder.Case Report: A 40-year-old female attended surgical outdoor with complain of pain right hypochondrium and dyspepsia since last four years patient followed-up with USG abdomen which was suggestive of chronic cholecystitis with cholelithiasis, and common bile duct (CBD) was normal in diameter on the basis of clinical symptoms and USG findings patient admitted and planned for laparoscopic cholecystectomy. On laparoscopy after removing the adhesions, exploration done up to the porta but gallbladder could not be visualized.On postoperative day-1 patient was sent for MRCP with MRI abdomen. On MRCP gallbladder and cystic duct were not visualized. CBD was normal in caliber. Liver and pancreas were normal. Hence the diagnosis of agenesis was made. Conclusion: Agenesis of gallbladder is an unusual anomaly in which about 23% presents as biliary disease. These patient frequently undergone surgery because of misinterpreted reports of USG abdomen, ERCP and CT abdomen. So, in cases with ultrasonographic diagnosis of scleroatrophic or non-visualization or suspicious of ectopic gallbladder and absence of wall echo shadow (WES) triad or double arc shadow, when non-visualization of gallbladder is present during laparoscopy or open exploration intraoperative cholangiogram, intraoperative ultrasound and postoperative MRCP or Endoscopic ultrasound (EUS) can help in the diagnosis of agenesis or ectopic gallbladder.