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Abstract Background Displaced anterior segmental bronchus and pulmonary artery is extremely rare. A keen knowledge of such variations is required in the field of pulmonary segmentectomy, for unawareness of the structural variation could lead to intra- and postoperative complications. Case presentation A 50-year-old female presented to our department with suspected lung adenocarcinoma. Preoperative 3-dimensional computed tomographic bronchography and angiography revealed anterior segmental bronchus and anterior segmental pulmonary artery variation: the anterior segmental bronchus derived from the middle lobe bronchus, accompanied by a distally distributed anterior segmental pulmonary artery branch. A right apical-posterior segmentectomy using inflation-deflation technique was performed successfully. Conclusions The keen observation and proper application of modern imaging technology and operative technique could greatly aid segmentectomy, preventing intra- and postoperative complications from happening.