Unilateral lung agenesis associated with congenital heart disease is very rare and the consequent malposition of the heart and mediastinal structures pose a technical challenge for definitive surgery. We describe a case of a three-month-old infant with right lung agenesis and a large ventricular septal defect who underwent successful surgical repair. The case highlights the importance of preoperative anatomic assessment and intraoperative adaption when presented with complex anatomy.
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