Purpose <p>A midline aberrant artery is an absolute contraindication to percutaneous dilatational tracheostomy (PDT). In this case report, we highlight a number of technical modifications that resulted in a successful PDT in a patient with a large midline aberrant artery.</p> Clinical features <p>A 72-yr-old woman with a posterior cranial fossa hematoma underwent PDT due to prolonged mechanical ventilation in the neurointensive care unit. On clinical examination, the patient had a huge, pulsatile midline neck mass. Ultrasonography (US) showed an aberrant artery that covered the entire tracheal length and deviated to the right, away from the midline, just below the cricoid cartilage at the level of the first tracheal ring. The patient’s family members were counseled, and following provision of informed consent, we planned PDT with technical modifications. After anesthesia induction, we replaced the endotracheal tube with a supraglottic airway device. We performed surface marking with US and chose a higher entry point between the first and second tracheal rings with a left anterolateral approach to the trachea. We made a 1-cm skin incision away from the midline towards the left side to aid with dilatation during the PDT procedure. We punctured the left anterolateral tracheal wall under real-time fibreoptic bronchoscopy and successfully performed PDT using a single-dilatation Ciaglia technique.</p> Conclusion <p>This report provides an anecdotal description of successful PDT in a patient with a large midline aberrant artery based on the use of US and a number of technical modifications. Nevertheless, PDT should continue to be considered contraindicated in patients with a midline aberrant artery, in whom surgical tracheostomy is the recommended technique.</p>