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Goldenhar syndrome or Oculo-auricular Vertebral Dysplasia (OAVD) is characterized by a wide range of congenital anomalies including ocular, auricular, facial, cranial, vertebral and cardiac abnormalities. Facial and oral abnormalities especially mandibular hypoplasia, micrognathia, high arch palate and limitation of neck movements resulting from vertebral anomalies, the difficult intubation, laryngoscopy and mask ventilation were expected. Hereby, presenting the case report of a 6-year-old male child, known to have goldenhar syndrome, who underwent drainage of left sided brain abscess with external ventricular drain insertion. In view of the anticipated difficult airway and cardiac anomalies, careful preoperative evaluation, preparation and well formulated contingency plans for airway maintenance, endotracheal intubation and intraoperative haemodynamic is required to combat the perioperative anaesthetic challenges in all cases of goldenhar syndrome.