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We present a rare case of aberrant left brachiocephalic vein and brachiocephalic artery, which crosses the trachea in the neck obliquely and closely related to lower border of thyroid gland. If not noticed while performing open or percutaneous dilatational tracheostomy or other neck surgeries, trauma to these vessel and subsequent hemorrhage can occur and may be fatal. Vascular compression of the airway causing obstructive symptoms can also occur due to this anomaly. In this report the case is presented along with its clinical significance.