Arterial variations in upper limbs are often reported commonly. Superficial arterial variations accounting for 4.2% of all arterial variations are hazardous during any invasive procedures of the upper limb, from routine intravenous injections to surgeries. Arterial variations are usually associated with inverted or absent palmaris longus. Palmaris profundus, a rare anomalous variation of palmaris longus has been reported in carpal tunnel syndrome as its tendon was associated with median nerve in the carpal tunnel. The authors reported a unique variation in the upper limb arterial pattern—the presence of bilateral superficial brachioulnar artery associated with unilateral palmaris profundus muscle and an abnormal radicle of musculocutaneous nerve to the median nerve in the left side.