ANEURYSM OF THE lNTERNAL JUGULAR VEIN MANIFESTING AFTER PROLONGED POSITIVE PRESSURE VENTILATION Scott A. Spiro, MD, Stephen F. Coccaro, MD, and Eugene Bogucki, MD zyxwvutsrqp zyxwvutsrqpo zyxwvutsrqp True internal jugular vein aneurysms are exceedingly rare anomalies. To date very few case reports have appeared regarding this entity. We present a case of a congenital internal jugular venous aneurysm which manifested itself after abdominal surgery and prolonged positive pressure ventilation. A theory regarding its manifestation is presented. We believe this case to be unique in its presentation. HEAD & NECK 1991;13:450452 CASEREPORT A 61-year-old black man was admitted with a large ventral incisional hernia and recurrent left inguinal hernia. He denied any previous trauma or surgery other than the inguinal hernia repair. There was no history of previous central line placement. He had a history of being briefly treated for hypertension and was a prior cigarette smoker. He denied any exposure to syphilis and reported a negative VDRL test. On physical exam his only significant findings were a soft large ventral incisional hernia which included the umbilicus and a left inguinal hernia which extended into the scrotum, both easily reducible. The patient underwent an uneventful ventral hernia repair with marlex graft patch and inguinal herniorrhaphy in the same procedure. His initial postoperative course was uneventful until day 4 when the patient complained of a lump in his left neck. Over the next 24 hours the mass (which was soft and nontender) increased in size to approximately 3 x 3 cm. It was located in the subclavian triangle of the neck and did not fluctuate in size during straining nor have any accompanying audible sounds on auscultation. There was no palpable lymphadenopathy. The patient denied any previous trauma or invasive procedures in the neck. An ultrasound of the neck was obtained. It revealed a mass with a cystic component. A computed tomographic (CT) scan of the neck (without contrast) was obtained on the next hospital day. It revealed a mass zyxwvuts Anomalies of the internal jugular vein are uncommon and infrequently encountered. Fusiform or saccular dilatation of veins has been termed phlebectasia by some investigators and aneurysm by others. Harris, in 1928, was the first to describe this condition, which he labeled congenital venous cyst.' Since then there has been relatively few reported cases of head and neck venous aneurysms. A comprehensive review in 1981 found 12 reported cases of head and neck aneurysms. Only 2 of these involved the internal jugular vein.2 We present a case of a congenital internal jugular vein aneurysm and describe its manifestation. A theory is then presented to explain its occurrence. From the Department of Surgery, St. Barnabas Medical Center, Livingston, New Jersey. zyxwvutsr zyxwvutsrqponm Address reprint requests to Dr. Spiro at the Department of Surgery, St. Barnabas Medical Center, Livingston, NJ 07039. Accepted for publication January 28, 1991 CCC 0148-6403/91/050450-03 $04.00 0 1991 John Wiley 8 Sons, Inc. 450 Aneurysm of Internal Jugular Vein HEAD & NECK September/October 1991 zyx the internal jugular vein. The proximal extent of the aneurysm was at the confluence of the internal jugular vein and subclavian vein. The external jugular vein joined the aneurysm as well. The aneurysm measured 2.5 x 3.0 cm. The nerves of the ansa cervicalis were splayed over the anterior surface of the aneurysm (see Figure 2). They were dissected free of the aneurysm and preserved. The aneurysm was resected at its base, preserving the integrity of the subclavian vein. The internal jugular vein was suture ligated distal to the aneurysm. The patient tolerated the procedure well. His postoperative recovery was uncomplicated, and he was discharged home 3 days later. At 5-month follow-up he had no further complaints and was completely asymptomatic. zyxwvutsrqpon FIGURE 1. CT scan delineates a mass in the left neck. The lesion was suggestive for an aneurysm of either the subclavian artery or vein. thought to be an aneurysm of the subclavian artery andlor vein, which was adherent t o the sternocleidomastoid muscle (Figure 1). The patient underwent a left neck exploration on the 7th postoperative day. A supraclavicular incision was used to expose the mass. Dissection revealed the mass to be a n aneurysm of FIGURE 2. Illustration depicting the anatomy found at surgery. Note the proximal extent of the aneurysm, the position of the external jugular vein, and the ansa cervicalis which was splayed over the aneurysm. Aneurysm of Internal Jugular Vein DISCUSSION The differential diagnosis of nonpulsatile soft neck masses includes arteriovenous malformations, thyroglossal duct cysts, cystic degeneration of neoplasms, retention cysts, cystic hygroma, branchial cleft cyst, pharyngocele, cavernous hemangioma, cupula inflation, laryngocele, external laryngeal diverticulum, tumors or cysts of the upper mediastinum, and venous ane u r y s m ~ . ~O-f~these only the last 5 are en- zyx zy zyxwvu FIGURE 3. Chest x-ray taken on admission revealed a soft tissue density in the left neck. This finding was identified in a retrospective review of the case. HEAD & NECK SeptemberlOctober 1991 451 hanced by maneuvers which increase intrathoracic pressure.6 The most common of which is a laryng~cele.~ Internal jugular vein aneurysms are thought to be more common on the left side of the neck in adult^.^ The workup usually includes plain x-rays, ultrasonography, and CT scan. Barium swallow and direct laryngoscopy may also be required.8 In our patient the neck mass did not fluctuate in size, so physical exam was of limited value. It was only on retrospective analysis of the patient's chest x-ray that a soft tissue density was appreciated (see Figure 3). We elected to resect the aneurysm based on 2 indications. First, thrombus had been found in the aneurysm, thereby creating a risk for embolism. Second, concerns on the part of the patient included the cosmetic appearance of his neck. Both indications for ligation and excision have been discussed by several author^.^.^,^.',^ The histologic findings vary. There may be varying degrees of thinning of the wall. Thrombus may or may not be present. The amount of connective tissue may be increased or decreased. There may also be an increase or decrease in elastic fiber^?,^,^," It has been stated that if the normal layers are all present, then aneurysm rather than phlebectasia is the more appropriate term.4 The microscopic specimen of vein from our case contained all the normal layers of anatomy; however, there was a paucity of connective tissue in the subendothelial layer suggestive of a congenital anomaly. The etiology of internal jugular vein aneurysm is only speculative. Most authors suggest a traumatic, inflammatory, or congenital etiology. If the structure of the vein is abnormal, then a congenital etiology is possible. Some authors suggest that the etiology is related to compression of the internal jugular vein by the cupula of the The cupula may extend 5 cm above the ~lavicle.~ We theorize that our patient had a congenital venous aneurysm which manifested itself only after the patient had been intubated and had positive pressure ventilation while undergoing lengthy abdominal surgery. The force of ventilation and expansion of the cupula may have either hastened the rate of growth of an exiting aneurysm or may have caused an aneurysm in a vein predisposed to aneurysm formation. In our case the internal jugular vein aneurysm became clinically significant during the postoperative recuperative period. It must be emphasized that our explanation for the etiology of an internal jugular vein aneurysm is speculative and based on a single case report. zyx zyxwvu zyxwvuts zyxwvutsrqp zyxwvuts zyxwvutsr zyxw REFERENCES 1. Harris RL. Congenital venous cyst of the mediastinum. Ann Surg 1928;88:953-956. 2. Zorn WGW, Zorn 'IT, Van Bellen B. Aneurysm of the anterior jugular vein. J Cardwvasc Surg 1981;22:546-549. 3. LaMonte SJ, Walker EA, Moran WE%.Internal jugular phlebectasia.Arch Otolaryngol 1976;102:706-708. 4. Stevens RK,Freid AM, Hood TR Jr. Ultrasonic diagnosis of jugular venous aneurysm. J Clin Ultrasound 1982;10:85-87. 5. Hughes PL, Qureshi SA, Galloway RW. Jugular venous aneurysm in children.Br J Radwl1988;61:1082- 1084. 452 Aneurysm of Internal Jugular Vein 6. Zohar Y, Ben-Tovim R, Talmi YP. Phlebectasia of the jugular system. J Cmnwmarillofm Surg 1989;17:9698. 7. Matsuba HM, Thawley, Smith PG. Internal jugular phlebectasia. Head Neck Surg 1985;7:431-433. 8. Mallik RC. A case of aneurysm of the internal jugular vein. J Luryngol Otol 1977;91:893-895. 9. Garrow E,Kirschtein M, Som ML. Internal jugular phlebectasia. Am J Surg 1964;108:380-383. 10. Schatz IJ, Fine G. Venous aneurysms. N Engl J Med 1962;266:1310- 1312. HEAD & NECK September/October 1991