- Email: [email protected]
Carotid artery aneurysm of granulomatous origin
Carotid Artery Aneurysm of Granulomatous Origin CAP1 M. K. Neugebauer, MD, USN, MC,’ Guam, Mariana Islands LCDR 1. W. Hoyt, MD, USNR, MC,+ Guam, Mariana Islands
The incidence of extracranial carotid aneurysms is impossible to document. Most are asymptomatic, and routine postmortem examination does not include the carotid bifurcations. Brindley and Stembridge [I] in 1956 reported the incidence of aortic aneurysms as 2 to 4 per cent; half of these were syphilitic. With the advent of chemotherapy, syphilitic aneurysms have been all but eliminated. This suggests a current incidence of about 1 per cent. Beall et al [2] reported that five cases of carotid artery aneurysm were other than traumatic of 2,300 cases of aneurysm surgically treated at Baylor University College of Medicine. Extrapolation of these data to the population of the United States (more than two hundred million), together with the fact that no carotid aneurysms have been reported in patients younger than forty years of age, indicates that at any one time approximately 2,000 people in the United States have carotid artery aneurysms. This certainly accounts for the absence of large series of this entity. There were only twelve cases in a thirty year survey at Johns Hopkins Hospital [3]. Within a twenty year period, there were only five cases at the University of Pennsylvania 141. None of these was granulomatous. The etiologic factors of carotid artery aneurysm are similar to those of aneurysm formation elsewhere, that is, arteriosclerosis, syphilis, mycosis, cystic medial necrosis, and granulomatous disease.
From the Department of Surgery, United States Naval Hospital, Guam, Marfana Islands. The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or as reflecting tfte views of the Navy Department or the Naval Service at large. Present address and address for reprint requests: 1948 Port Chelsea Place, Newport Beach. California 92660. 7 Present address: Department of Thoracic Surgery, University of Alabama, Birmingham, Ahbama 35233. l
362
Arteriosclerosis accounts for 90 per cent of these unique aneurysms [5-81. There are numerous reports of tuberculous aneurysms, which almost invariably result from the inflammation of adjacent lymph nodes. The reported cases have been aortic, with fatal rupture. This entity is associated with active and/or miliary tuberculosis in a high percentage of cases [+11]. In a review of eleven cases of mycotic aneurysms, Cliff, Soulen, and Finestone [12] may have encountered one common carotid artery aneurysm caused by tuberculosis. The following report describes a case of mycotic aneurysm of granulomatous origin involving the right common carotid artery and bifurcation, treated successfully by excision and saphenous vein graft replacement. If this aneurysm is tuberculous in origin, it is a rare entity. If its origin is sarcoid, it is the first such case reported. Case Report The patient, a forty-four year old female native of Palau, Western Caroline Islands, was admitted to this hospital in January 1972 because of a mass in the right side of the neck that occasionally was tender. She first noted swelling three years prior to admission and associated it with a tooth extraction. The mass had enlarged slowly. The patient reported occasional syncope. A purified protein derivative (PPD) skin test had been strongly positive, and she received isoniaxid for six weeks prior to referral, without reduction in the size of the mass. Physical examination revealed no abnormalities except for a pulsatile mass measuring 3 by 6 cm over the right carotid bifurcation. Serologic study was noncontributory. An extensive radiologic survey failed to show any evidence of active or old tuberculosis. Carotid arteriography revealed an extensive aneurysm involving the distal two thirds of the right common carotid artery a& bi-
The American Journal 01 Surgery
Granulomatous Carotid Aneurysm
Figure 7. Anteropostet’ior and laterat carotid arteriograms showing extensive aneurysm of the d/&al common carotid artery and bifurcation.
furcation. (Figure 1.) With local anesthesia and normal blood pressure, the common carotid was occluded for eight minutes. The patient had no loss of sensorium or paresis of the contralateral side. General endotracheal anesthesia and hypertension were then induced. Hypercarbia was not utilized. The aneurysm, together with densely adherent lymph nodes, was excised (Figure 2) and replaced with a reversed segment of saphenous vein without the use of an internal or external shunt. Total occlusion time was twenty-four minutes. Back pressure from the internal carotid artery was obviously elevated. There were no neurologic sequelae in the postoperative period and recovery was uneventful. An arteriogram six months postoperatively revealed a patent graft without dilatation. (Figure 3.) Histologic evidence strongly indicates that a granulomatous process was responsible for this-carotid artery
Figure 3. Carotid arteriogram six months postopera tlvely showing patent vein graft wIthout dilatation. Arrows denote extent of vein graft replacement.
vohmo 180, soptombu 1975
i
^
.I~*
‘1
.t.*t.,
..‘_J
Figure 2. Gross specimen showing extent and size of the rtght carotid artery aneurysm.
Neugebauer and Hoyt
Figure 4. Medium-power view of lymph node immediately adjacent to carotid aneurysm showing muitipie well formed granuiomas with occasional giant ceiis. No necrosis or caseation is noted. (Hematoxyiin and eosin stain; original magnification approximately X 100.)
Figure 5. Low-power view of entire vessel wail from carotid aneurysm showing subtntimai hemorrhage with organization and m&pie foci of perivascuiar inflammatory infiitrate in the media and adventitiai layers. (Hematoxy/in and eosin stain; original magnification approximately x 5.)
aneurysm. Although tuberculosis is most likely the etiologic agent, sarcoid cannot be ruled out because of the noncaseating granulomas noted in the lymph nodes surrounding the aneurysm. (Figure 4.) Multinucleated giant cells were noted in the wall of the aneurysm, but they were not typical of the giant cells associated with tuberculosis. Unfortunately, the specimen was placed in formalin prior to culture for acid-fast bacteria, but a scrupulous search of multiple slides of the aneurysm and lymph nodes failed to reveal acid-fast organisms. (Figures 5 and 6.)
We used this last test because of the patient’s young age and the complete lack of arteriosclerotic findings elsewhere. The many surgical approaches to the treatment of carotid artery aneurysms have been reviewed by Kirby, Johnson, and Donald [4] and Thompson and Austin [15]. These approaches include: (1) proximal ligation; (2) proximal and distal ligation with or without excision of the aneurysm; (3) wrapping with fascia lata; (4) excision with restoration of circulation. With the advances in current vascular surgical technics, it seems that excision and restoration of circulation are mandatory except when technically impossible.
Comments The presence of a carotid artery aneurysm must be considered in the differential diagnosis of any mass in the neck, especially one that is pulsatile. This entity must be differentiated from abscess, lymphadenopathy, carotid body tumor, branchial cleft cyst, and tortuous carotid artery [13]. Arteriography is mandatory in diagnosis and is essential in planning the operative procedure and approach ]2,5,71. Operation in the carotid arteries is unique because of the inability of the brain to tolerate even short periods of ischemia. Only 40 to 70 per cent of patients can undergo complete occlusion of the common carotid artery without neurologic deficits [14]. Although various tests have been devised to identify those patients who can tolerate carotid occlusion, no test is reliable, as evidenced by the many different approaches to carotid operation. These tests include: (1) cross-compression angiography; (2) manual compression preoperatively (Matas test); (3) occlusion using local anesthesia.
364
Figure 5. Medium-power view of infiammatory infiitrate in the medial kyers showing predomkant iymphocytk popuktkn and muttinucieated giant ceits. (Hematoxyiin and eodn stain; original magnifkation approxbnateiy X 100.)
The American Journal of Sqery
Granulomatous Carotid Aneurysm
Prevention of ischemic changes in the brain during reconstruction of the carotid artery has been approached by a variety of methods. The use of an internal shunt [16] during reconstruction reduces total occlusion time but lengthens the operative procedure and involves a reduced blood flow to the brain during the period of anastomosis. Construction of an external bypass shunt prior to excision also lengthens the procedure [2,5]. The external shunt precludes a significant period of ischemia but may predispose to subsequent thrombosis. The induction of hypothermia is known to increase the ability of the brain to tolerate ischemia; however, the procedure itself is not without risks, which may outweigh the benefits obtained. In patients such as the one reported herein, when adequate collateral circulation is demonstrated, simple occlusion with excision and vein graft replacement appears to carry the least risk. The various technics reported for reconstruction of the cervical carotid artery include primary end to end anastomosis and utilization of the external carotid artery, Dacron@ prostheses, homografts, and autogenous vein grafts [ 7,8,16,17]. Autogenous saphenous vein grafts have been reported only infrequently but seem to be the ideal replacement. They are immediately available, of appropriate size, and autogenous, and they do not serve as a foreign body. Reconstruction is best individualized depending on the extent of the aneurysm and availability of materials, as well as the experience and preference of the surgeon. Summary A native of the Western Caroline Islands presented with a granulomatous aneurysm of the right common carotid artery measuring 7 to 8 cm, which was resected and replaced with a reversed segment of saphenous vein. Adequacy of the collateral circulation to the brain was established by occlusion of the common carotid artery with local anesthesia. This was followed by definitive operation with general endotracheal anesthesia and induced hypertension. Although tuberculosis was
Vofume 130.60~amber
1975
the most likely etiologic agent, sarcoid could not be ruled out. Granulomatous aneurysms of the common carotid are extremely rare, and if this case was sarcoid in origin, it is the first such case reported. Only one other similar aneurysm could be found in the literature. Of the various methods of reconstruction of the common carotid artery reported, autogenous reversed saphenous vein is recommended strongly. References 1. Brindley P, Stembridge VA: Aneurysms of the aorta, a clinicoaatholoaicstudv of 369 necroosv . r cases. Am J Pafhol 32: 67, 19-56. 2. Beall AC, Crawford ES, Cooley DA, DeBakey ME: Extracranial aneurysms of the carotid artery. Postgrad Med 32: 93, 1962. 3. Reid MR: Aneurysms in the Johns Hopkins Hospital: all cases treated in the surgical service from the opening of the hospital to January, 1922. Arch Surg 12: 1, 1946. 4. Kirby CK, Johnson J, Donald JG: Aneurysm of the common carotid artery. Ann Surg 130: 913, 1949. 5. Raphael HA, Bernatz PE, Spittell JA, Ellis FH: Cervical carotid aneurysms: treatment by excision and restoration of arterial continuity. AmJ Surg 105: 771, 1963. 6. Spencer FC: Aneurysm of the common carotid artery treated with excision and primary anastomosis. Ann Surg 145: 254, 1957. 7. Wilson JR, Jordan PH Jr: Excision of internal carotid artery aneurysm: restitution of continuity by substitution of external for internal carotid artery. Ann Surg 154: 45, 1961. 6. Barnes WT. Jacoby GE: Aneurysm of the common carotid artery due to cystic medial necrosis treated with excision and graft. Ann surg 155: 62, 1962. 9. Stiefel JW: Rupture of a tuberculous aneurysm of the aorta. Arch Pathol65: 506, 1956. 10. German JL. Green CL: Fatal rupture of a tuberculous aortic aneurysm. Ann Intern Med 45: 496.1956. 11. DeProphetis N, Arm&age HV, Triboletti ED: Rupture of a tuberculous aottic aneurysm into the lung. Ann Surg 150: 1046, 1959. 12. Cliff MM, Soulen RL, Finestone AJ: Mycotic aneurysms, a challenge and a clue. Arch Intern Med 126: 977, 1970. 13. Deterling RA: Tortuous right common carotid artery simulating an aneurysm. Angiology 3: 463, 1952. 14. Brachett CE: Complications of carotid artery ligation in the neck. J Neurosurg 10: 91, 1953. 15. Thompson JE, Austin DJ: Surgical management of cervical carotid aneurysms. Arch Surg 74: 60, 1957. 16. Eiseman B, Paton BC, Hogshead H: Use of an internal polyethylene shunt during resection of a carotid artery aneurysm.Am J Surg 102: 702. 1961. 17. Cifarelli F: Bilateral internal carotid artery aneurysms resected and replaced with prostheses. Arch Surg 102: 74, 1971.
365
The incidence of extracranial carotid aneurysms is impossible to document. Most are asymptomatic, and routine postmortem examination does not include the carotid bifurcations. Brindley and Stembridge [I] in 1956 reported the incidence of aortic aneurysms as 2 to 4 per cent; half of these were syphilitic. With the advent of chemotherapy, syphilitic aneurysms have been all but eliminated. This suggests a current incidence of about 1 per cent. Beall et al [2] reported that five cases of carotid artery aneurysm were other than traumatic of 2,300 cases of aneurysm surgically treated at Baylor University College of Medicine. Extrapolation of these data to the population of the United States (more than two hundred million), together with the fact that no carotid aneurysms have been reported in patients younger than forty years of age, indicates that at any one time approximately 2,000 people in the United States have carotid artery aneurysms. This certainly accounts for the absence of large series of this entity. There were only twelve cases in a thirty year survey at Johns Hopkins Hospital [3]. Within a twenty year period, there were only five cases at the University of Pennsylvania 141. None of these was granulomatous. The etiologic factors of carotid artery aneurysm are similar to those of aneurysm formation elsewhere, that is, arteriosclerosis, syphilis, mycosis, cystic medial necrosis, and granulomatous disease.
From the Department of Surgery, United States Naval Hospital, Guam, Marfana Islands. The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or as reflecting tfte views of the Navy Department or the Naval Service at large. Present address and address for reprint requests: 1948 Port Chelsea Place, Newport Beach. California 92660. 7 Present address: Department of Thoracic Surgery, University of Alabama, Birmingham, Ahbama 35233. l
362
Arteriosclerosis accounts for 90 per cent of these unique aneurysms [5-81. There are numerous reports of tuberculous aneurysms, which almost invariably result from the inflammation of adjacent lymph nodes. The reported cases have been aortic, with fatal rupture. This entity is associated with active and/or miliary tuberculosis in a high percentage of cases [+11]. In a review of eleven cases of mycotic aneurysms, Cliff, Soulen, and Finestone [12] may have encountered one common carotid artery aneurysm caused by tuberculosis. The following report describes a case of mycotic aneurysm of granulomatous origin involving the right common carotid artery and bifurcation, treated successfully by excision and saphenous vein graft replacement. If this aneurysm is tuberculous in origin, it is a rare entity. If its origin is sarcoid, it is the first such case reported. Case Report The patient, a forty-four year old female native of Palau, Western Caroline Islands, was admitted to this hospital in January 1972 because of a mass in the right side of the neck that occasionally was tender. She first noted swelling three years prior to admission and associated it with a tooth extraction. The mass had enlarged slowly. The patient reported occasional syncope. A purified protein derivative (PPD) skin test had been strongly positive, and she received isoniaxid for six weeks prior to referral, without reduction in the size of the mass. Physical examination revealed no abnormalities except for a pulsatile mass measuring 3 by 6 cm over the right carotid bifurcation. Serologic study was noncontributory. An extensive radiologic survey failed to show any evidence of active or old tuberculosis. Carotid arteriography revealed an extensive aneurysm involving the distal two thirds of the right common carotid artery a& bi-
The American Journal 01 Surgery
Granulomatous Carotid Aneurysm
Figure 7. Anteropostet’ior and laterat carotid arteriograms showing extensive aneurysm of the d/&al common carotid artery and bifurcation.
furcation. (Figure 1.) With local anesthesia and normal blood pressure, the common carotid was occluded for eight minutes. The patient had no loss of sensorium or paresis of the contralateral side. General endotracheal anesthesia and hypertension were then induced. Hypercarbia was not utilized. The aneurysm, together with densely adherent lymph nodes, was excised (Figure 2) and replaced with a reversed segment of saphenous vein without the use of an internal or external shunt. Total occlusion time was twenty-four minutes. Back pressure from the internal carotid artery was obviously elevated. There were no neurologic sequelae in the postoperative period and recovery was uneventful. An arteriogram six months postoperatively revealed a patent graft without dilatation. (Figure 3.) Histologic evidence strongly indicates that a granulomatous process was responsible for this-carotid artery
Figure 3. Carotid arteriogram six months postopera tlvely showing patent vein graft wIthout dilatation. Arrows denote extent of vein graft replacement.
vohmo 180, soptombu 1975
i
^
.I~*
‘1
.t.*t.,
..‘_J
Figure 2. Gross specimen showing extent and size of the rtght carotid artery aneurysm.
Neugebauer and Hoyt
Figure 4. Medium-power view of lymph node immediately adjacent to carotid aneurysm showing muitipie well formed granuiomas with occasional giant ceiis. No necrosis or caseation is noted. (Hematoxyiin and eosin stain; original magnification approximately X 100.)
Figure 5. Low-power view of entire vessel wail from carotid aneurysm showing subtntimai hemorrhage with organization and m&pie foci of perivascuiar inflammatory infiitrate in the media and adventitiai layers. (Hematoxy/in and eosin stain; original magnification approximately x 5.)
aneurysm. Although tuberculosis is most likely the etiologic agent, sarcoid cannot be ruled out because of the noncaseating granulomas noted in the lymph nodes surrounding the aneurysm. (Figure 4.) Multinucleated giant cells were noted in the wall of the aneurysm, but they were not typical of the giant cells associated with tuberculosis. Unfortunately, the specimen was placed in formalin prior to culture for acid-fast bacteria, but a scrupulous search of multiple slides of the aneurysm and lymph nodes failed to reveal acid-fast organisms. (Figures 5 and 6.)
We used this last test because of the patient’s young age and the complete lack of arteriosclerotic findings elsewhere. The many surgical approaches to the treatment of carotid artery aneurysms have been reviewed by Kirby, Johnson, and Donald [4] and Thompson and Austin [15]. These approaches include: (1) proximal ligation; (2) proximal and distal ligation with or without excision of the aneurysm; (3) wrapping with fascia lata; (4) excision with restoration of circulation. With the advances in current vascular surgical technics, it seems that excision and restoration of circulation are mandatory except when technically impossible.
Comments The presence of a carotid artery aneurysm must be considered in the differential diagnosis of any mass in the neck, especially one that is pulsatile. This entity must be differentiated from abscess, lymphadenopathy, carotid body tumor, branchial cleft cyst, and tortuous carotid artery [13]. Arteriography is mandatory in diagnosis and is essential in planning the operative procedure and approach ]2,5,71. Operation in the carotid arteries is unique because of the inability of the brain to tolerate even short periods of ischemia. Only 40 to 70 per cent of patients can undergo complete occlusion of the common carotid artery without neurologic deficits [14]. Although various tests have been devised to identify those patients who can tolerate carotid occlusion, no test is reliable, as evidenced by the many different approaches to carotid operation. These tests include: (1) cross-compression angiography; (2) manual compression preoperatively (Matas test); (3) occlusion using local anesthesia.
364
Figure 5. Medium-power view of infiammatory infiitrate in the medial kyers showing predomkant iymphocytk popuktkn and muttinucieated giant ceits. (Hematoxyiin and eodn stain; original magnifkation approxbnateiy X 100.)
The American Journal of Sqery
Granulomatous Carotid Aneurysm
Prevention of ischemic changes in the brain during reconstruction of the carotid artery has been approached by a variety of methods. The use of an internal shunt [16] during reconstruction reduces total occlusion time but lengthens the operative procedure and involves a reduced blood flow to the brain during the period of anastomosis. Construction of an external bypass shunt prior to excision also lengthens the procedure [2,5]. The external shunt precludes a significant period of ischemia but may predispose to subsequent thrombosis. The induction of hypothermia is known to increase the ability of the brain to tolerate ischemia; however, the procedure itself is not without risks, which may outweigh the benefits obtained. In patients such as the one reported herein, when adequate collateral circulation is demonstrated, simple occlusion with excision and vein graft replacement appears to carry the least risk. The various technics reported for reconstruction of the cervical carotid artery include primary end to end anastomosis and utilization of the external carotid artery, Dacron@ prostheses, homografts, and autogenous vein grafts [ 7,8,16,17]. Autogenous saphenous vein grafts have been reported only infrequently but seem to be the ideal replacement. They are immediately available, of appropriate size, and autogenous, and they do not serve as a foreign body. Reconstruction is best individualized depending on the extent of the aneurysm and availability of materials, as well as the experience and preference of the surgeon. Summary A native of the Western Caroline Islands presented with a granulomatous aneurysm of the right common carotid artery measuring 7 to 8 cm, which was resected and replaced with a reversed segment of saphenous vein. Adequacy of the collateral circulation to the brain was established by occlusion of the common carotid artery with local anesthesia. This was followed by definitive operation with general endotracheal anesthesia and induced hypertension. Although tuberculosis was
Vofume 130.60~amber
1975
the most likely etiologic agent, sarcoid could not be ruled out. Granulomatous aneurysms of the common carotid are extremely rare, and if this case was sarcoid in origin, it is the first such case reported. Only one other similar aneurysm could be found in the literature. Of the various methods of reconstruction of the common carotid artery reported, autogenous reversed saphenous vein is recommended strongly. References 1. Brindley P, Stembridge VA: Aneurysms of the aorta, a clinicoaatholoaicstudv of 369 necroosv . r cases. Am J Pafhol 32: 67, 19-56. 2. Beall AC, Crawford ES, Cooley DA, DeBakey ME: Extracranial aneurysms of the carotid artery. Postgrad Med 32: 93, 1962. 3. Reid MR: Aneurysms in the Johns Hopkins Hospital: all cases treated in the surgical service from the opening of the hospital to January, 1922. Arch Surg 12: 1, 1946. 4. Kirby CK, Johnson J, Donald JG: Aneurysm of the common carotid artery. Ann Surg 130: 913, 1949. 5. Raphael HA, Bernatz PE, Spittell JA, Ellis FH: Cervical carotid aneurysms: treatment by excision and restoration of arterial continuity. AmJ Surg 105: 771, 1963. 6. Spencer FC: Aneurysm of the common carotid artery treated with excision and primary anastomosis. Ann Surg 145: 254, 1957. 7. Wilson JR, Jordan PH Jr: Excision of internal carotid artery aneurysm: restitution of continuity by substitution of external for internal carotid artery. Ann Surg 154: 45, 1961. 6. Barnes WT. Jacoby GE: Aneurysm of the common carotid artery due to cystic medial necrosis treated with excision and graft. Ann surg 155: 62, 1962. 9. Stiefel JW: Rupture of a tuberculous aneurysm of the aorta. Arch Pathol65: 506, 1956. 10. German JL. Green CL: Fatal rupture of a tuberculous aortic aneurysm. Ann Intern Med 45: 496.1956. 11. DeProphetis N, Arm&age HV, Triboletti ED: Rupture of a tuberculous aottic aneurysm into the lung. Ann Surg 150: 1046, 1959. 12. Cliff MM, Soulen RL, Finestone AJ: Mycotic aneurysms, a challenge and a clue. Arch Intern Med 126: 977, 1970. 13. Deterling RA: Tortuous right common carotid artery simulating an aneurysm. Angiology 3: 463, 1952. 14. Brachett CE: Complications of carotid artery ligation in the neck. J Neurosurg 10: 91, 1953. 15. Thompson JE, Austin DJ: Surgical management of cervical carotid aneurysms. Arch Surg 74: 60, 1957. 16. Eiseman B, Paton BC, Hogshead H: Use of an internal polyethylene shunt during resection of a carotid artery aneurysm.Am J Surg 102: 702. 1961. 17. Cifarelli F: Bilateral internal carotid artery aneurysms resected and replaced with prostheses. Arch Surg 102: 74, 1971.
365