Mycotic aneurysms of the internal carotid artery Case report SATOSHi SHIBUYA, M.D., SEISHI IGARASHI, M.D., TADASHI AMO, M.D., HIDEO SATO, M.D., AND TARO FUKUMITSU, M.D. Department of Neurosurgery, Shizuoka Rosai Hospital, Hamamatsu, Japan t,, The authors report a case with two mycotic aneurysms in the cavernous portion of the internal carotid artery, presumably secondary to a transient bacteremia from pneumonia. The strikingly rapid development of the aneurysms was demonstrated by angiography. Painful total ophthalmoplegia and exophthalmoswere the main clinical features. KEYWORDS 9 m y c o t i c a n e u r y s m 9 c a v e r n o u s sinus
9
totalophthalmoplegia
history of pulmonary disease or cardiac abnormalities. Examination. On admission, the patient was drowsy and complained of severe headache and chest pains. She had a moderately stiff neck, nystagmus to the left, and flaccid tendon reflexes of the extremities. Blood pressure was 140/92, pulse 96, temperature 98.8~ and white blood cell count 20,600. Lumbar puncture revealed an opening pressure of 180 mm H20. Examination of the spinal fluid showed 65 white blood cells, while sugar and protein amounts were within normal limits. Chest x-ray films Case Report showed a homogenous density increase in the A 42-year-old woman was admitted to right lower lobe. Blood cultures grew hospital on January 29, 1973, with chest pains staphylococcus, so antibiotic therapy was in the right side, after a prolonged and com- started with penicillin and chloramphenicol. On the fourth day in hospital, the patient plicated illness characterized by headache and fever. She had no previous documented complained of retro-orbital pain and tinnitus
i
NTRACRANIALmycotic aneurysms of extravascular origin are rare, 5 and a multiple incidence is particularly unusual. 7 We have been able to find reports of only seven cases of aneurysm of the intracavernous portion of the internal carotid artery secondary to infection in the cavernous sinus.1,4,1~ We are reporting a case of multiple mycotic aneurysms with angiographic evidence of rapid development in size in only 10 days. Such manifest angiographic demonstration has not been reported previously.
J. Neurosurg. / Volume 44 /January, 1976
105
S. Shibuya, et al.
Fi~. 1. Left carotid angiogram. Frontal (left) and lateral (right) projections in the initial arterial phase, demonstrating the two aneurysms of the intracavernous portion of the internal carotid artery. The upper arrow indicates the saccular aneurysm and the lower arrow the elongated cylindrical aneurysm.
in the left side, and a mild left blepharoptosis was found. Within a week the patient had developed inflammatory swelling of the left infra-auricular region. The left eyelids were edematous, the eyeball protruded and lateral ocular movements were restricted. No bruit was audible over either eye. A left carotid angiography on February 5 revealed a saccular and a cylindrical aneurysm in the intracavernous part of the internal carotid artery (Fig. 1). The carotid siphon was of a caliber less than average, suggesting the presence of arterial spasm. On the 16th hospital day, the patient had a total left ophthalmoplegia, with complete ptosis and an enlarged fixed pupil. She developed mild facial palsy on the left, and sensory impairment over the second and third divisions of the left fifth nerve. Corneal reflex on the left was also reduced. On February 15, a second left carotid arteriogram was performed (Fig. 2); the two aneurysms had increased in size over a period of 10 days. There was marked arterial narrowing of the supraclinoid portion of the carotid siphon and very poor opacification of the distal internal carotid branches, due to spasm. Operation. On February 16, we carried out ligations of the left internal and external carotid arteries in the neck, and intracranial ]06
ligation of the ipsilateral internal carotid artery above the cavernous sinus. The left cavernous sinus was ballooned, causing a depression and a flattening of the left third nerve with mild inflammation, but no abscess formation. On extradural exploration of the foramen ovale at the base of the skull, no conspicuous inflammatory foci were disclosed. Postoperative Course. Postoperatively the patient remained lethargic for a week. She then slowly improved, the protrusion of the eyeball diminished, and some ocular movement returned. The ligations of the carotid artery did not produce any recognizable neurological deficits. Postoperative fourvessel angiography demonstrated no remaining aneurysm. A year after the operation the patient was almost fully recovered, although mild limitation of left ocular movement to the left persisted. Discussion In this patient, fever and subsequent total ophthalmoplegia were the main symptoms, and a diagnosis of infective cavernous sinus syndromes was considered. In the early stages, it was difficult to determine whether the inability to move the eye and the eyelids was due to mechanical compression by the J. Neurosurg. / Volume 44 /January, 1976
Mycotic aneurysms of the internal carotid artery
FIG. 2. Repeat left carotid angiogram 10 days later, showing the strikingly rapid development of the two aneurysms (arrows).
aneurysms or to inflammatory involvement of the cavernous sinus. 12 The most probable cause for aneurysmal formation was a secondary transient bacteremia from pneumonia. It seemed likely that bacterial infection invaded the left cavernous sinus from neighboring inflammatory foci, particularly a petrosal inflammation. The carotid angiitis, of extravascular origin, might cause disintegration of the tissue until the vessel wall was weakened enough to yield to mycotic aneurysm." Another possible cause is an infective microembolus in the vasa vasorum, 1~ but our patient never showed any evidence of embolism, so this is unlikely. Hannesson and Sachs 5 stated that only organisms of relatively low virulence 8 or attenuated by antibiotic therapy 6 were capable of forming an aneurysm. They thought that such a complication might become more frequent in the future. In this case, blood cultures taken at the time of admission were positive for staphylococcus and culture from the left tympanic paracentesis revealed the same low-virulence bacterial flora. On repeat angiography? the strikingly rapid development of aneurysms was noted. Both aneurysms had increased three to five times their size in only 10 days. Such manifest J. Neurosurg. / Volume 44 / January, 1976
angiographic evidence of rapid aneurysm growth has not been reported previously. With the aneurysms expanding so rapidly, early surgical attack was indicated. Ligations of the carotid artery in the neck and intracranially were the preferred treatment, as the patient's cerebrovascular system was capable of supplying the necessary cross-flow from the contralateral carotid artery. References
1. Barker WF: Mycotic aneurysms. Ann Surg 139:84-89, 1954 2. Barr HWK, Blackwood W, Meadows SP: Intracavernous carotid aneurysms. A clinical pathological report. Brain 94:607-622, 1971 3. Cantu RC, LeMay M, Wilkinson HA: The importance of repeated angiography in the treatment of mycotic-embolic intracranial aneurysms. J Neurosurg 25:189-193, 1966 4. Devadiga KV, Mathai KV, Chandy J: Spontaneous cure of intracavernous aneurysm of the internal carotid in a 14-month-old child. Case report. J Neurosurg 30:165-168, 1969 5. Hannesson B, Sachs E Jr: Mycotic aneurysms following purulent meningitis. Report of a case with recovery and review of the literature. Acta Neurochir (Wein) 24:305-313, 1971 6. Heidelberger KP, Layton WM Jr, Fisher RG: Multiple cerebral mycotic aneurysms complicating posttraumatic pseudomonas menin]07
S. Shibuya, et al.
7.
8. 9.
10.
]08
gitis. Case report. J Neurosurg 29:631-635, 1968 King AB: Successful surgical treatment of an intracranial mycotic aneurysm complicated by a subdural hematoma. J Neurosurg 17:788-791, 1960 Molinari GF, Smith L, Goldstein MN, et al: Pathogenesis of cerebral mycotic aneurysms. Neurology (Minneap) 23:325-332, 1973 Ojemann RG, New PFJ, Fleming TC: Intracranial aneurysms associated with bacterial meningitis. Neurology (Minneap) 16:1222-1226, 1966 Ohono K, Ito U, Fukushima Y: Multiple mycotic aneurysms of internal carotid arteries caused by osteomyelitis of skull: report of a case. Bull Tokyo Med Dent Univ 20:51-58, 1973
11. Roach MR, Drake CG: Ruptured cerebral aneurysms caused by micro-organisms. N Engl J Med 273:240-244, 1965 12. Sondheimer FK, Knapp J: Angiographic findings in the Tolosa-Hunt syndrome: painful ophthalmoplegia. Radiology 106:105-112, 1973 13. Suwanwela C, Suwanwela N, Charuchinda S, et al: Intracranial mycotic aneurysms of extravascular origin. J Neurosurg 36:552-559, 1972
Address reprint requests to." Satoshi Shibuya, M.D., Department of Neurosurgery, Shizuoka Rosai Hospital, Hamamatsu, Japan.
J. Neurosurg. / Volume 44 / January, 1976
9
totalophthalmoplegia
history of pulmonary disease or cardiac abnormalities. Examination. On admission, the patient was drowsy and complained of severe headache and chest pains. She had a moderately stiff neck, nystagmus to the left, and flaccid tendon reflexes of the extremities. Blood pressure was 140/92, pulse 96, temperature 98.8~ and white blood cell count 20,600. Lumbar puncture revealed an opening pressure of 180 mm H20. Examination of the spinal fluid showed 65 white blood cells, while sugar and protein amounts were within normal limits. Chest x-ray films Case Report showed a homogenous density increase in the A 42-year-old woman was admitted to right lower lobe. Blood cultures grew hospital on January 29, 1973, with chest pains staphylococcus, so antibiotic therapy was in the right side, after a prolonged and com- started with penicillin and chloramphenicol. On the fourth day in hospital, the patient plicated illness characterized by headache and fever. She had no previous documented complained of retro-orbital pain and tinnitus
i
NTRACRANIALmycotic aneurysms of extravascular origin are rare, 5 and a multiple incidence is particularly unusual. 7 We have been able to find reports of only seven cases of aneurysm of the intracavernous portion of the internal carotid artery secondary to infection in the cavernous sinus.1,4,1~ We are reporting a case of multiple mycotic aneurysms with angiographic evidence of rapid development in size in only 10 days. Such manifest angiographic demonstration has not been reported previously.
J. Neurosurg. / Volume 44 /January, 1976
105
S. Shibuya, et al.
Fi~. 1. Left carotid angiogram. Frontal (left) and lateral (right) projections in the initial arterial phase, demonstrating the two aneurysms of the intracavernous portion of the internal carotid artery. The upper arrow indicates the saccular aneurysm and the lower arrow the elongated cylindrical aneurysm.
in the left side, and a mild left blepharoptosis was found. Within a week the patient had developed inflammatory swelling of the left infra-auricular region. The left eyelids were edematous, the eyeball protruded and lateral ocular movements were restricted. No bruit was audible over either eye. A left carotid angiography on February 5 revealed a saccular and a cylindrical aneurysm in the intracavernous part of the internal carotid artery (Fig. 1). The carotid siphon was of a caliber less than average, suggesting the presence of arterial spasm. On the 16th hospital day, the patient had a total left ophthalmoplegia, with complete ptosis and an enlarged fixed pupil. She developed mild facial palsy on the left, and sensory impairment over the second and third divisions of the left fifth nerve. Corneal reflex on the left was also reduced. On February 15, a second left carotid arteriogram was performed (Fig. 2); the two aneurysms had increased in size over a period of 10 days. There was marked arterial narrowing of the supraclinoid portion of the carotid siphon and very poor opacification of the distal internal carotid branches, due to spasm. Operation. On February 16, we carried out ligations of the left internal and external carotid arteries in the neck, and intracranial ]06
ligation of the ipsilateral internal carotid artery above the cavernous sinus. The left cavernous sinus was ballooned, causing a depression and a flattening of the left third nerve with mild inflammation, but no abscess formation. On extradural exploration of the foramen ovale at the base of the skull, no conspicuous inflammatory foci were disclosed. Postoperative Course. Postoperatively the patient remained lethargic for a week. She then slowly improved, the protrusion of the eyeball diminished, and some ocular movement returned. The ligations of the carotid artery did not produce any recognizable neurological deficits. Postoperative fourvessel angiography demonstrated no remaining aneurysm. A year after the operation the patient was almost fully recovered, although mild limitation of left ocular movement to the left persisted. Discussion In this patient, fever and subsequent total ophthalmoplegia were the main symptoms, and a diagnosis of infective cavernous sinus syndromes was considered. In the early stages, it was difficult to determine whether the inability to move the eye and the eyelids was due to mechanical compression by the J. Neurosurg. / Volume 44 /January, 1976
Mycotic aneurysms of the internal carotid artery
FIG. 2. Repeat left carotid angiogram 10 days later, showing the strikingly rapid development of the two aneurysms (arrows).
aneurysms or to inflammatory involvement of the cavernous sinus. 12 The most probable cause for aneurysmal formation was a secondary transient bacteremia from pneumonia. It seemed likely that bacterial infection invaded the left cavernous sinus from neighboring inflammatory foci, particularly a petrosal inflammation. The carotid angiitis, of extravascular origin, might cause disintegration of the tissue until the vessel wall was weakened enough to yield to mycotic aneurysm." Another possible cause is an infective microembolus in the vasa vasorum, 1~ but our patient never showed any evidence of embolism, so this is unlikely. Hannesson and Sachs 5 stated that only organisms of relatively low virulence 8 or attenuated by antibiotic therapy 6 were capable of forming an aneurysm. They thought that such a complication might become more frequent in the future. In this case, blood cultures taken at the time of admission were positive for staphylococcus and culture from the left tympanic paracentesis revealed the same low-virulence bacterial flora. On repeat angiography? the strikingly rapid development of aneurysms was noted. Both aneurysms had increased three to five times their size in only 10 days. Such manifest J. Neurosurg. / Volume 44 / January, 1976
angiographic evidence of rapid aneurysm growth has not been reported previously. With the aneurysms expanding so rapidly, early surgical attack was indicated. Ligations of the carotid artery in the neck and intracranially were the preferred treatment, as the patient's cerebrovascular system was capable of supplying the necessary cross-flow from the contralateral carotid artery. References
1. Barker WF: Mycotic aneurysms. Ann Surg 139:84-89, 1954 2. Barr HWK, Blackwood W, Meadows SP: Intracavernous carotid aneurysms. A clinical pathological report. Brain 94:607-622, 1971 3. Cantu RC, LeMay M, Wilkinson HA: The importance of repeated angiography in the treatment of mycotic-embolic intracranial aneurysms. J Neurosurg 25:189-193, 1966 4. Devadiga KV, Mathai KV, Chandy J: Spontaneous cure of intracavernous aneurysm of the internal carotid in a 14-month-old child. Case report. J Neurosurg 30:165-168, 1969 5. Hannesson B, Sachs E Jr: Mycotic aneurysms following purulent meningitis. Report of a case with recovery and review of the literature. Acta Neurochir (Wein) 24:305-313, 1971 6. Heidelberger KP, Layton WM Jr, Fisher RG: Multiple cerebral mycotic aneurysms complicating posttraumatic pseudomonas menin]07
S. Shibuya, et al.
7.
8. 9.
10.
]08
gitis. Case report. J Neurosurg 29:631-635, 1968 King AB: Successful surgical treatment of an intracranial mycotic aneurysm complicated by a subdural hematoma. J Neurosurg 17:788-791, 1960 Molinari GF, Smith L, Goldstein MN, et al: Pathogenesis of cerebral mycotic aneurysms. Neurology (Minneap) 23:325-332, 1973 Ojemann RG, New PFJ, Fleming TC: Intracranial aneurysms associated with bacterial meningitis. Neurology (Minneap) 16:1222-1226, 1966 Ohono K, Ito U, Fukushima Y: Multiple mycotic aneurysms of internal carotid arteries caused by osteomyelitis of skull: report of a case. Bull Tokyo Med Dent Univ 20:51-58, 1973
11. Roach MR, Drake CG: Ruptured cerebral aneurysms caused by micro-organisms. N Engl J Med 273:240-244, 1965 12. Sondheimer FK, Knapp J: Angiographic findings in the Tolosa-Hunt syndrome: painful ophthalmoplegia. Radiology 106:105-112, 1973 13. Suwanwela C, Suwanwela N, Charuchinda S, et al: Intracranial mycotic aneurysms of extravascular origin. J Neurosurg 36:552-559, 1972
Address reprint requests to." Satoshi Shibuya, M.D., Department of Neurosurgery, Shizuoka Rosai Hospital, Hamamatsu, Japan.
J. Neurosurg. / Volume 44 / January, 1976
