ACTA NEUROCHIRURGICA

Acta Neurochirurgica 45, 259--275 (1979)

9 by Springer-Verlag 1979

Regional Neurological Centre Newcastle General Hospital, Newcastle upon Tyne, England

A n U n u s u a l Case o f M u l t i p l e Intracranial A n e u r y s m s By

R. P. Sengupta and J. Hankinson With 8 Figures

Summary A 49-year-old female patient with six intracranial aneurysms, all of which were treated successfully by direct surgery, is reported. She had a major subarachnoid haemorrhage 10 years ago. Because of difficulty in identifying the offending aneurysm she was not surgically treated at that time. The natural progression of intracranial aneutysms, the problem of identifying the lesion in a patient with multiple aneurysms, and an example of excellent recovery of visual function following surgical treatment are discussed.

With the improved surgical and anaesthetic techniques of recent years, more and more patients with multiple aneurysms are being treated by direct obliteration of all accessible lesions. The maximum number of aneurysms treated by direct surgery in any one patient is seldom more than two or three 4, 5. Mount and Brisbane 2 have mentioned a case in which five aneurysms were treated, but the details of treatment were not given. This paper reports a patient who was found to have six intracranial aneurysms. All of these were treated successfully by direct surgery. Apart from the large number of aneurysms, this patient showed an unusual sequence of presenting symptoms, an initial subarachnoid haemorrhage followed 10 years later by visual failure. The case also illustrates the natural progression of intracranial aneurysms, the problem of identifying the offending lesion in a patient with multiple aneurysms, and an example of excellent recovery of visual function following surgical treatment.

Case Report Mrs. E. G., a 49-year-old housewife, was admitted to the Regional Neurological Centre of the Newcastle General Hospital on 10 June 0001-6268/79/0045/0259/$ 03.40

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1976 for investigation of intermittent headache and failing vision in her right eye. Her troubles had started 10 years previously. On 4 March 1966 she developed a mild frontal headache, not severe, but enough to keep her awake all night. The next day she developed a severe occipital and frontal headache with neck stiffness lasting for two hours. The following day she again had severe headache, neck stiffness, and photophobia. These persisted at the time of her admission to the local hospital on 7 March. Four years before this admission she had had a migrainous type of attack consisting of pain behind the right eye and flashing of light in front of both eyes. On admission to the local hospital she was found to be fully alert, with normal vital functions. Her blood pressure was 130/90mmHg. There was severe neck stiffness. No abnormal neurological findings were detected. A lumbar puncture confirmed subarachnoid Memo> rhage, and she was transferred to the Neurosurgical Unit on 9 March. Bilateral carotid angiography was performed on the next day. This showed multiple aneurysms on both sides of the carotid circulation (Fig. 1). On the right side there was a large internal carotid aneurysm close to the bifurcation, a large anterior communicating aneurysm, a large aneurysm arising from the middle cerebral trifurcation, and possibly a fourth middle cerebral aneurysm projecting more laterally. On the left side there was a bilocular aneurysm arising from the bifurcation of the internal carotid artery, and a small aneurysmal dilatation at the point of origin of the posterior communicating artery. There was no spasm or displacement of vessels to identify the offending aneurysm. In view of this difficulty of localization, it was felt that surgical intervention was contro-indicated and she was returned to the local hospital. In fact she continued to do well, apart from having intermittent frontal headache, until December 1975, when she had an episode of severe pain behind the right eye with blurring of vision. This was followed by generalized headache. She had no neurological deficit. The CSF was clear on lumbar puncture, but the cell count showed 75 lymphocytes per mm a and a protein of 0.7 g/1. She recovered from this episode uneventfully. On 8 June 1976 she was readmitted with a five day history of severe pain behind the right eye, mild neck stiffness, and loss of vision in the right eye. Apart from a blind right eye, she again showed no abnormal neurological deficit, and her blood pressure was 160/90 mm Hg. Because of this loss of vision and intermittent headache she was referred again to the Neurosurgical Unit. There she was found to be co-operative and alert; visual acuity in the left eye was normal. The vision in the right eye had improved to N 10. Examination of general and neurological systems gave a normal

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response. Bilateral carotid angiography was performed on 1l June. The findings were as follows: (Fig. 2) right carotid angiogram: multiple saccular aneurysms still demonstrable on the origin of the right posterior communicating artery, the anterior communicating artery, and the trifurcation of the right middle cerebral artery. The sac of the posterior communicating aneurysm was definitely larger than in 1966, as was the main aneurysm of the right middle cerebral artery. Left carotid angiogram: there were two aneurysms, one at the region of the origin of the left posterior communicating artery and another at the terminal bifurcation of the left internal carotid artery. These aneurysms were both larger than at the previous examination 10 years ago. Angiographicallly there was no evidence to suggest which of the two aneurysms, the internal carotid or the anterior communicating, was responsible for her loss of vision in the right eye, since the patient's internal carotid aneurysm was projecting directly laterally away from the optic nerve and the anterior communicating aneurysm was projecting to the left. In spite of these difficulties in localization we felt that the right optic nerve should be explored and the offending aneurysm dealt with to relieve compression of the optic nerve. First operation: Clipping of the right internal carotid aneurysm. On 16 June a right fronto-temporal craniotomy was performed. After retraction of the frontal lobe, the optic nerve was found to be arched upwards, stretched and flattened by the dome of a large aneurysm lying underneath it. The nerve was very pale, and the sac of the aneurysm was found to be firm in consistency. The identity of the aneurysm remained obscure. The sac was so large that exploration of the internal carotid artery was not yet possible. As a considerable part of the sac lay medial to the optic nerve, the initial impression was that it could not be the internal carotid aneurysm that was causing compression of the nerve. So the alternative was the anterior communicating aneurysm. However, exploration of the anterior cerebral artery up to the point of origin of the anterior communicating artery revealed no aneurysmal sac that could compress the right optic nerve. We decided to decompress the optic nerve by reducing the size of the offending sac with bipolar coagulation. With slow and gentle manoeuvres, under hypotension at the 70 mm Hg systolic level, we succeeded in reducing the size by one third, following which the whole of the right optic nerve became free. With the reduction in the size of the sac, it was also possible now to delineate the anatomy more clearly. The aneurysm in question was in fact arising from the internal carotid artery, which 17 Ac~aNeurochirurgica, Vol. 45, Fasc. 8--4

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Fig. 1 a. Right Carotid Angiogram--March 1966. (Upper) Lateral projection shows multiple superimposed aneurysms. (Lower) Oblique Towne's view shows origins of aneurysms from internal carotid, middle cerebral and anterior communicating arteries. Arrow points to the location of a second middle cerebral aneuysm

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Fig. 1 b. Left Carotid Angiogram--March 1966. (Upper) Lateral projection shows small aneurysmal dilatation at the point of origin of the posterior communicating artery, and a bilocular aneurysm arising from the bifurcation of internal carotid artery (arrow). (Lower) Towne's view--developing aneurysms not clearly visible

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Fig. 2 a. Right Carotid Angiogram--June i976. Laterai (upper), Towne's (lower left) and oblique Towne's (lower right) show changes in the size of the aneurysms. The internal carotid and anterior communicating aneurysms are slightly smaller and the proximal middle cerebral aneurysm is bigger. Note that the distal middle cerebral aneurysm is overlapped by the proximal aneurysm and not visible. The arrow points to the displacement of the internal carotid artery medially

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Fig. 2 b. Left Carotid Angiogram--June 1976. Similar views as in Fig. 2 a show enlargement of internal carotid bifurcation aneurysm. The aneurysmal dilatation seen 10 years previously has now developed into a larger aneurysm

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Fig. 3. 1st Post-Operative Right Carotid Angiogram--23 June 1976. LaterM (upper) and Towne's views show total obliteration of the internal carotid aneurysm

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Fig. 4. 2nd Post-Operative Right Carotid Angiogram--16 July 1976. Lateral (upper), Towne's (lower left) and oblique Towne's (lower right) views show obliteration of internal carotid and anterior communicating aneurysm. The clip on the middle cerebral aneurysm is not misplaced. In fact it is occluding the distal middle cerebral aneurysm

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Fig. 5 a

Fig. 5 b

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had been pushed considerably medially and downwards, displacing the optic nerve laterally (Fig. 8). Once the anatomy had become clear, the neck of the aneurysm was isolated without difficulty, and a spring clip was applied across it. Because of the prolonged dissection, we decided to leave the other aneurysms alone for the time being. The patient recovered uneventfully, but the vision in her right eye was further diminished. She had no other deficit. A check anglo-

Fig. 5 c Fig. 5. 3rd Post-Operative Carotid Angiogram--6 August 1976. a) Right side: Towne's view shows total obliteration of all four aneurysms in the right carotid system, b) Left side: lateral view shows filling of the bifurcation and posterior communicating aneurysms, c) Left side: Towne's view shows partial obliteration of posterior communicating aneurysm (arrow)

gram on 23 June 1976 showed that the aneurysm had been successfully clipped (Fig. 3). With this aneurysm satisfactorily obliterated, we planned the obliteration of her other aneurysm in stages. Second operation: Clipping of the anterior communicating and right middle cerebral aneurysms. On 5 July the right frontal craniotomy was re-opened. After retraction of the frontal lobe, the previously clipped internal carotid aneurysm was found to be thrombosed. The optic nerve was lying free. The anterior communicating aneurysm was approached through the gyrus rectus. After exposure of the anterior communicating region, the bilocular aneurysm projecting to the left was dissected at its neck and obliterated with a spring clip without difficulty. The middle cerebral aneurysm

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was then approached through the Sylvian fissure. As the fissure was opened, a small aneurysm with a broad base at the point of middle cerebral bifurcation was encountered. Although this aneurysm was much smaller and more distal than the one visible on the angiograms, we considered it to be the aneurysm in question, and clipped it at the neck. The patient recovered from this operation uneventfully and without deficit. A check angiogram on 16 July showed successful obliteration of the anterior communicating and a previously

Fig. 6 a unrecognized middle cerebral aneurysm. The larger and more proximal middle cerebral aneurysm still filled as before (Fig. 4). Third operation: Clipping of the right middle cerebral, left posterior communicating, and left carotid bifurcation aneurysms. On 26 July a bifrontal coronal scalp flap was elevated, incorporating the previously turned flap on the right side. A left fronto-temporal osteoplastic craniotomy was then performed. The posterior communicating aneurysm was found to have a wide base and to be projecting postero-laterally. A spring clip was applied. By following the internal carotid artery distally, the bifurcation point was reached, and the origins of the anterior cerebral and middle cerebral arteries were seen. The aneurysm projecting ba&wards was dissected at the neck, and a spring clip was applied. The right sided craniotomy was then re-opened. By separating the Sylvian fissure, a previously placed clip on the distal middle cerebral aneurysm was seen. The middle

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Fig. 6 b Fig. 6. 4th Post-Operative Left Carotid Angiogram--12 October 1976. a) lateral, b) Towne's (upper) and b) oblique Towne's (lower) views show total obliteration of the bifurcation aneurysm. A small part of this posterior communicating aneurysm is still filling

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cerebral artery was then followed proximally. Near the point of trifurcation the dome of the large aneurysm came into view. It was almost spherical in shape and obscured the origin of the middle cerebral branches. With bipolar coagulation and systemic hypotension, the size of the aneurysm was reduced, and it became possible to locate the he& and apply a spring clip. The aneurysm was then aspirated to prevent any slipping of the clip. The patient

recovered well without any deficit. A check angiogram on 6 August revealed that the right middle cerebral aneurysm had been obliterated, the left posterior communicating aneurysm had been only partially obliterated, and that the clip on the bifurcation aneurysm was not in the correct position (Fig. 5). After full discussion with the patient and her husband she was re-admitted on 28 September for further obliteration of the left-sided aneurysms. Fourth operation: Clipping of the left bifurcation and posterior communicating aneurysms. On 4 October the left sided craniotomy was re-opened. The clip from the bifurcation aneurysm was found to be lying free and was removed. The neck of the aneurysm was dissected more clearly, and another clip was applied to its neck. The

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clip on the posterior communicating artery aneurysm was thought to be occluding part of the aneurysm and was firmly adherent to it so, without removal of this first clip, a second clip was applied across the neck. The base of the aneurysm was so large that it was difficult to narrow the neck, even with bipolar coagulation. For fear of producing a tear at the base, we decided to keep dissection to a minimum. The patient recovered from this operation uneventfully. The last check angiogram on 12 October showed successful obliteration of the bifurcation aneurysm and an almost total obliteration of the posterior communicating one (Fig. 6). We were satisfied that no further surgical procedure was warranted.

Fig. 8. Diagramatic sketch at operation. This shows that the large aneurysm of the internal carotid has pushed the artery medially to the optic nerve As the posterior circulation in this patient was not studied by vertebral angiography, it is not possible to exclude other aneurysms in the vertebro-basilar system. Since we have isolated and obliterated the offending lesion, we felt it would be unwise to make a direct surgical attack on any intact aneurysm in the posterior circulation even if one were detected by vertebral angiography. The patient was finally discharged home on 15 October. She was given Phenytoin 100 mg three times a day as a prophylactic anticonvulsant. At the follow-up clinic on 17 February 1977 she looked well (Fig. 7). She had no complaints. Her visual acuity showed marked improvement to right 6/6 and left 6/5. The visual field on the right side remained constricted.

Discussion Direct surgical obliteration of six aneurysms (including three large ones) by four intracranial procedures and five sessions of

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carotid angiography in a period of four months bears testimony to the endurance of a patient in modern times with improved surgical and anaesthetic techniques. The most gratifying outcome, however, was the dramatic improvement in vision in the right eye. This patient presents several inteesting features. Contrary to the accepted view of recurring haemorrhage from a ruptured aneurysm 5, she remained well for 10 years until she developed visual symptoms. However, the occasional severe headaches during this period may indicate minor bleeds into the false sac caused by thrombosed clot within the aneurysm. These could have increased the overall size of the aneurysm without causing a haemorrhage into the subarachnoid space. This case also shows the natural progression of intracranial aneurysms. In 10 years all the aneurysms, whether intact or otherwise, showed some increase in size. The left posterior communicating aneurysm, which appeared merely as an infundibular dilatation in 1966, had blossomed into a full blown aneurym by 1976. Identification of the offending lesion proved difficult both in 1966 and in 1976, albeit for different reasons. In 1966 the patient had no neurological deficit, and carotid angiograms showed no localizing spasm or haematoma to identify it. In the light of modern knowledge 6, several points from the patient's clinical picture and angiograms can be detected which could have identified the offending lesion. Firstly, the patient complained of pain behind the right eye before the haemorrhage and she also gave a past history of migrainous headaches on the right side for four years. In the carotid angiograms it was evident that the right internal carotid aneurysm was the largest and most proximal one in the carotid system. These factors might have indicated that the right internal carotid aneurysm was responsible for the subarachnoid haemorrhage. Even today these factors do not give absolute indications to localization. With failing vision in the right eye in 1976, there was no doubt that an aneurysm close to the right optic nerve was responsible for her latest symptoms. The difficulty however was to explain how an internal carotid aneurysm projecting laterally could cause compression of the optic nerve, which normally lies medially to the internal carotid artery. Even at operation the problem was not resolved until the optic nerve had been decompressed by the hazardous process of gradual shrinking of the sac of the unidentified aneurysm. It became clear that the relationship of the optic nerve and internal carotid artery had become reversed as the aneurysm of the internal carotid pushed the artery medially by progressive enlargement over the years. Careful study of the right carotid angiogram shows that the carotid siphon is unfolded, and the internal carotid artery shows curvilinear displacement

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medially by the partly thrombosed sac of the aneurysm. A diagramatic sketch, drawn at operation, superimposed on the anteroposterior view of the angiogram, clarifies the position (Fig. 8). Recently several authors writing on the management of patients with multiple aneurysms have recommended surgical obliteration of all accessible aneurysms present. We agree with this view, maintaining that all intact aneurysms should be obliterated, provided that the surgeon is able to do so with a minimum of risk to life or vital structures. For the same reason we have refrained from advocating vertebro-basilar studies in this patient who has already endured enough from surgical intervention. The patient was gratified with recovery of her vision; and the risk of bleeding from a posterior circulation aneurysm, if present, is less than 1% References

1. Griffith, H., The surgery of intracraniaI aneurysms and arteriovenous malformations. In: Recent advances in clinical neurology, pp. 187--217 (Matthews, W. B., ed.). Edinburgh: Churchill Livingstone. 1975. 2. Mount, L. A., Brisman, R., Treatment of multiple aneurysms--symptomatic and asymptomatic. Clin. Neurosurg. 21 (1974), 166--170. 3. Moyes, P.D., Surgical treatment of multiple aneurysms and of incidentally discovered unruptured aneurysms. J. Neurosurg. 35 (1971), 291--295. 4. Pool, J.L., Potts, D.G., Aneurysms and arteriovenous anomalies of the brain. Diagnosis and treatment, pp. 279. New York: Harper and Row. 1965. 5. Sahs, A.L., Perret, G. E., Locksley, H.B., Nishioka, H., Intracranial aneurysms and subaraehnoid .lvaemo~rha~e:-.A. co~perafive--s~u-dy;--pp. --1-o~ "~Ph~-l~d-elpi~ia: J. B. Lippincott. 1969. 6. Sengupta, R.P., Lassman, L.P., Identification of the source of bleeding in multiple intracranial aneurysms. Vasc. Surg. 8 (1974), 177--183. Authors' address: R. P. Sengupta, F.R.C.S., and J. Hankinson, F.R.C.S., Regional Neurological Centre, Newcastle General Hospital, Newcastle upon Tyne, England.

An unusual case of multiple intracranial aneurysms.

ACTA NEUROCHIRURGICA Acta Neurochirurgica 45, 259--275 (1979) 9 by Springer-Verlag 1979 Regional Neurological Centre Newcastle General Hospital, Ne...
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