NEUROCHIRURGIA •
F O R T S C H R I T T E
S u p p l e m e n t u m ad Fortschritte der N e u r o l o g i e u n d Psychiatrie Vol. 18
•
P R O G R E S
© Georg Thieme Verlag, Stuttgart 1975
Stuttgart, im N o v e m b e r 1975
Heft 6
Neurochirurgia 18 (1975), 175-189 © Georg Thieme Verlag Stuttgart
Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae K. Roosen, W. Grote Department of Neurosurgery at the University of Essen (Director: Prof. Dr. W. Grote)
Summary The authors report the diagnosis and successful treatment of a case of traumatic bilateral carotidcavernous sinus fistula. Direct tamponade of the fistula with a Fogarty catheter and ligation of all cervical carotid vessels was carried out on the left side following ligation on the right side of the common carotid, the internal carotid extraand intracranially, and of the external carotid artery. The pre-requisite for this procedure was the development of a functional collateral circulation via the posterior communicating arteries from the basilar system. The 18 months follow-up report andthe rare reports in the world literature on the operative techniques and the results of treatment of similar cases are discussed. Key words: Carotid-cavernous sinus fistula cerebral arteriovenous malformation - head injuries
Zusammenfassung Die Autoren berichten über Diagnostik und erfolgreiche Therapie einer traumatischen, bilateralen Carotis-Sinus cavernosus-Fistel. Nach Unter-
bindung der Aa. carotides communes, interna extra- wie intrakraniell und externa auf der rechten Seite erfolgte auf der Gegenseite die direkte Fisteltamponade mit einem Fogarty-Kuthetei sowie die Ligatur aller zervikalen Karotisgefäße. Voraussetzung für dieses Vorgehen war die Ausbildung eines funktionstüchtigen Kollateralkreislaufs über die Rami comm. posteriores aus dem Basilarisgefäßsystem. Der 18monatige postoperative Beobachtungszeitraum des beschwerdefreien, arbeitsfähigen Patienten wird geschildert. Die in der Weltliteratur seltenen Mitteilungen über Operationstechniken und Behandlungsergebnisse bei ähnlichen Fällen werden diskutiert.
Résumé Diagnostic et traitement des fistules carotidocaverneuses posttraumatiques bilatérales. Les auteurs rendent compte du diagnostic et du traitement d'un cas de fistulas carotido-caverneuses posttraumatiques bilatérales. On procéda à gauche à un tamponnement direct de la fistule avec la sonde Fogarty et à la ligature de tous les vaisseaux carotidiens après avoir, à droite, ligaturé successivement les artères carotide commune et externe ainsi que l'artère carotide interne extra-
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A D V A N C E S
K. Roosen, W. Grote
et intracranienne. La condition requise pour le succès d'une telle procédure fut le développement dÄune vicariance par l'artère fut le développement d'une vicariance par l'artère communicante postérieure et le système basilaire. La catamnèse de 18 mois de ce cas ainsi que les techniques opératoires et les résultats des rares cas semblables relevés dans la littérature mondiale donnent lieu à une discussion. The first description (62) of a fistula of the carotid artery in the cavernous sinus stimulated discussion of the diagnostic and therapeutic problems of this clinical condition. Our understanding of this disease has been facilitated by anatomical considerations (7, 14, 37), clinical studies (12, 16, 20, 21, 27) and aetiological and physiological data (3, 22, 54, 64), as well as by reports of therapeutic experience (2, 5, 7, 9, 12, 16-18, 21, 23, 25, 36, 38, 40, 41, 43, 45, 46, 53, 57, 58, 60, 61). Important additions to our knowledge have come from radiology (6, 8, 27, 56, 59) and ophthalmology (30, 32, 47-49). Assessment of the cases reported has been rendered difficult by the varying terminology employed. An open connection between the carotid artery and the cavernous sinus is described as a fistula in recent Anglo-American and French Literature, while the term frequently used in German literature is carotidcavernous aneurysm. Bettelbeim (1968) gave preference to the term "aneurysm" on the grounds of pathological anatomy, but we support the expression "carotid-cavernous sinus fistula" for three reasons: It facilitates the clear, conceptual differentiation of real infra-clinoidal, intra-cavernous, unilateral (13, 41, 42) or bilateral (1, 22, 24, 31, 35, 51) carotid aneurysm, from which a carotidcavernous sinus fistula may arise. Secondly, the expression accords better with the haemodynamics, and finally, it facilitates understanding in international usage. If there were agreement on a single terminology, confusion, such as is seen in several studies (12, 22, 63) would be avoided. The case described by Hellner (1962) of a
bilateral, symmetrical carotid aneurysm in the cavernous sinus is quoted as an example of a bilateral arteriovenous fistula. On consideration of the clinical data reported by Hellner, and with reference to the literature quoted by him (51) we believe that he was describing a case of the rare, spontaneous bilateral infraclinoid aneurysms of the carotid. A personal observation of a traumatic, bilateral carotid-cavernous sinus fistula led us to review the accounts in the literature concerning the diagnosis and treatment of similar cases.
a) Patients Table 1 summarizes the 20 cases so far described of bilateral carotid-cavernous sinus fistulas. It shows the number, age and sex of patients, and details concerning the pathogenesis, diagnosis and treatment of the condition. Trauma preceded the condition in 13 patients, while the symptoms arose spontaneously in 7. The diagnosis was confirmed clinically and by arteriography in 16 cases, and clinically, by angiography and at autopsy in 3. The diagnosis was first made post mortem in one case.
(b) Symptoms The clinical picture of carotid-cavernous sinus fistula has been fully described in the literature already quoted. Basically, there is no difference from the signs of a unilateral shunt. Three patients showed unilateral signs with angiographically demonstrable bilateral involvement (Patients 5, I; 7; 16). The clinical signs appeared on the opposite side only after unilateral surgical closure of the fistula, in three cases.
c) Treatment No reference is made to therapeutic measures in the report by Clemens and Lodin.
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176
Surgical repair appeared to be impossible in six patients. T w o patients died before treatment w a s begun. Voigt et al. describe the s p o n t a n e o u s closure of a bilateral fistula immediately after angiography. T h e patients referred to by Stolpmann were treated conservatively, by carotid compression, on acc o u n t of their age and because of the operative risks. Closure of t h e fistula w a s d e m o n s t r a t e d arteriographically in one case 15 m o n t h s after the diagnosis h a d been m a d e and t r e a t m e n t begun. T h e second patient left the clinic after one m o n t h ' s t r e a t m e n t : there w a s n o change in her clinical state. In eight cases surgical t r e a t m e n t was performed. Closure of the opposite fistula was impossible in one patient on account of inadequate collateral circulation following unilateral fistula repair ( T a b 1 + 2; Pat 5, II). T w o other patients (Tab 1: 11 + 13) died after successful o p e r a t i o n on t h e first side. Bilateral closure of the fistulas w a s carried o u t on five occasions (Tab 2). T h e d u r a t i o n of the postoperative observation period in 3 patients varied between 30 days a n d 15 m o n t h s . O n e a u t h o r r e p o r t s n o postoperative information and a n o t h e r c o m m e n t s t h a t his patient w a s able t o return to w o r k . T h e external clinical changes and t h e neurological lesions showed satisfactory resolution in all patients.
Personal
Observation
L., K., male aged 19. H o s p . - N o : 6 1 5 / 7 3 . Traffic accident o n 2 1 . 4. 1972. Findings on admission the patient:
to hospital first treating
Unconscious. Protrusion of right (R) bulbus oculi. Pupils: equal, circular and moderately dilated, with positive reaction to light. Corneal reflex: + , both sides. Facial paresis R. Motor restlessness. X-ray of skull: right fronto-temporal fracture extending into base of skull. Progress: 25.4. - Patient responsive, orientated. Organic psychosyndrome. Massive exophthalmos R. 12 Neurochirurgia 18,6
177
Chemosis R. Amaurotic iridoplegia R. Left (L) eye: externally n. a. d. Able to count fingers. 28. 4. - Increasing exophthalmos R. Protrusion of bulbus L. Congestion of conjunctival veins. 4.5. - Carotid Angiography on account of recurrence of drowsiness and unilateral signs. 5. 5. - Because of suspicion of temporal spaceoccupying lesion right temporo-occipital trepanation: no haematoma. 22. 6. - Deterioration in visual acuity L, progressive exophthalmos - therefore investigation at a University Clinic, Ophthalmologics! and Surgical Department. 23. 6. - Two months after accident diagnosis of a bilateral traumatic carotid-cavernous sinus fistula verified by serial angiography. 7.7.-1st operation - Right-sided muscle embolisation 'Brooks'; unable to close fistula. 28.7. - 2nd operation - Ligation of common carotid artery R. 31.7. -3rd operation - Ligation of internal and external carotid arteries R. 15. 9. - 4th operation - Brooks operation L. No fistula closure. Because of unchanged symptoms and signs admission to the Neurosurgical Department of the University Clinic, Essen, 11. 10. Conscious, orientated patient (Figs la + b). Internal examination: N. A. D. ECG normal. Neurological condition: Diminished hearing L. Firstly described: Pulse-synchronous murmur, audible all over the skull; point of maximal intensity both temporal regions; louder L than on the R. Discrete intensification of tendon reflexes in right extremities. Other findings: N. A. D. Local findings: 5 cm long, non-tender scar in the temporo-occipital region R (following exploratory trephination). Wide keloidal scar on R side of neck along the sternomastoid muscle (following ligation of carotid vessels). Ophthalmological findings: Bilateral pulsatile exophthalmos; excessive conjunctival Chemosis. Congestion of conjunctival vessels. Active closure of eyelids incomplete, passive closure complete. Movements in all directions reduced. Extra-orbital prominence: R 25 mm L 26 mm on base 115 (Hertl). Vision: L. 0 . 6 ; R amaurosis. Phthisis oculi R, Fundus not visible. L. e.: Caput Medusae-like dilatation of temporal vessels. Pupil moderately dilated. Prompt reaction to light and convergence. Optic disc 1 dioptre of swelling. EEG: Marked signs of impairment of cerebral bloodflow, more pronounced on R than on L. X-ray: 1. Skull - Fracture line no longer visi-
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Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
K. Roosen, W. Grote
178
Patient
Origin
Diagnosis
Treatment
Subsequent History
1. Clemens, Lodin (1968)
51 year old woman
traumatic
Clinical Arteriography
Not reported
Not reported
2. Dott [1969)
2 cases No details
traumatic
Clinical Arteriography
Nil
Not reported
3. Friedmann, Frowein, Luster (1970)
60 year old woman
Clinical Arteriography
Nil
First investigated 13 years after trauma. No details concerning further progress
4. Grote, Schiefer (1959)
63 year old woman
Clinical
Nil
Not reported
5. Hamby (1966)
Pat. I Young female Pat. 11 59 year old man
Nil Unilateral operation (v. Table 3)
No further details
Author
spontaneous
Clinical Arteriography
traumatic
Clinical Arteriography
Not reported
6. Khilko (1968)
22 year old man
traumatic
Clinical Arteriography
Bilateral operation (v. Table 3)
Discharged fit
7. Kubin, Ortner (1973)
30 year old man
traumatic
Clinical Arteriography Autopsy
Nil
Patient died 6 weeks after trauma
8. Madsen (1970)
70 year old man
traumatic
Clinical Arteriography
Nil
Patient died a few days after accident
9. Mason, Swain, Osheroff (1954)
18 year old man
Clinical Arteriography
Right and leftsided operation (v. Table 3)
Discharged cured 30 days after operation
10. Penzholz (1954)
26 year old man
traumatic
Clinical Arteriography
Bilateral operation (v. Table 3)
Subjectively well one year later
11. Poppen (1951)
66 year old woman
Spontaneous Clinical bilateral rupture Arteriography of carotid Autopsy aneurysms in arterial hypertension
Right-sided operation
Died 22 days after operation from pulmonary embolus
12. Reclus (1908)
18 year old man
Spontaneous Clinical rupture. Vascular Autopsy damage due to syphilis
Nil
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Table 1: Published Bilateral Carotid-Cavernous Sinus Fistulae
Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
Patient
Origin
Diagnosis
I reatment
Subsequent History
13. Schoolman, Kepes (1967)
39 year old woman
Spont. bilat. rupture of the carotids in Ehlers-Danlos syndrome
Clinical Arteriography Autopsy
Operation on left side
Died 3 weeks after op. Cause of death: rupture of aorta with pericardial haemorrhage and tamponade
14. Stolpmann (1972)
Pat. I: 66 year old woman
spontaneous. Arteriosclerosis
Clinical Arteriography
Conservative: Digital compression of the carotid arteries
Subjectively asymptomatic 15 months later. Arteriography: r i g h t - fistula closed left - not performed on account of age and absence of symptoms
Pat. II: 65 year old woman
Clinical spontaneous rupture. Sclero- Arteriography sis. Possible congenital malformation
Conservative: Digital compression of carotids on alternate sides
No change in findings 4 weeks after beginning treatment. No information concerning subsequent progress
45 year old woman
traumatic
Bilateral operation
Patient asymptomatic 1 year after operation. Neurology improved
15. T'aptas (1971)
Clinical Arteriography
(v. Tab. 3)
'. Voigt, Sauer, Dichgans (1971)
17. Zander (1959)
12*
53 year old woman
Spontaneous. Clinical Congenital vas- Arteriography cular anomalies together with labile hypertension
19 year old man
traumatic
Clinical Arteriography
Nil
Spontaneous closure of fistula following angiography. Symptoms improved over 6 weeks. 1 year later: angiogram, EEG and clinical findings normal
Bilateral operation (v. Tab. 3)
Rapid resolution of all lesions except Vlth cranial nerve bilateral lesion. Patient able to work. No longterm follow-up
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Author
179
180
K. Roosen, W. Grote
Table 2: Review of Reported Surgical Treatment in 6 Patients Case Author
Treatment (in chronological order)
Post-operative
Clinical Findings
5, II Hamby
a. Ligation of right common carotid artery. b. Intracranial clipping of right internal carotid artery.
Systolic vascular bruit softer. Other symptoms increased.
6 Kbilko
a. Ligation in neck of internal carotid artery. b. Intracranial clamping of internal carotid and ophthalmic arteries. Contralateral side: c. Intracranial clamping of internal carotid artery. d. Brooks' operation.
9 Mason
a. Ligation of right internal carotid artery in neck. b. Intracranial clamping of right internal carotid artery. c. Ligation of left carotid artery after 5 weeks carotid compression training. d. 2 days later, ligation of left common carotid artery in order to exclude external circulation.
10 Penzbolz
Ligation of left common carotid artery.
b. Ligation of right internal carotid artery. c. Supra-clinoid clamping of internal carotid artery, (right) d. Ligation of right common carotid artery.
15
Taptas
a. Brooks' operation right side. b. Brooks' operation left side.
c. Ligation of right common carotid artery to exclude external carotid system.
17 Zander
Brooks' operations on both sides, without ligation of carotid vessels.
Finally patient discharged cured, subjectively and objectively normal. Vertebral angiography demonstrated adequate collateral blood supply via basilar artery. Vascular bruit softer. Bruit: R L increased Arteriography demonstrated fistula on left, therefore operation c. and d. Patient discharged fit 32 days after the last operation. Exophthalamos and vascular bruit no longer present. Bilateral Vlth nerve paresis improving. Bruit softer on L side. Regression of all signs in L. eye. Now: Signs of right-sided fistula. Increasing signs on right side. Progressive deterioration on right. Now regression of all signs. Improvement in visual acuity. Last findings: Patient asymptomatic. Vlth nerve palsy L.e. Visual acuity: R.e. 1/20, L.e. 5/7. Occlusion of internal carotid artery following the embolisation and fistula occlusion. Right spastic hemiparesis, speech disturbance. Discharged 3 months after last operation. Follow-up investigation: - 1 year later Visual acuity: R.e. N.A.D., L.e. 5/10. R sided ptosis. Increased tendon reflexes right side, no paresis, no dysarthria. All signs regressed post-operatively, apart from Vlth nerve palsy. Patient later able to return to work.
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No further surgical interference on account of inadequacy of collateral circulation and occlusion of posterior communicating artery.
a)
181
b)
Fig. 1: Photograph of patient before operative treatment (a) Full gace (b) Right profile. ble. Trepanation defect R temporo-occipital. 2 metal-clips in infraclinoidal position (marked muscle emboli). 2. Thorax - N . A. D., in particular no cardiac signs of peripheral arteriovenous shunt. 3. Cerebral serial angiography a) Right brachial artery (Figs 2 a + b) - Retrograde filling through the Circle of Willis of bilateral cavernous sinus fistulas, cerebral arteries and carotid vessels as far as site of ligation. b) Left brachial artery (Figs 3 a + b ) - Visualisation of left cavernous sinus system on route through basilar artery and right posterior communicating a., and in the AP projection the rightsided venous outflow can also be seen, in the form of a bilateral arteriovenous fistula. The differences in filling of the posterior vessels (later on the left, and less well contrasted than on the right) in the AP view clearly demonstrates the "steal-syndrome" due to the fistula. Treatment
(Fig. 4)
7 - 1st operation 11.12.72. Exposure of the right cervical carotid vessels. Release of the pre-
viously applied ligatures on the external and internal carotid arteries. Under fluoroscopy control it was attempted to pass a Fogarty catheter along the right internal carotid artery into the fistulous region, so as to interrupt the arteriovenous shunt. In spite of using catheters of various sizes, it was not possible to pass the catheter over the curve of the siphon into the fistulous region. The internal and external carotids were again ligated to prevent proximal inflow. Í7 - 2nd operation 9. 1. 73. Intracranial rightsided internal carotid ligation by two metal clips proximal to the origin of the posterior communicating artery. During the operation, the rightsided vascular murmur became much softer. The bruit still perceptible was probably transmitted from the opposite side. A few hours later there was striking improvement in the exophthalmos (R). The patient withstood the treatment without any neurological manifestations. To develop the formation of collateral vessels "cerebral vascular training" in the form of digital compression of the left carotid artery was carried out several times daily under electro-encephalo-
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Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
K. Roosen, W. Grote
Fig. 2: First angiogram after preliminary treatment, described in text, and admission to Department of Neurosurgery, Essen. Angiogram of right brachial artery. (a) lateral view (b) AP view
graphical control. The patient initially complained of giddiness and headache after only three minutes compression, and the EEG showed beta and delta waves. It immediately returned to normal alpha rhythm as soon as the pressure on the carotid was relieved. The duration of compression was continuously increased up to a period of one hour. After eight weeks the patient was able to tolerate compression for one hour without subjective complaints or pathological signs of the EEG. Ill - 3rd operation 15. 3. 73. Continuous EEG control. Ligation of the common carotid artery. Delta waves immediately appeared, but disappeared again two minutes later. 30 min after closure of the external carotid artery, the internal carotid was opened, and a radiopaque Fogarty catheter was successfully advanced to the fistulous region. Following inflation of the catheter balloon the vascular bruit stopped. After 1 hour the EEG remaining normal, the distal end of the
b)
catheter was stopped off, and the left internal carotid a. was ligated. The patient's post-operative condition was good. Exophthalmos L improved. Bulbar protrusion: R 20 mm, L 26 mm, on base of 85. Visual acuity: unchanged. Congestion of the conjunctival, episcleral and fundal veins was no longer visible. Improvement in ocular mobility. The EEG was without abnormalities.
Postoperative serial
angiography
a) L-sided brachial arteriogram (Fig. 5 a + b) The cerebral areas supplied by the carotid are filled through the posterior communicating artery. The catheter balloon, filled with contrast medium, has closed the fistula. b) R-sided brachial arteriogram (Figs 6 a + b) The intra-cerebral branches of the carotid a. are filled via the basilar and the right posterior com-
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182
Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
>T Fig. 3: As in Fig. 4. Angiogram of left brachial artery, (a) lateral view (b) AP view municating artery. No filling of the fistula. The film of the neck region demonstrates the proximal course of the external carotid as far as the site of ligation, via extensive extra-cranial anastomoses with the vertebral artery, without any shunt-inflow through these non-functional collaterals. The patient was discharged symptom-free on 9. 4. 73.
Catamnesis According to details provided by his general practitioner, Mr. L's general and ophthalmological conditions improved markedly over succeeding months. The patient (Figs 7a + b) was admitted for follow-up investigations 18 months after discharge. He reported having been free from symptoms for the past six months and that vision in the L eye had recovered well. He had not yet returned to
b) his work as a joiner, but had done some occasional light work. Clinical and neurological conditions: normal. No vascular bruit could be heard. Ophthalmological findings: Eyelid closure possible actively and passively on both sides. Exophthalmos: R. e. 20 mm, L. e. 21 mm, on base 85. Vision: R. e. amaurotic, probably due to the traumatic lesion of the optic nerve. L. e. 0,8. Phthisis oculi R. Vitreous and fundus L: N. A. D. EEG: Low voltage EEG of alpha type with slight general changes showing no evidence of laterality, focal changes or epileptiform potentials. X-rays: 1. Skull and 2. Thorax: N. A. D. Since the patient felt subjectively well, and all the investigations showed no evidence of any recurrence of the fistula, and since it was intended to carry out further clinical investigations at regular intervals, we did not feel that follow-up angiography was indicated, on account of the risk of cerebral damage as a result of impairment of oxygen supply by angiography or anaesthesia.
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183
Fig. 4: Graphic representation of surgical procedure. Common carotid artery Internal carotid a. External carotid a. Vertebral a. Basilar a. Ant. inf. cerebellar a. Anterior cerebral a. Middle cerebral a. Odontoid process Maxillary sinus Orbit
I - 1st op. - Ligation of the right cervical carotid arteries. II - 2nd op. - Intracranial clipping of right internal carotid artery. Ill - 3rd op. - Closure of the left fistula by a Fogarty catheter. Ligation of common, internal, and external carotid arteries on the left in the cervical region.
Fig. 5: Left brachial arteriogram at end of surgical treatment (a) lateral view (b) AP view
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1 2 3 4 5 6 7 8 9 10 11
185
Fig. 6: Right brachial arteriogram after surgery (a) AP view (b) lateral view including cervical arteriogram. Fig. 7: Photograph of patient 18 months after surgery, (a) Full gace (b) right profile.
a)
M0 -
m
Bp
F O R T S C H R I T T E
S u p p l e m e n t u m ad Fortschritte der N e u r o l o g i e u n d Psychiatrie Vol. 18
•
P R O G R E S
© Georg Thieme Verlag, Stuttgart 1975
Stuttgart, im N o v e m b e r 1975
Heft 6
Neurochirurgia 18 (1975), 175-189 © Georg Thieme Verlag Stuttgart
Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae K. Roosen, W. Grote Department of Neurosurgery at the University of Essen (Director: Prof. Dr. W. Grote)
Summary The authors report the diagnosis and successful treatment of a case of traumatic bilateral carotidcavernous sinus fistula. Direct tamponade of the fistula with a Fogarty catheter and ligation of all cervical carotid vessels was carried out on the left side following ligation on the right side of the common carotid, the internal carotid extraand intracranially, and of the external carotid artery. The pre-requisite for this procedure was the development of a functional collateral circulation via the posterior communicating arteries from the basilar system. The 18 months follow-up report andthe rare reports in the world literature on the operative techniques and the results of treatment of similar cases are discussed. Key words: Carotid-cavernous sinus fistula cerebral arteriovenous malformation - head injuries
Zusammenfassung Die Autoren berichten über Diagnostik und erfolgreiche Therapie einer traumatischen, bilateralen Carotis-Sinus cavernosus-Fistel. Nach Unter-
bindung der Aa. carotides communes, interna extra- wie intrakraniell und externa auf der rechten Seite erfolgte auf der Gegenseite die direkte Fisteltamponade mit einem Fogarty-Kuthetei sowie die Ligatur aller zervikalen Karotisgefäße. Voraussetzung für dieses Vorgehen war die Ausbildung eines funktionstüchtigen Kollateralkreislaufs über die Rami comm. posteriores aus dem Basilarisgefäßsystem. Der 18monatige postoperative Beobachtungszeitraum des beschwerdefreien, arbeitsfähigen Patienten wird geschildert. Die in der Weltliteratur seltenen Mitteilungen über Operationstechniken und Behandlungsergebnisse bei ähnlichen Fällen werden diskutiert.
Résumé Diagnostic et traitement des fistules carotidocaverneuses posttraumatiques bilatérales. Les auteurs rendent compte du diagnostic et du traitement d'un cas de fistulas carotido-caverneuses posttraumatiques bilatérales. On procéda à gauche à un tamponnement direct de la fistule avec la sonde Fogarty et à la ligature de tous les vaisseaux carotidiens après avoir, à droite, ligaturé successivement les artères carotide commune et externe ainsi que l'artère carotide interne extra-
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A D V A N C E S
K. Roosen, W. Grote
et intracranienne. La condition requise pour le succès d'une telle procédure fut le développement dÄune vicariance par l'artère fut le développement d'une vicariance par l'artère communicante postérieure et le système basilaire. La catamnèse de 18 mois de ce cas ainsi que les techniques opératoires et les résultats des rares cas semblables relevés dans la littérature mondiale donnent lieu à une discussion. The first description (62) of a fistula of the carotid artery in the cavernous sinus stimulated discussion of the diagnostic and therapeutic problems of this clinical condition. Our understanding of this disease has been facilitated by anatomical considerations (7, 14, 37), clinical studies (12, 16, 20, 21, 27) and aetiological and physiological data (3, 22, 54, 64), as well as by reports of therapeutic experience (2, 5, 7, 9, 12, 16-18, 21, 23, 25, 36, 38, 40, 41, 43, 45, 46, 53, 57, 58, 60, 61). Important additions to our knowledge have come from radiology (6, 8, 27, 56, 59) and ophthalmology (30, 32, 47-49). Assessment of the cases reported has been rendered difficult by the varying terminology employed. An open connection between the carotid artery and the cavernous sinus is described as a fistula in recent Anglo-American and French Literature, while the term frequently used in German literature is carotidcavernous aneurysm. Bettelbeim (1968) gave preference to the term "aneurysm" on the grounds of pathological anatomy, but we support the expression "carotid-cavernous sinus fistula" for three reasons: It facilitates the clear, conceptual differentiation of real infra-clinoidal, intra-cavernous, unilateral (13, 41, 42) or bilateral (1, 22, 24, 31, 35, 51) carotid aneurysm, from which a carotidcavernous sinus fistula may arise. Secondly, the expression accords better with the haemodynamics, and finally, it facilitates understanding in international usage. If there were agreement on a single terminology, confusion, such as is seen in several studies (12, 22, 63) would be avoided. The case described by Hellner (1962) of a
bilateral, symmetrical carotid aneurysm in the cavernous sinus is quoted as an example of a bilateral arteriovenous fistula. On consideration of the clinical data reported by Hellner, and with reference to the literature quoted by him (51) we believe that he was describing a case of the rare, spontaneous bilateral infraclinoid aneurysms of the carotid. A personal observation of a traumatic, bilateral carotid-cavernous sinus fistula led us to review the accounts in the literature concerning the diagnosis and treatment of similar cases.
a) Patients Table 1 summarizes the 20 cases so far described of bilateral carotid-cavernous sinus fistulas. It shows the number, age and sex of patients, and details concerning the pathogenesis, diagnosis and treatment of the condition. Trauma preceded the condition in 13 patients, while the symptoms arose spontaneously in 7. The diagnosis was confirmed clinically and by arteriography in 16 cases, and clinically, by angiography and at autopsy in 3. The diagnosis was first made post mortem in one case.
(b) Symptoms The clinical picture of carotid-cavernous sinus fistula has been fully described in the literature already quoted. Basically, there is no difference from the signs of a unilateral shunt. Three patients showed unilateral signs with angiographically demonstrable bilateral involvement (Patients 5, I; 7; 16). The clinical signs appeared on the opposite side only after unilateral surgical closure of the fistula, in three cases.
c) Treatment No reference is made to therapeutic measures in the report by Clemens and Lodin.
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176
Surgical repair appeared to be impossible in six patients. T w o patients died before treatment w a s begun. Voigt et al. describe the s p o n t a n e o u s closure of a bilateral fistula immediately after angiography. T h e patients referred to by Stolpmann were treated conservatively, by carotid compression, on acc o u n t of their age and because of the operative risks. Closure of t h e fistula w a s d e m o n s t r a t e d arteriographically in one case 15 m o n t h s after the diagnosis h a d been m a d e and t r e a t m e n t begun. T h e second patient left the clinic after one m o n t h ' s t r e a t m e n t : there w a s n o change in her clinical state. In eight cases surgical t r e a t m e n t was performed. Closure of the opposite fistula was impossible in one patient on account of inadequate collateral circulation following unilateral fistula repair ( T a b 1 + 2; Pat 5, II). T w o other patients (Tab 1: 11 + 13) died after successful o p e r a t i o n on t h e first side. Bilateral closure of the fistulas w a s carried o u t on five occasions (Tab 2). T h e d u r a t i o n of the postoperative observation period in 3 patients varied between 30 days a n d 15 m o n t h s . O n e a u t h o r r e p o r t s n o postoperative information and a n o t h e r c o m m e n t s t h a t his patient w a s able t o return to w o r k . T h e external clinical changes and t h e neurological lesions showed satisfactory resolution in all patients.
Personal
Observation
L., K., male aged 19. H o s p . - N o : 6 1 5 / 7 3 . Traffic accident o n 2 1 . 4. 1972. Findings on admission the patient:
to hospital first treating
Unconscious. Protrusion of right (R) bulbus oculi. Pupils: equal, circular and moderately dilated, with positive reaction to light. Corneal reflex: + , both sides. Facial paresis R. Motor restlessness. X-ray of skull: right fronto-temporal fracture extending into base of skull. Progress: 25.4. - Patient responsive, orientated. Organic psychosyndrome. Massive exophthalmos R. 12 Neurochirurgia 18,6
177
Chemosis R. Amaurotic iridoplegia R. Left (L) eye: externally n. a. d. Able to count fingers. 28. 4. - Increasing exophthalmos R. Protrusion of bulbus L. Congestion of conjunctival veins. 4.5. - Carotid Angiography on account of recurrence of drowsiness and unilateral signs. 5. 5. - Because of suspicion of temporal spaceoccupying lesion right temporo-occipital trepanation: no haematoma. 22. 6. - Deterioration in visual acuity L, progressive exophthalmos - therefore investigation at a University Clinic, Ophthalmologics! and Surgical Department. 23. 6. - Two months after accident diagnosis of a bilateral traumatic carotid-cavernous sinus fistula verified by serial angiography. 7.7.-1st operation - Right-sided muscle embolisation 'Brooks'; unable to close fistula. 28.7. - 2nd operation - Ligation of common carotid artery R. 31.7. -3rd operation - Ligation of internal and external carotid arteries R. 15. 9. - 4th operation - Brooks operation L. No fistula closure. Because of unchanged symptoms and signs admission to the Neurosurgical Department of the University Clinic, Essen, 11. 10. Conscious, orientated patient (Figs la + b). Internal examination: N. A. D. ECG normal. Neurological condition: Diminished hearing L. Firstly described: Pulse-synchronous murmur, audible all over the skull; point of maximal intensity both temporal regions; louder L than on the R. Discrete intensification of tendon reflexes in right extremities. Other findings: N. A. D. Local findings: 5 cm long, non-tender scar in the temporo-occipital region R (following exploratory trephination). Wide keloidal scar on R side of neck along the sternomastoid muscle (following ligation of carotid vessels). Ophthalmological findings: Bilateral pulsatile exophthalmos; excessive conjunctival Chemosis. Congestion of conjunctival vessels. Active closure of eyelids incomplete, passive closure complete. Movements in all directions reduced. Extra-orbital prominence: R 25 mm L 26 mm on base 115 (Hertl). Vision: L. 0 . 6 ; R amaurosis. Phthisis oculi R, Fundus not visible. L. e.: Caput Medusae-like dilatation of temporal vessels. Pupil moderately dilated. Prompt reaction to light and convergence. Optic disc 1 dioptre of swelling. EEG: Marked signs of impairment of cerebral bloodflow, more pronounced on R than on L. X-ray: 1. Skull - Fracture line no longer visi-
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Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
K. Roosen, W. Grote
178
Patient
Origin
Diagnosis
Treatment
Subsequent History
1. Clemens, Lodin (1968)
51 year old woman
traumatic
Clinical Arteriography
Not reported
Not reported
2. Dott [1969)
2 cases No details
traumatic
Clinical Arteriography
Nil
Not reported
3. Friedmann, Frowein, Luster (1970)
60 year old woman
Clinical Arteriography
Nil
First investigated 13 years after trauma. No details concerning further progress
4. Grote, Schiefer (1959)
63 year old woman
Clinical
Nil
Not reported
5. Hamby (1966)
Pat. I Young female Pat. 11 59 year old man
Nil Unilateral operation (v. Table 3)
No further details
Author
spontaneous
Clinical Arteriography
traumatic
Clinical Arteriography
Not reported
6. Khilko (1968)
22 year old man
traumatic
Clinical Arteriography
Bilateral operation (v. Table 3)
Discharged fit
7. Kubin, Ortner (1973)
30 year old man
traumatic
Clinical Arteriography Autopsy
Nil
Patient died 6 weeks after trauma
8. Madsen (1970)
70 year old man
traumatic
Clinical Arteriography
Nil
Patient died a few days after accident
9. Mason, Swain, Osheroff (1954)
18 year old man
Clinical Arteriography
Right and leftsided operation (v. Table 3)
Discharged cured 30 days after operation
10. Penzholz (1954)
26 year old man
traumatic
Clinical Arteriography
Bilateral operation (v. Table 3)
Subjectively well one year later
11. Poppen (1951)
66 year old woman
Spontaneous Clinical bilateral rupture Arteriography of carotid Autopsy aneurysms in arterial hypertension
Right-sided operation
Died 22 days after operation from pulmonary embolus
12. Reclus (1908)
18 year old man
Spontaneous Clinical rupture. Vascular Autopsy damage due to syphilis
Nil
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Table 1: Published Bilateral Carotid-Cavernous Sinus Fistulae
Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
Patient
Origin
Diagnosis
I reatment
Subsequent History
13. Schoolman, Kepes (1967)
39 year old woman
Spont. bilat. rupture of the carotids in Ehlers-Danlos syndrome
Clinical Arteriography Autopsy
Operation on left side
Died 3 weeks after op. Cause of death: rupture of aorta with pericardial haemorrhage and tamponade
14. Stolpmann (1972)
Pat. I: 66 year old woman
spontaneous. Arteriosclerosis
Clinical Arteriography
Conservative: Digital compression of the carotid arteries
Subjectively asymptomatic 15 months later. Arteriography: r i g h t - fistula closed left - not performed on account of age and absence of symptoms
Pat. II: 65 year old woman
Clinical spontaneous rupture. Sclero- Arteriography sis. Possible congenital malformation
Conservative: Digital compression of carotids on alternate sides
No change in findings 4 weeks after beginning treatment. No information concerning subsequent progress
45 year old woman
traumatic
Bilateral operation
Patient asymptomatic 1 year after operation. Neurology improved
15. T'aptas (1971)
Clinical Arteriography
(v. Tab. 3)
'. Voigt, Sauer, Dichgans (1971)
17. Zander (1959)
12*
53 year old woman
Spontaneous. Clinical Congenital vas- Arteriography cular anomalies together with labile hypertension
19 year old man
traumatic
Clinical Arteriography
Nil
Spontaneous closure of fistula following angiography. Symptoms improved over 6 weeks. 1 year later: angiogram, EEG and clinical findings normal
Bilateral operation (v. Tab. 3)
Rapid resolution of all lesions except Vlth cranial nerve bilateral lesion. Patient able to work. No longterm follow-up
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Author
179
180
K. Roosen, W. Grote
Table 2: Review of Reported Surgical Treatment in 6 Patients Case Author
Treatment (in chronological order)
Post-operative
Clinical Findings
5, II Hamby
a. Ligation of right common carotid artery. b. Intracranial clipping of right internal carotid artery.
Systolic vascular bruit softer. Other symptoms increased.
6 Kbilko
a. Ligation in neck of internal carotid artery. b. Intracranial clamping of internal carotid and ophthalmic arteries. Contralateral side: c. Intracranial clamping of internal carotid artery. d. Brooks' operation.
9 Mason
a. Ligation of right internal carotid artery in neck. b. Intracranial clamping of right internal carotid artery. c. Ligation of left carotid artery after 5 weeks carotid compression training. d. 2 days later, ligation of left common carotid artery in order to exclude external circulation.
10 Penzbolz
Ligation of left common carotid artery.
b. Ligation of right internal carotid artery. c. Supra-clinoid clamping of internal carotid artery, (right) d. Ligation of right common carotid artery.
15
Taptas
a. Brooks' operation right side. b. Brooks' operation left side.
c. Ligation of right common carotid artery to exclude external carotid system.
17 Zander
Brooks' operations on both sides, without ligation of carotid vessels.
Finally patient discharged cured, subjectively and objectively normal. Vertebral angiography demonstrated adequate collateral blood supply via basilar artery. Vascular bruit softer. Bruit: R L increased Arteriography demonstrated fistula on left, therefore operation c. and d. Patient discharged fit 32 days after the last operation. Exophthalamos and vascular bruit no longer present. Bilateral Vlth nerve paresis improving. Bruit softer on L side. Regression of all signs in L. eye. Now: Signs of right-sided fistula. Increasing signs on right side. Progressive deterioration on right. Now regression of all signs. Improvement in visual acuity. Last findings: Patient asymptomatic. Vlth nerve palsy L.e. Visual acuity: R.e. 1/20, L.e. 5/7. Occlusion of internal carotid artery following the embolisation and fistula occlusion. Right spastic hemiparesis, speech disturbance. Discharged 3 months after last operation. Follow-up investigation: - 1 year later Visual acuity: R.e. N.A.D., L.e. 5/10. R sided ptosis. Increased tendon reflexes right side, no paresis, no dysarthria. All signs regressed post-operatively, apart from Vlth nerve palsy. Patient later able to return to work.
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No further surgical interference on account of inadequacy of collateral circulation and occlusion of posterior communicating artery.
a)
181
b)
Fig. 1: Photograph of patient before operative treatment (a) Full gace (b) Right profile. ble. Trepanation defect R temporo-occipital. 2 metal-clips in infraclinoidal position (marked muscle emboli). 2. Thorax - N . A. D., in particular no cardiac signs of peripheral arteriovenous shunt. 3. Cerebral serial angiography a) Right brachial artery (Figs 2 a + b) - Retrograde filling through the Circle of Willis of bilateral cavernous sinus fistulas, cerebral arteries and carotid vessels as far as site of ligation. b) Left brachial artery (Figs 3 a + b ) - Visualisation of left cavernous sinus system on route through basilar artery and right posterior communicating a., and in the AP projection the rightsided venous outflow can also be seen, in the form of a bilateral arteriovenous fistula. The differences in filling of the posterior vessels (later on the left, and less well contrasted than on the right) in the AP view clearly demonstrates the "steal-syndrome" due to the fistula. Treatment
(Fig. 4)
7 - 1st operation 11.12.72. Exposure of the right cervical carotid vessels. Release of the pre-
viously applied ligatures on the external and internal carotid arteries. Under fluoroscopy control it was attempted to pass a Fogarty catheter along the right internal carotid artery into the fistulous region, so as to interrupt the arteriovenous shunt. In spite of using catheters of various sizes, it was not possible to pass the catheter over the curve of the siphon into the fistulous region. The internal and external carotids were again ligated to prevent proximal inflow. Í7 - 2nd operation 9. 1. 73. Intracranial rightsided internal carotid ligation by two metal clips proximal to the origin of the posterior communicating artery. During the operation, the rightsided vascular murmur became much softer. The bruit still perceptible was probably transmitted from the opposite side. A few hours later there was striking improvement in the exophthalmos (R). The patient withstood the treatment without any neurological manifestations. To develop the formation of collateral vessels "cerebral vascular training" in the form of digital compression of the left carotid artery was carried out several times daily under electro-encephalo-
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Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
K. Roosen, W. Grote
Fig. 2: First angiogram after preliminary treatment, described in text, and admission to Department of Neurosurgery, Essen. Angiogram of right brachial artery. (a) lateral view (b) AP view
graphical control. The patient initially complained of giddiness and headache after only three minutes compression, and the EEG showed beta and delta waves. It immediately returned to normal alpha rhythm as soon as the pressure on the carotid was relieved. The duration of compression was continuously increased up to a period of one hour. After eight weeks the patient was able to tolerate compression for one hour without subjective complaints or pathological signs of the EEG. Ill - 3rd operation 15. 3. 73. Continuous EEG control. Ligation of the common carotid artery. Delta waves immediately appeared, but disappeared again two minutes later. 30 min after closure of the external carotid artery, the internal carotid was opened, and a radiopaque Fogarty catheter was successfully advanced to the fistulous region. Following inflation of the catheter balloon the vascular bruit stopped. After 1 hour the EEG remaining normal, the distal end of the
b)
catheter was stopped off, and the left internal carotid a. was ligated. The patient's post-operative condition was good. Exophthalmos L improved. Bulbar protrusion: R 20 mm, L 26 mm, on base of 85. Visual acuity: unchanged. Congestion of the conjunctival, episcleral and fundal veins was no longer visible. Improvement in ocular mobility. The EEG was without abnormalities.
Postoperative serial
angiography
a) L-sided brachial arteriogram (Fig. 5 a + b) The cerebral areas supplied by the carotid are filled through the posterior communicating artery. The catheter balloon, filled with contrast medium, has closed the fistula. b) R-sided brachial arteriogram (Figs 6 a + b) The intra-cerebral branches of the carotid a. are filled via the basilar and the right posterior com-
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182
Diagnosis and Treatment of Bilateral Traumatic Carotid-Cavernous Sinus Fistulae
>T Fig. 3: As in Fig. 4. Angiogram of left brachial artery, (a) lateral view (b) AP view municating artery. No filling of the fistula. The film of the neck region demonstrates the proximal course of the external carotid as far as the site of ligation, via extensive extra-cranial anastomoses with the vertebral artery, without any shunt-inflow through these non-functional collaterals. The patient was discharged symptom-free on 9. 4. 73.
Catamnesis According to details provided by his general practitioner, Mr. L's general and ophthalmological conditions improved markedly over succeeding months. The patient (Figs 7a + b) was admitted for follow-up investigations 18 months after discharge. He reported having been free from symptoms for the past six months and that vision in the L eye had recovered well. He had not yet returned to
b) his work as a joiner, but had done some occasional light work. Clinical and neurological conditions: normal. No vascular bruit could be heard. Ophthalmological findings: Eyelid closure possible actively and passively on both sides. Exophthalmos: R. e. 20 mm, L. e. 21 mm, on base 85. Vision: R. e. amaurotic, probably due to the traumatic lesion of the optic nerve. L. e. 0,8. Phthisis oculi R. Vitreous and fundus L: N. A. D. EEG: Low voltage EEG of alpha type with slight general changes showing no evidence of laterality, focal changes or epileptiform potentials. X-rays: 1. Skull and 2. Thorax: N. A. D. Since the patient felt subjectively well, and all the investigations showed no evidence of any recurrence of the fistula, and since it was intended to carry out further clinical investigations at regular intervals, we did not feel that follow-up angiography was indicated, on account of the risk of cerebral damage as a result of impairment of oxygen supply by angiography or anaesthesia.
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183
Fig. 4: Graphic representation of surgical procedure. Common carotid artery Internal carotid a. External carotid a. Vertebral a. Basilar a. Ant. inf. cerebellar a. Anterior cerebral a. Middle cerebral a. Odontoid process Maxillary sinus Orbit
I - 1st op. - Ligation of the right cervical carotid arteries. II - 2nd op. - Intracranial clipping of right internal carotid artery. Ill - 3rd op. - Closure of the left fistula by a Fogarty catheter. Ligation of common, internal, and external carotid arteries on the left in the cervical region.
Fig. 5: Left brachial arteriogram at end of surgical treatment (a) lateral view (b) AP view
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1 2 3 4 5 6 7 8 9 10 11
185
Fig. 6: Right brachial arteriogram after surgery (a) AP view (b) lateral view including cervical arteriogram. Fig. 7: Photograph of patient 18 months after surgery, (a) Full gace (b) right profile.
a)
M0 -
m
Bp
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