J Neurosurg76:714-717, 1992

Spontaneous intracerebral hematoma from occult carotidcavernous fistula during pregnancy and puerperium Case report Tzu-KANG LIN, M.D., CHEN-NEN CHANG, M.D., AND YAU-YAU WAI, M.D. Divisions of Neurosurgery and Neuroradiology. Chang Gung Medical College and Memorial Hospital, Taipei, Taiwan ~" In most cases, intracerebral hemorrhage during pregnancy or puerpefium results from cerebral aneurysms or arteriovenous malformations. The authors present a case of a 30-year-old woman whose symptoms from a traumatic carotid-cavernous fistula had completely resolved 189years after the event, but recurred 4 years later, causing two hemorrhages during pregnancy (33rd and 35th week of gestation) and one during the postpartum period (10 days after Caesarean section). Partial thrombosis of the cavernous sinus with obliteration of most of the drainage from the fistula accounted for the resolution of clinical symptoms, but also promoted backflow to the preserved drainage of superficial cortical veins. The hemodynamic changes and the hormonal effects due to the patient's subsequent pregnancy further aggravated the venous engorgement and finally caused rupture. All three hematomas occurred in the vicinity of the extremely dilated veins, suggesting that back-flow with venous hypertension was the probable cause for the intracerebral hematomas. Spontaneous healing of the carotid-cavernous fistula should be confirmed with cerebral angiography.

KEY WORDS carotid-cavernousfistula venous hypertension

LTHOUGH the carotid-cavernous fistula (CCF) has been a well-known entity since it was first reported by Travers in 1809, 23 there have been only a few papers documenting the life-threatening complication of an intracerebral hematoma. ~6'2~ A CCF complicated by an intracerebral hematoma during pregnancy and shortly after childbirth has not yet been reported. We report a clinically asymptomatic traumatic CCF associated with intracerebral hemorrhage twice during a subsequent pregnancy and once shortly after Caesarean section. The mechanisms of these bleeds and the literature are reviewed.

A

Case Report This 30-year-old woman was involved in a traffic accident in April, 1985. Four months later, she was brought to our hospital because of progressive development of right tinnitus, double vision, blurred vision, and protrusion and congestion of the right eye. First Admission. On admission, a bruit could be auscultated around the right eye. There was oculomotor 714

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and trochlear nerve paresis with abducens nerve palsy on the right. Visual acuity was 201100 in the right eye and 20/20 in the left, and exophthalmos of the fight eye was 2 mm greater than that of the left. The conjunctiva of the fight eye was hyperemic. Right carotid angiography revealed early opacification of the right cavernous sinus with high flow. Venous drainage was by way of the superior ophthalmic vein, petrosal sinus, sylvian vein, and basal vein of Rosenthal (Fig. 1). The patient refused treatment because of improvement in her symptoms. About 6 months after examination, double vision and hyperemia of the right eye were completely gone. The bruit was no longer detectable and vision in the right eye was normal 189 years later. She remained asymptomatic for 3 years. Second Admission. On August 8, 1989, during the 33rd week of her seventh pregnancy, the patient suffered a sudden loss of consciousness associated with a left hemiplegia. There was no hyperemia in the conjunctiva of the fight eye, and no bruit could be auscultated. Emergency computerized tomography showed a large right frontal intracerebral hematoma (Fig. 2 left).

J. Neurosurg. / Volume 76/April, 1992

Intracerebral hematoma from CCF daring pregnancy Operations. The hematoma was evacuated immediately and the patient regained consciousness gradually, although she was still very drowsy and the left hemiplegla persisted. About 2 weeks postoperatively (in the 35th week of pregnancy), the patient suddenly became comatose again. Computerized tomography revealed a recurrent right frontal hematoma (Fig. 2 right). Cerebral angiography showed only partial filling of the right cavernous sinus. The early venous drainage was by way of the sylvian vein and then to the superior sagittal sinus and the transverse sinus via the veins of Trolard and Labbr, respectively, which remained filled in the late arterial phase. The superficial cortical veins were extremely dilated (Fig. 3). Venous drainage to the superior ophthalmic vein, petrosal sinus, and basal vein of Rosenthal, demonstrated in the previous angiogram, were not evident in the second angiogram. A cross-compression study showed good collateral circulation from left to fight through the anterior communicating artery. The extreme swelling of the brain made definitive surgical treatment of the CCF using a Jaeger-Hamby procedure 1~'~ difficult even after the hematoma was removed. A Caesarean section was performed immediately after the craniotomy at the request of the family due to the extremely critical condition of the patient. Surprisingly, the patient gradually recovered consciousness, but 10 days after the Caesarean section, she suddenly again became comatose due to a right frontotemporal intracerebral hematoma (Fig. 4). The hematoma was removed immediately, followed by trapping and muscle embolization of the right internal carotid artery. Postoperative Course. The patient recovered gradually after the last operation. At a follow-up examination 1) years postoperatively, she was alert, but the dense left hemiparesis persisted. The baby developed normally.

FIG. 1. Right carotid angiogram, lateral view, showing opacification of the right cavernous sinus, draining via the superior ophthalmic vein (large open arrow), petrosal sinus (small open arron9, sylvian vein (large closed arrow), and basal vein of Rosenthal (small closed arrow).

FIG. 2. Left: Cranial computerized tomography (CT) scan obtained during the 33rd week of the patient's pregnancy showing a large right frontal intracerebral hematoma. Right: Cranial CT scan obtained 2 weeks later showing a recurrent right frontal intracerebral hematoma.

FIG. 3. Cerebral angiograms, early arterial (A), arterial (B), and late arterial (C) phases, obtained during the 35th week of the patient's pregnancy revealing early partial filling of the right cavernous sinus. Venous drainage is by way of the extremely dilated sylvian vein, and then to the superior sagittal sinus and the transverse sinus via the dilated veins of Trolard and LobbY, respectively. The late arterial phase angiogram clearly demonstrates the dilated cortical veins (arrowhead), indicating venous stasis and hypertension.

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T. K. Lin, C. N. Chang, and Y. Y. Wai Discussion

Veins as a Source of Hemorrhage Intracerebral hemorrhage during pregnancy and puerperium is a rare but well-documented entity. Most of the cases result from cerebral aneurysms or arteriovenous malformations. Spontaneous intracerebral hemorrhage from an occult CCF during pregnancy and puerperium has not previously been reported. In general, catastrophic complications from CCF are uncommon. The goal of CCF treatment usually includes improvement or preservation of vision, return of the orbit and contents to normal, and elimination of bruit. Lifethreatening intracerebral hemorrhage from CCF is reported to occur in approximately 3% of patients. 9'-~ In our case, the second angiography obtained during pregnancy demonstrated only a partial filling of the right cavernous sinus. Moreover, venous drainage via the superior ophthalmic vein, petrosal sinus, and basal vein of Rosenthal observed on the first angiogram was no longer evident. The only drainage preserved from the right cavernous sinus was the back-flow to the extremely dilated sylvian vein, and then through the quite dilated veins of Trolard and Labb~ to the superior sagittal sinus and the transverse sinus, respectively. The dilated right cortical veins remained filled through the late arterial phase (Fig. 3C). Spontaneous intracerebral hemorrhage, occurring three times in this case, was limited to the vicinity of the extremely dilated and engorged cortical veins, demonstrating that the backflow with venous hypertension was the principal cause of bleeding. We deduce that partial thrombosis of the right cavernous sinus associated with obliteration of most of the venous drainage, especially the superior ophthalmic vein, gave rise to the resolution of the patient's eye signs and the bruit. Nevertheless, this also rendered the fight cortical veins, which were the only

preserved drainage routes of the occult CCF, more dilated and engorged. In the subsequent pregnancy, the hemodynamic changes and the hormonal effects of pregnancy further aggravated the dilatation and engorgement of the right superficial cortical veins, finally causing the intracerebral hemorrhage.

Systemic Changes With Pregnancy In a recent extensive analysis of reported cases of intracerebral hemorrhage during pregnancy, good correlation is found between the increased frequency of both aneurysmal and angiomatous intracerebral hemorrhages and the advancing gestational age. 7 This fact suggests that the hemodynamic changes during pregnancy may be an important cause of intracerebral hemorrhage. Cardiac output increases by 60% in the first two trimesters and remains at a high level in the third trimester. Venous pressure and blood volume rise rather steadily throughout pregnancy and reach a maximum level near term. t7'25 Hormonal effects during pregnancy are also suggested to play a role in the cause of intracerebral hemorrhage. Circulating estrogen may dilate the abnormal blood vessels. The secretion of relaxin, which induces a general change in fibrous tissue and a relaxation of pelvic ligaments, also may be a causative factor? '8'9 Conclusions

Although spontaneous resolution of CCF reportedly occurs in 5% to 60% of patients, 3-5'lz~s'26only a few reports have demonstrated the spontaneous disappearance of CCF on cerebral angiography. ~~ We stress the importance of cerebral angiography to confirm the true disappearance of CCF. Otherwise, the occult CCF may be missed and is likely to result in the catastrophic complication of intracerebral hemorrhage if back-flow occurs and causes venous hypertension. References

1. Ambler MW, Moon AC, Stumer WQ: Bilateral carotidcavernous fistulae of mixed types with unusual radiological and neuropathologieal findings. Case report. J Neurosurg 48:117-124, 1978 2. Amias AG: Cerebral vascular disease in pregnancy. I. Haemorrhage. J Obstet Gynaeeol Br Commonw 77: 100-120, 1970 3. Aminoff MJ: Vascular anomalies in the intracranial dura mater. Brain 96:601-612, 1973 4. Barrow DL, Spector RH, Braun IF, et al: Classification and treatment of spontaneous carotid-cavernous sinus fistulas. J Neurosurg 62:248-256, 1985 5. Day AL, Rhoton AL Jr: Aneurysms and arteriovenous fistulae of the intracavernous carotid artery and its branches, in Youmans JR (ed): Neurological Surgery, ed 3. Philadelphia: WB Saunders, 1990, Vol 3, pp 1807-1830 6. deSchweinitz GE, Holloway TB: Pulsating ExophthalFIG. 4. Cranial computerized tomography obtained 10 days after Caesarean section showing a right frontmemporal hematoma.

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7. Dias MS, Sekhar LN: Intracranial hemorrhage from aneurysms and arteriovenous malformations during preg-

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nancy and the puerperium. Neurosurgery 27:855-865, 1990 Dimsdale H: Pregnancy and lesions of the nervous system. Neurological emergencies in pregnancy. Proc R SOc Med 55:571-575, 1962 Dohrmann PJ, Batjer HH, Samson D, et al: Recurrent subarachnoid hemorrhage complicating a traumatic carotid-cavernous fistula. Neurosurgery 17:480-483, 1985 Fromm H, Habel 3: Angiographischen Nachweis eines sackfrrmigen Aneurysmas als Ursache einer spontanen Carotis-Sinus cavernosus-Fistel und Spontanheilung dieser Fistel nach Angiographie. Nervenarzt 36:170-172, 1965 Hamby WB: Carotid-cavernous fistula. Report of 32 surgically treated cases and suggestions for definitive operation. J Neurusurg 21:859-866. 1964 Hamby WB: Carotid Cavernous Fistula. Springfield, Ill: Charles C Thomas, 1966, 139 pp Jaeger R: Intracranial aneurysms. South Surg 15: 205-217, 1949 Kohgo T, Kowada M, Momma F, et al: [Spontaneously cured carotid-cavernous fistula verified by angiography - - report of a case.] Neural Surg 7:187-190, 1979 (Jpn) Nukui H, Shibasald T, Kaneko M, et al: Long-term observations in cases with spontaneous carotid-cavernous fistulas. Surg Neurol 21:543-552, 1984 Ohta T, Kajikawa H: [Dural arteriovenous malformation.] Neurol Med Chit 18:439-472, 1978 (Jpn) Pedersen H, Finster M: Anesthetic risk in the pregnant surgical patient. Anestheslolagy 51:439-451, 1979 Peeters FL, KrOger R: Dural and direct cavernous sinus fistulas. AJR 132:599-606, 1979 Robb JP: Neurologic complications of pregnancy. Neurology 5:679-690, 1955

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20. Saltier CH: Pulsierender exophthalmus. Handbuch der Gesamten Augenheilkunde. Berlin: Julius Springer, 1920 21. Schrader KE, Wiedenmann O: Spontanheilung einer Karotis-Kavernosus-Fistel. Kiln Monatsbl Augenheilkd 146: 826-833, 1965 22. Toya S, Shiobara R, Izumi J, et al: Spontaneous carotidcavernous fistula during pregnancy or in the postpartum stage. Report of two cases. J Neurosurg 54:252-256, 1981 23. Travers B: A case of aneurysm by anastomosis in the orbit, cured by the ligature of the common carotid artery.. Trans Med Chir SUe 2:1-16, 1809 24. Turner DM, Vangilder JC, Mojtahedi S, et al: Spontaneous intracerebral hematoma in carotid-cavernous fistula. Report of three cases. J Neurosarg 59:680-686, 1983 25. Ueland K, Metcalfe J: Circulatory changes in pregnancy. Clin Obstet Gyneeol 18 (3):41-50, 1975 26. Vifiuela F, Fox AJ, Debrun GM, et al: Spontaneous carotid-cavernous fistulas: clinical, radiological, and therapeutic considerations. Experience with 20 cases. J Neurosarg 60:976-984, 1984 27. Voigt K, Sauer M, Dichgans J: Spontaneous occlusion of a bilateral caroticocavernous fistula studied by serial angiography. Neuroradiology 2:207-211, 1971

Manuscript received June 6, 1991. Accepted in final form August 19, 1991. Address reprint requests to: Tzu-Kang Lin, M.D., Division of Neurosurgery, Department of Surgery, Chang Gung Medical College and Memorial Hospital, 199 Tung Hwa North Road, Taipei 10591, Taiwan.

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Spontaneous intracerebral hematoma from occult carotid-cavernous fistula during pregnancy and puerperium. Case report.

In most cases, intracerebral hemorrhage during pregnancy or puerperium results from cerebral aneurysms or arteriovenous malformations. The authors pre...
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