Bilateral spontaneous carotid-cavernous fistula(CCF) presenting as painful ophthalmoplegia -Two cases-

J Korean Neurol Assoc > Volume 15(5); 1997 > Article

Journal of the Korean Neurological Association 1997;15(5): 1148-1154.

동통성 안근마비를 나타낸 양측성 자발성 경동맥해면정맥동루 2례

전성만, 박경원, 차재관, 김상호, 김재우, 최순섭

동아대학교 의대 신경과. 진단방사선과

Bilateral spontaneous carotid-cavernous fistula(CCF) presenting as painful ophthalmoplegia -Two cases-

Sung Man Jun, M.D. , Kyung Won Park, M.D. , Jae Kwan Gha, M.D. , Sang Ho Kim, M.D. , Jae Woo Kim , M.D. , Sun Sep Choi, M.D.*

Dept of Neurology and neuroradiology*, Dong-A Univ. College of Medicine

Abstract

Carotid cavernous fistula(CCF) is a syndrome in which arteriovenous sinus shunts exist between the carotid artery and the cavernous sinus. These shunts have very wide spectrums in pathogenesis, angiografic findings, hemodynamics and teatment. Spontaneous dural CCF is dural vascular malformations that usually runs a benign course with variable clinical findings. We experienced two case of bilateral spontaneous CCF presenting as painful ophthalmoplegia. In the first case, a 53-years old hypertensive, diabetic woman had previous history of unilateral painful ophthalmoplegia eight months before admission and as partially recovered. On examination, she had slowly progressive bilateral chemosis ,exophthalmos, and sixth nerve palsy. MRI showed abnormal vessels in the cavernous sinus from prominent superior ophthalmic vein. Angiographic findings showed fistulas of both external carotid and left internal carotid arteries draining into the cavernous sinus. We had multiple trials of transarterial and transvenous embolization with gelfom material or platinum coils with partial improvement of chemosis and exophthalmos. In the second case, a 45-years old woman noted slowly progressive headache, ptosis, dilatation of pupil in the left side, and diplopia suggesting paralysis of the left inferior rectus muscle. MRI showed signal voids in the cavernous sinus. Angiographic findings showed fistulars of the right and left internal carotid arteries draining into the cavernous sinus. CCF should be consider as a underlying cause in patient presenting as painful ophthalmoplegia. We report two cases of bilateral spontaneous CCF presenting as painful ophthalmoplegia.