Neuroradiology 14, 113-116 (1977)
Nearoradiologg
© by Springer-Verlag 1977
Angiographic Manifestations of Intracerebral Cavernous Hemangioma Y. Numaguchi,* M. Fukui,** E. Miyake,** T. Kishikawa,* J. Ikeda,* K. Matsuura,* M. Tomonaga,*** and K. Kitamura** Departments of Radiology* and Neurosurgery**, Kyushu University Hospital, Fukuoka and Neurosurgery***, Fukuoka University Hospital, Fukuoka, Japan
Summary. A case of intraventricular cavernous hemangioma is reported, with a review of the literature, especially that dealing with angiography of intracerebral lesions. Dense venous poolings and a localized area of venous stain were observed in this case.
Key words: Cerebral cavernous hemangioma Cavernoma - Cerebral angiography.
Intracerebral cavernous hemangiomas, or "cavernomas", are rare. Voigt and Yasargil [15] recently reviewed 164 cases of intracranial cavernous hemangiomas reported in the literature, for their incidence, pathology, location, diagnosis, clinical features and treatment. The angiograms were described in only 31 cases in 17 reports. In 20 cases the angiograms were normal or showed avascular masses; in the remainder, their findings were nonspecific. Reported here is a patient with a huge cavernous hemangioma of the lateral ventricle. On cerebral angiography the large avascular mass contained small venous poolings of contrast medium. To our knowledge, this finding has never been reported before.
ity with increased deep tendon reflexes, incoordination of the left extremities and he was unsteady sitting, standing und walking. Skull radiographs revealed several small calcifications just to the right of the midline, and enlargement of the sella (Fig. 1). Brain scintigraphy disclosed a large area of high activity on the right, near the midline. Right carotid and left vertebral angiography revealed displacement of the midline structures to the left, especially in the venous phase, and the internal cerebral vein was markedly depressed. The asceding branches of the right middle cerebral artery were stretched, particularly in the parietal region (Figs. 2 and 3). The findings suggested a large mass in the right lateral ventricle. No large feeding
Case Report A man, aged 43, had had a hearing impairment and tinnitus on the left for six years before admission. During the year prior to admission, there was clumsiness of the left lower extremity and his walking became unsteady. Neurological examination on admission revealed bilateral papilledema, left hearing impairment, muscle weakness of the left lower extrem-
Fig. 1. Skull radiograph showing several small calcifications (arrows) and an enlarged sella with demineralization of the dorsum sellae
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Y. Numaguchi et al.: Angiographic Manifestations of Intracerebral Cavernous Hemangioma
Fig. 2 a and b Carotid angiograms, arterial phase, demonstrate very minimal midline shift of pericallosal artery. The sylvian triangle is displaced anteriorly and there is stretching of branches of middle cerebral artery in parietal region
Fig. 3 a and b. Carotid angiogram, venous phase, shows marked shift of internal cerebral vein to left, a localized area of venous stain (large arrows) and venous poolings of contrast medium (small arrows)
artery was observed but a small venous stain and small dense venous poolings in the inferior aspect of the presumed mass were well demonstrated (Fig. 3 a and b). At operation a huge nodular and encapsulated tumor was found arising from the floor of the trigone of the right lateral ventricle. Histologically the tumor was a cavernous hemangioma. The patient is alive five years after operation, but has had left hemiplegia and left homonymous hemianopia as postoperative sequelae.
Discussion In their extensive review of the world literature on intracranial cavernous hemangiomas, Voigt and Yasargil [15] described several features. There was no sex or age predominance. In 126 of 164 cases (76.8%) the cavernomas were supratentorial; in 34 (20.7%), infratentorial; and in 4 (2.5%), they were multiple in various locations. In 4 cases (2.5%), the lesions were intraventricular. Various symptoms
Y. Numaguchi et al.: Angiographic Manifestations of Intracerebral Cavernous Hemangioma
115
Fig. 4. Histological section shows tumor tissue composed of numerous vascular channels intersected by hyalinized connective tisst~_~ (H & E × 120)
were encountered among the Patients. Macroscopic calcifications were observed in 18 cases (11%). Among 31 cases ( 1 8 . 9 % ) w i t h cerebral angiography, 20 (12.2%) were normal or had avascular masses. In the remainder there was an assortment of angiographic findings. They included cases histologically unverified, and those with extracerebral lesions such as in the orbit, at the base ef the skull and tentorium. We reviewed 17 cases in 12 reports [2, 4-10, 12-15], and our own in which the lesions were strictly intracerebral and whose angiographic and histological descriptions were included. There is general agreement that, unlike most of the cases of arteriovenous malformations, no large feeding arteries are observed with intracerebral cavernomas. Angiography was either normal or suggestive of a mass effect with arterial displacement in 10 of the 17 cases. In one of three histologically verified intracerebral cavernomas, Krayenbiihl and Siebenmann [9] found tiny strands of contrast medium in an avascular mass in the capillary and venous phases. Their description of the contrast medium in the venous phase does not appear to indicate the presence of "venous lakes' or "venous poolings" as seen in our case.
Jonutis et al. [7] reported a case of cavernoma with a tumor stain in the parietal region, containing irregular vessels which were supplied by an enlarged posterior choroidal artery. Segall et al. [14] described a case with "vague puddling of contrast material" in a large avascular mass. Bogren et al. [4] referred to two cases; Roberson et al. [10] to one, when stating that an enlarged draining vein and/or early venous filling around an avascular mass might be features of cavernoma. Voigt and Yasargil [15] reported a case in which angiographic was normal initially, and in which an avascular mass was noted on the fifth examination. They opined that the angiographic findings with cavernomas depend upon when angiography is performed in relation to hemorrhage. In the case we report here, though dense venous poolings and a localized area of venous stain were noted, we could not detect large draining veins or early venous filling. Bartley and Wickbom [3] and Abrams et al. [1] reported venous poolings of contrast medium in some of the soft tissue cavernomas, and in most of the liver cavernomas. If one sees this finding in a mass on cerebral angiography, the possibility of a cavernoma must be strongly considered. The tech-
116
Y. Numaguchi et al.: Angiographic Manifestations of Intracerebral Cavernous Hemangioma
nic of p r o l o n g e d i n j e c t i o n of c o n t r a s t m e d i u m to d e m o n s t r a t e c a v e r n o u s h e m a n g i o m a as s h o w n b y R o s e n b a u m et al. [11] with calvarial lesions m a y also b e useful for i n t r a c e r e b r a l lesions.
Acknowledgement: We are grateful to Walter J. Russell, M.D., Radiation Effect Research Foundation, Hiroshima, Japan for editing this manuscript. We thank Miss Fusae Aramaki for her secretarial assistance.
References 1. Abrams, R.M., Beranbaum, E.R., Santos, J.S., Lipson, J.: Angiographic features of cavernous hemangioma of liver. Radiology 92, 308-312 (1969) 2. Aoki, H., Ogashiwa, M., Wakutani, K., Ito, E.: Cavernous hemangioma of the cerebral hemisphere. A case report. Brain and Nerve (Jap.) 23, 927-932 (1971) 3. Bartley, O., Wickbom, I.: Angiography in soft tissue hemangiomas. Acta radiol. (Diagn.) 5I, 81-94 (1959) 4. Bogren, H., Svalander, C., Wickbom, I.: Angiography in intracranial cavernous hemangiomas. Acta radiol. (Diagn.) 10, 81-89 (1970) 5. Jain, K.K.: Intraventricular cavernous hemangioma of the lateral ventricle. Case report. J. Neurosurg. 24, 762-764 (1966) 6. Jain, K.K., Robertson, E." Recurrence of an excised cavernous hemangioma in the opposite cerebral hemisphere. J. Neurosurg. 33, 453-456 (1970) 7. Jonutis, A.J., Sondheimer, F.K., Klein, H.Z., Wise, B.L.: Intracerebral cavernous hemangioma with angiographically demonstrated pathologic vasculature. Neuroradiology 3, 57-63 (1971)
8. Kamrin, R.B., Buchsbaum, H.W.: Large vascular malformations of the brain not visualized by serial angiography. Arch. Neurol. (Chicago) 13, 413-420 (1965) 9. Krayenbiihl, H., Siebenmann, R.: Small vascular malformations as a cause of primary intracerebral hemorrhage. J. Neurosurg. 22, 7-20 (1965) 10. Roberson, G.H., Kase, C. S., Wolpow, E.R.: Teleangiectasis and cavernous angiomas of the brain stem: "Cryptic" vascular malformations. Neuroradiology 8, 83-89 (1974) 11. Rosenbaum, A.E., Rossi, P., Schechter, M.M., Sheehan, J.P.: Angiography of haemangiomata of the calvarium. Brit. J. Radiol. 42, 682~587 (1969) 12. Runnels, J.B., Gifford, D. B., Forsberg, P. L., Hanbery, J. W.: Dense calcification in a large cavernous angioma: Case report. J. Neurosurg. 30, 293-298 (1969) 13. Schneider, R.C., Liss, L.: Cavernous hemangiomas of the cerebral hemispheres. J. Neurosurg. 15, 392-399 (1958) 14. SegaU, H.D., Segal H.L., Teal, J.S., Rumbaugh, C.L., Bergeron, R.T.: Calcifying cerebral cavernous hemangioma with brain scan and angiographic findings. Neuroradiology 7, 133-138 (1974) 15. Voigt, K., Yasargil, M. G.: Cerebral cavernous haemangiomas or cavernomas. Neurochirurgia 19, 59-68 (1976) Received: June 1, 1977
Yuji Numaguchi, M.D. Neuroradiology section Department of Radiology Kyushu University Hospital 1-1, Maidashi 3 Higashi-ku Fukuoka 812, Japan
Nearoradiologg
© by Springer-Verlag 1977
Angiographic Manifestations of Intracerebral Cavernous Hemangioma Y. Numaguchi,* M. Fukui,** E. Miyake,** T. Kishikawa,* J. Ikeda,* K. Matsuura,* M. Tomonaga,*** and K. Kitamura** Departments of Radiology* and Neurosurgery**, Kyushu University Hospital, Fukuoka and Neurosurgery***, Fukuoka University Hospital, Fukuoka, Japan
Summary. A case of intraventricular cavernous hemangioma is reported, with a review of the literature, especially that dealing with angiography of intracerebral lesions. Dense venous poolings and a localized area of venous stain were observed in this case.
Key words: Cerebral cavernous hemangioma Cavernoma - Cerebral angiography.
Intracerebral cavernous hemangiomas, or "cavernomas", are rare. Voigt and Yasargil [15] recently reviewed 164 cases of intracranial cavernous hemangiomas reported in the literature, for their incidence, pathology, location, diagnosis, clinical features and treatment. The angiograms were described in only 31 cases in 17 reports. In 20 cases the angiograms were normal or showed avascular masses; in the remainder, their findings were nonspecific. Reported here is a patient with a huge cavernous hemangioma of the lateral ventricle. On cerebral angiography the large avascular mass contained small venous poolings of contrast medium. To our knowledge, this finding has never been reported before.
ity with increased deep tendon reflexes, incoordination of the left extremities and he was unsteady sitting, standing und walking. Skull radiographs revealed several small calcifications just to the right of the midline, and enlargement of the sella (Fig. 1). Brain scintigraphy disclosed a large area of high activity on the right, near the midline. Right carotid and left vertebral angiography revealed displacement of the midline structures to the left, especially in the venous phase, and the internal cerebral vein was markedly depressed. The asceding branches of the right middle cerebral artery were stretched, particularly in the parietal region (Figs. 2 and 3). The findings suggested a large mass in the right lateral ventricle. No large feeding
Case Report A man, aged 43, had had a hearing impairment and tinnitus on the left for six years before admission. During the year prior to admission, there was clumsiness of the left lower extremity and his walking became unsteady. Neurological examination on admission revealed bilateral papilledema, left hearing impairment, muscle weakness of the left lower extrem-
Fig. 1. Skull radiograph showing several small calcifications (arrows) and an enlarged sella with demineralization of the dorsum sellae
114
Y. Numaguchi et al.: Angiographic Manifestations of Intracerebral Cavernous Hemangioma
Fig. 2 a and b Carotid angiograms, arterial phase, demonstrate very minimal midline shift of pericallosal artery. The sylvian triangle is displaced anteriorly and there is stretching of branches of middle cerebral artery in parietal region
Fig. 3 a and b. Carotid angiogram, venous phase, shows marked shift of internal cerebral vein to left, a localized area of venous stain (large arrows) and venous poolings of contrast medium (small arrows)
artery was observed but a small venous stain and small dense venous poolings in the inferior aspect of the presumed mass were well demonstrated (Fig. 3 a and b). At operation a huge nodular and encapsulated tumor was found arising from the floor of the trigone of the right lateral ventricle. Histologically the tumor was a cavernous hemangioma. The patient is alive five years after operation, but has had left hemiplegia and left homonymous hemianopia as postoperative sequelae.
Discussion In their extensive review of the world literature on intracranial cavernous hemangiomas, Voigt and Yasargil [15] described several features. There was no sex or age predominance. In 126 of 164 cases (76.8%) the cavernomas were supratentorial; in 34 (20.7%), infratentorial; and in 4 (2.5%), they were multiple in various locations. In 4 cases (2.5%), the lesions were intraventricular. Various symptoms
Y. Numaguchi et al.: Angiographic Manifestations of Intracerebral Cavernous Hemangioma
115
Fig. 4. Histological section shows tumor tissue composed of numerous vascular channels intersected by hyalinized connective tisst~_~ (H & E × 120)
were encountered among the Patients. Macroscopic calcifications were observed in 18 cases (11%). Among 31 cases ( 1 8 . 9 % ) w i t h cerebral angiography, 20 (12.2%) were normal or had avascular masses. In the remainder there was an assortment of angiographic findings. They included cases histologically unverified, and those with extracerebral lesions such as in the orbit, at the base ef the skull and tentorium. We reviewed 17 cases in 12 reports [2, 4-10, 12-15], and our own in which the lesions were strictly intracerebral and whose angiographic and histological descriptions were included. There is general agreement that, unlike most of the cases of arteriovenous malformations, no large feeding arteries are observed with intracerebral cavernomas. Angiography was either normal or suggestive of a mass effect with arterial displacement in 10 of the 17 cases. In one of three histologically verified intracerebral cavernomas, Krayenbiihl and Siebenmann [9] found tiny strands of contrast medium in an avascular mass in the capillary and venous phases. Their description of the contrast medium in the venous phase does not appear to indicate the presence of "venous lakes' or "venous poolings" as seen in our case.
Jonutis et al. [7] reported a case of cavernoma with a tumor stain in the parietal region, containing irregular vessels which were supplied by an enlarged posterior choroidal artery. Segall et al. [14] described a case with "vague puddling of contrast material" in a large avascular mass. Bogren et al. [4] referred to two cases; Roberson et al. [10] to one, when stating that an enlarged draining vein and/or early venous filling around an avascular mass might be features of cavernoma. Voigt and Yasargil [15] reported a case in which angiographic was normal initially, and in which an avascular mass was noted on the fifth examination. They opined that the angiographic findings with cavernomas depend upon when angiography is performed in relation to hemorrhage. In the case we report here, though dense venous poolings and a localized area of venous stain were noted, we could not detect large draining veins or early venous filling. Bartley and Wickbom [3] and Abrams et al. [1] reported venous poolings of contrast medium in some of the soft tissue cavernomas, and in most of the liver cavernomas. If one sees this finding in a mass on cerebral angiography, the possibility of a cavernoma must be strongly considered. The tech-
116
Y. Numaguchi et al.: Angiographic Manifestations of Intracerebral Cavernous Hemangioma
nic of p r o l o n g e d i n j e c t i o n of c o n t r a s t m e d i u m to d e m o n s t r a t e c a v e r n o u s h e m a n g i o m a as s h o w n b y R o s e n b a u m et al. [11] with calvarial lesions m a y also b e useful for i n t r a c e r e b r a l lesions.
Acknowledgement: We are grateful to Walter J. Russell, M.D., Radiation Effect Research Foundation, Hiroshima, Japan for editing this manuscript. We thank Miss Fusae Aramaki for her secretarial assistance.
References 1. Abrams, R.M., Beranbaum, E.R., Santos, J.S., Lipson, J.: Angiographic features of cavernous hemangioma of liver. Radiology 92, 308-312 (1969) 2. Aoki, H., Ogashiwa, M., Wakutani, K., Ito, E.: Cavernous hemangioma of the cerebral hemisphere. A case report. Brain and Nerve (Jap.) 23, 927-932 (1971) 3. Bartley, O., Wickbom, I.: Angiography in soft tissue hemangiomas. Acta radiol. (Diagn.) 5I, 81-94 (1959) 4. Bogren, H., Svalander, C., Wickbom, I.: Angiography in intracranial cavernous hemangiomas. Acta radiol. (Diagn.) 10, 81-89 (1970) 5. Jain, K.K.: Intraventricular cavernous hemangioma of the lateral ventricle. Case report. J. Neurosurg. 24, 762-764 (1966) 6. Jain, K.K., Robertson, E." Recurrence of an excised cavernous hemangioma in the opposite cerebral hemisphere. J. Neurosurg. 33, 453-456 (1970) 7. Jonutis, A.J., Sondheimer, F.K., Klein, H.Z., Wise, B.L.: Intracerebral cavernous hemangioma with angiographically demonstrated pathologic vasculature. Neuroradiology 3, 57-63 (1971)
8. Kamrin, R.B., Buchsbaum, H.W.: Large vascular malformations of the brain not visualized by serial angiography. Arch. Neurol. (Chicago) 13, 413-420 (1965) 9. Krayenbiihl, H., Siebenmann, R.: Small vascular malformations as a cause of primary intracerebral hemorrhage. J. Neurosurg. 22, 7-20 (1965) 10. Roberson, G.H., Kase, C. S., Wolpow, E.R.: Teleangiectasis and cavernous angiomas of the brain stem: "Cryptic" vascular malformations. Neuroradiology 8, 83-89 (1974) 11. Rosenbaum, A.E., Rossi, P., Schechter, M.M., Sheehan, J.P.: Angiography of haemangiomata of the calvarium. Brit. J. Radiol. 42, 682~587 (1969) 12. Runnels, J.B., Gifford, D. B., Forsberg, P. L., Hanbery, J. W.: Dense calcification in a large cavernous angioma: Case report. J. Neurosurg. 30, 293-298 (1969) 13. Schneider, R.C., Liss, L.: Cavernous hemangiomas of the cerebral hemispheres. J. Neurosurg. 15, 392-399 (1958) 14. SegaU, H.D., Segal H.L., Teal, J.S., Rumbaugh, C.L., Bergeron, R.T.: Calcifying cerebral cavernous hemangioma with brain scan and angiographic findings. Neuroradiology 7, 133-138 (1974) 15. Voigt, K., Yasargil, M. G.: Cerebral cavernous haemangiomas or cavernomas. Neurochirurgia 19, 59-68 (1976) Received: June 1, 1977
Yuji Numaguchi, M.D. Neuroradiology section Department of Radiology Kyushu University Hospital 1-1, Maidashi 3 Higashi-ku Fukuoka 812, Japan
