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Intracranial aneurysm due to metastatic choriocarcinoma
74
Surg Neurol 1986;25:74-6
Intracranial Aneurysm Due to Metastatic Choriocarcinoma Fumiyuki Momma, M.D., Hiroichi Beck, M.D., Toshihiko Miyamoto, M.D., and Seigo Nagao, M.D. Department of Neurological Surgery, Okayama University Medical School, Okayama, Japan
Momma F, Beck H, Miyamoto T, Nagao S. Intracranial aneurysm due to metastatic choriocarcinoma. Surg Neurol 1986;25:74-6.
The case of a woman with an episode of subarachnoid hemorrhage 2 days after delivery is presented. An emergency operation was performed, and an intracerebral clot from a ruptured aneurysm was removed. Histologic examination of the resected aneurysm showed metastasis from a choriocarcinoma to the aneurysmal wall. Six cases of intracranial aneurysm due to metastasis of choriocarcinoma are reviewed. KEY WORDS: Choriocarcinoma; aneurysm
Metastasis;
Intracranial
mass in the right frontoparietal area and a saccular aneurysm on the distal middle cerebral artery (Figure 1). X-ray examination of the chest showed no abnormal findings such as metastatic nodules. An emergency frontoparietal craniotomy was performed. After the dura mater was opened, fresh subarachnoid hemorrhage was seen. A small cortical incision was made in the p r e m o t o r area, and an intracerebral clot was removed. A saccular aneurysm, approximately 2 m m in diameter, was visible in the posteromedial portion of the clot. The parent artery was clipped and the aneurysm was resected for histologic examination. N o tumor mass was seen in the clot or in the periphery of the aneurysm. External decompression was also carried out.
Choriocarcinoma has a high predilection to metastasize by way of the bloodstream. Brain metastases of this tumor are reported to occur in approximately 2 0 % of cases [6]. The t u m o r cells have a tendency to invade the blood vessels; therefore, the incidence ofintracranial hemorrhage is greater than with other metastatic or primary brain tumors. A case of ruptured aneurysm due to metastasis of the choriocarcinoma and a review of six cases of aneurysm associated with metastatic choriocarcinoma constitute this report.
Case R e p o r t A 29-year-old woman, who had been delivered of a normal full-term infant 2 days before, was admitted immediately after an episode of subarachnoid hemorrhage on January 22, 1980. In May 1978, a benign hydatidiform mole had been removed from the uterus, and she had received chemotherapy. On physical examination she was semicomatose and had hemiparesis of the left side. Lumbar puncture yielded bloody cerebrospinal fluid. Emergency cerebral angiography revealed an avascular
She improved gradually after the operation. Postoperative cerebral angiography revealed moderate ventricular dilatation. T h e r e was no mass effect or vascular abnormalities, such as aneurysms or varices. On February 19, cranioplasty and ventriculoperitoneal shunting were performed. Despite a mild hemiparesis of the left side she became alert on February 21, and was transferred to the gynecologic service for chemotherapy on February 23, 1980. Ten days later, she suddenly died of shock due to massive intraperitoneal hemorrhage. An autopsy demonstrated numerous hemorrhagic necrotic tumor nodules on the mesentery of the small intestine. A few hemorrhagic nodules, which had ruptured and caused a massive hemorrhage, were seen in the liver. These tumor masses showed typical features of choriocarcinoma on histologic examination. T h e r e were no tumor nodules in the lungs macroscopically. Microscopic examination of the resected aneurysm showed typical trophoblasts invading the intima of the aneurysmal wall. T u m o r cells also invaded the media with interruption of the internal elastic m e m b r a n e (Figure 2).
Discussion Address reprint requests to: Fumiyuki Momma, M.D., Department
of Neurological Surgery, Okayama University Medical School, 2-5-1 Shikato-cho, Okayama 700, Japan. (<~ 1986 by Elsevier Science Publishing Co., Inc.
Metastatic choriocarcinoma has an inherent tendency to cause hemorrhage. Teacher [5] first described the mechanism of bleeding in cerebral choriocarcinomas. H e em0090-3019/86/$3.50
Aneurysm and Choriocarcinoma
Figure 1. Right carotid angiogram showing a saccular aneurysm on the operculofrontal artery and an avascular mass in the frontoparietal area (arrows).
phasized rupture of neoplastic aneurysms or varices as a cause of intracerebral hemorrhage. Six patients with cerebral aneurysms associated with metastatic choriocarcinoma reported in the literature were all female, their ages ranging from 18 to 29 years
Figure 2. (A) Photomicrograph of the resected aneurysm showing the atypical trophoblasts invading the arterial intima. (Hematoxylin and eosin, x 200.) (B) Choriocarcinoma invading the media with interruption of the internal elastic membrane (arrow). (Van Gieson Elastica, x I00.)
(A)
Surg Neurol 1986;25:74-6
75
(Table 1). Location of the aneurysm was on the distal middle cerebral artery in all cases. The shape of the aneurysm was fusiform in four cases, and saccular in our case. The location and the shape of the aneurysms are in striking contrast with those of congenital aneurysms, and resemble those of mycotic aneurysms. The initial symptoms were those of subarachnoid hemorrhage in all cases. An intracerebral h e m a t o m a was found in four out of six cases. Cerebral choriocarcinoma should be considered in the differential diagnosis in a woman of child-bearing age with an episode of subarachnoid hemorrhage. Histologic examination of the aneurysm showed proliferation of the tumor cells in the intima [ 1 - 3 ], invasion
(B)
76
Surg N e u r o l 1986;25:74-6
M o m m a et al
Table 1. Reported Cases of Intracranial Aneurysm Due to Metastasis of Choriocarcinoma Case
Author
Age/sex
Onset
1 2 3 4 5 6
Montaut et al Montaut et at Stilp et al Nakahara et al Bryce et al Presented case
21/F 18/F 22/F 22/F 25/F
SAH SAH SAH SAH SAH SAH
29/F
Location of aneurysm MCA, MCA, MCA, MCA, MCA, MCA,
distal distal distal distal distal distal
Shape of aneurysm
Intracerebral hematoma
Operation
Fusiform Fusiform -Fusiform Fusiform Saccular
(- ) (+) (+ ) (- ) (+ ) (+ )
(+ (+ (+ (+ (+ (+
) ) ) ) ) )
Abbreviations: F, female; SAH, subarachnoid hemorrhage; MCA, middle cerebral artery.
to the media with interruption of the internal elastic m e m b r a n e [ 1,3], and neoplastic dissection of the media from the adventitia [1]. These features show the vascular-invasive capacity of the choriocarcinoma. In the management of these neoplastic aneurysms, an emergency craniotomy should be selected in the patient who has an intracerebral h e m a t o m a from a ruptured aneurysm. C h e m o t h e r a p y and radiation therapy are r e c o m m e n d e d for patients who have an aneurysm without a tumor mass or a clot. Four out of six cases improved neurologically within a few days after the surgical treatment. Longterm postoperative course was excellent in two [1,3] out of six cases. They were alive 3 years after the operation. The other four cases died within 3 months. The outcome of these patients seemed to be determined by the site and n u m b e r of metastases including the brain. A combination of chemotherapy, with methotrexate and actinomycin-D, and radiotherapy to the whole brain is reco m m e n d e d after surgical treatment [1,3,4].
We would like to thank Mr. Hideki Wakimoto for manuscript preparation.
References 1. Bryce W, Nell M, Bruce M. lntracranial vascular complications of choriocarcinoma. Neurosurgery 1978;2:138-42. 2. Montant J, Hepner H, Tridon P, Picard L, Floquet J, Lepoire J. Aspects pseudovasculaires des mdtastases intracraniennes des choriodpithdliomes. Neurochirurgie 1971 ; 17:119-28. 3. Nakahara T, Nosaka N, Kinoshita K, Matsukado Y. Subarachnoid hemorrhage and aneurysmal change of cerebral arteries due to metastases of choriocarcinoma. N o Shinkei Geka 1975;3:777-82. 4. Stilp TJ, Buoy PC, Brewer JI. Cure of metastatic choriocarcinoma of the brain. J A M A 1972;22l:276-9. 5. Teacher JH. On chorionepithelioma and the occurrence of chorionepitheliomatous and hydatidiform mole-like structures in teratomata: a pathological and clinical study. J Obstet Gynaecol Br Emp 1903;4:1-64. 6. Vaughan HG, Howard RG. intracranial hemorrhage due to metastatic choriocarcinoma. Neurology 1962;12:771-7.
Surg Neurol 1986;25:74-6
Intracranial Aneurysm Due to Metastatic Choriocarcinoma Fumiyuki Momma, M.D., Hiroichi Beck, M.D., Toshihiko Miyamoto, M.D., and Seigo Nagao, M.D. Department of Neurological Surgery, Okayama University Medical School, Okayama, Japan
Momma F, Beck H, Miyamoto T, Nagao S. Intracranial aneurysm due to metastatic choriocarcinoma. Surg Neurol 1986;25:74-6.
The case of a woman with an episode of subarachnoid hemorrhage 2 days after delivery is presented. An emergency operation was performed, and an intracerebral clot from a ruptured aneurysm was removed. Histologic examination of the resected aneurysm showed metastasis from a choriocarcinoma to the aneurysmal wall. Six cases of intracranial aneurysm due to metastasis of choriocarcinoma are reviewed. KEY WORDS: Choriocarcinoma; aneurysm
Metastasis;
Intracranial
mass in the right frontoparietal area and a saccular aneurysm on the distal middle cerebral artery (Figure 1). X-ray examination of the chest showed no abnormal findings such as metastatic nodules. An emergency frontoparietal craniotomy was performed. After the dura mater was opened, fresh subarachnoid hemorrhage was seen. A small cortical incision was made in the p r e m o t o r area, and an intracerebral clot was removed. A saccular aneurysm, approximately 2 m m in diameter, was visible in the posteromedial portion of the clot. The parent artery was clipped and the aneurysm was resected for histologic examination. N o tumor mass was seen in the clot or in the periphery of the aneurysm. External decompression was also carried out.
Choriocarcinoma has a high predilection to metastasize by way of the bloodstream. Brain metastases of this tumor are reported to occur in approximately 2 0 % of cases [6]. The t u m o r cells have a tendency to invade the blood vessels; therefore, the incidence ofintracranial hemorrhage is greater than with other metastatic or primary brain tumors. A case of ruptured aneurysm due to metastasis of the choriocarcinoma and a review of six cases of aneurysm associated with metastatic choriocarcinoma constitute this report.
Case R e p o r t A 29-year-old woman, who had been delivered of a normal full-term infant 2 days before, was admitted immediately after an episode of subarachnoid hemorrhage on January 22, 1980. In May 1978, a benign hydatidiform mole had been removed from the uterus, and she had received chemotherapy. On physical examination she was semicomatose and had hemiparesis of the left side. Lumbar puncture yielded bloody cerebrospinal fluid. Emergency cerebral angiography revealed an avascular
She improved gradually after the operation. Postoperative cerebral angiography revealed moderate ventricular dilatation. T h e r e was no mass effect or vascular abnormalities, such as aneurysms or varices. On February 19, cranioplasty and ventriculoperitoneal shunting were performed. Despite a mild hemiparesis of the left side she became alert on February 21, and was transferred to the gynecologic service for chemotherapy on February 23, 1980. Ten days later, she suddenly died of shock due to massive intraperitoneal hemorrhage. An autopsy demonstrated numerous hemorrhagic necrotic tumor nodules on the mesentery of the small intestine. A few hemorrhagic nodules, which had ruptured and caused a massive hemorrhage, were seen in the liver. These tumor masses showed typical features of choriocarcinoma on histologic examination. T h e r e were no tumor nodules in the lungs macroscopically. Microscopic examination of the resected aneurysm showed typical trophoblasts invading the intima of the aneurysmal wall. T u m o r cells also invaded the media with interruption of the internal elastic m e m b r a n e (Figure 2).
Discussion Address reprint requests to: Fumiyuki Momma, M.D., Department
of Neurological Surgery, Okayama University Medical School, 2-5-1 Shikato-cho, Okayama 700, Japan. (<~ 1986 by Elsevier Science Publishing Co., Inc.
Metastatic choriocarcinoma has an inherent tendency to cause hemorrhage. Teacher [5] first described the mechanism of bleeding in cerebral choriocarcinomas. H e em0090-3019/86/$3.50
Aneurysm and Choriocarcinoma
Figure 1. Right carotid angiogram showing a saccular aneurysm on the operculofrontal artery and an avascular mass in the frontoparietal area (arrows).
phasized rupture of neoplastic aneurysms or varices as a cause of intracerebral hemorrhage. Six patients with cerebral aneurysms associated with metastatic choriocarcinoma reported in the literature were all female, their ages ranging from 18 to 29 years
Figure 2. (A) Photomicrograph of the resected aneurysm showing the atypical trophoblasts invading the arterial intima. (Hematoxylin and eosin, x 200.) (B) Choriocarcinoma invading the media with interruption of the internal elastic membrane (arrow). (Van Gieson Elastica, x I00.)
(A)
Surg Neurol 1986;25:74-6
75
(Table 1). Location of the aneurysm was on the distal middle cerebral artery in all cases. The shape of the aneurysm was fusiform in four cases, and saccular in our case. The location and the shape of the aneurysms are in striking contrast with those of congenital aneurysms, and resemble those of mycotic aneurysms. The initial symptoms were those of subarachnoid hemorrhage in all cases. An intracerebral h e m a t o m a was found in four out of six cases. Cerebral choriocarcinoma should be considered in the differential diagnosis in a woman of child-bearing age with an episode of subarachnoid hemorrhage. Histologic examination of the aneurysm showed proliferation of the tumor cells in the intima [ 1 - 3 ], invasion
(B)
76
Surg N e u r o l 1986;25:74-6
M o m m a et al
Table 1. Reported Cases of Intracranial Aneurysm Due to Metastasis of Choriocarcinoma Case
Author
Age/sex
Onset
1 2 3 4 5 6
Montaut et al Montaut et at Stilp et al Nakahara et al Bryce et al Presented case
21/F 18/F 22/F 22/F 25/F
SAH SAH SAH SAH SAH SAH
29/F
Location of aneurysm MCA, MCA, MCA, MCA, MCA, MCA,
distal distal distal distal distal distal
Shape of aneurysm
Intracerebral hematoma
Operation
Fusiform Fusiform -Fusiform Fusiform Saccular
(- ) (+) (+ ) (- ) (+ ) (+ )
(+ (+ (+ (+ (+ (+
) ) ) ) ) )
Abbreviations: F, female; SAH, subarachnoid hemorrhage; MCA, middle cerebral artery.
to the media with interruption of the internal elastic m e m b r a n e [ 1,3], and neoplastic dissection of the media from the adventitia [1]. These features show the vascular-invasive capacity of the choriocarcinoma. In the management of these neoplastic aneurysms, an emergency craniotomy should be selected in the patient who has an intracerebral h e m a t o m a from a ruptured aneurysm. C h e m o t h e r a p y and radiation therapy are r e c o m m e n d e d for patients who have an aneurysm without a tumor mass or a clot. Four out of six cases improved neurologically within a few days after the surgical treatment. Longterm postoperative course was excellent in two [1,3] out of six cases. They were alive 3 years after the operation. The other four cases died within 3 months. The outcome of these patients seemed to be determined by the site and n u m b e r of metastases including the brain. A combination of chemotherapy, with methotrexate and actinomycin-D, and radiotherapy to the whole brain is reco m m e n d e d after surgical treatment [1,3,4].
We would like to thank Mr. Hideki Wakimoto for manuscript preparation.
References 1. Bryce W, Nell M, Bruce M. lntracranial vascular complications of choriocarcinoma. Neurosurgery 1978;2:138-42. 2. Montant J, Hepner H, Tridon P, Picard L, Floquet J, Lepoire J. Aspects pseudovasculaires des mdtastases intracraniennes des choriodpithdliomes. Neurochirurgie 1971 ; 17:119-28. 3. Nakahara T, Nosaka N, Kinoshita K, Matsukado Y. Subarachnoid hemorrhage and aneurysmal change of cerebral arteries due to metastases of choriocarcinoma. N o Shinkei Geka 1975;3:777-82. 4. Stilp TJ, Buoy PC, Brewer JI. Cure of metastatic choriocarcinoma of the brain. J A M A 1972;22l:276-9. 5. Teacher JH. On chorionepithelioma and the occurrence of chorionepitheliomatous and hydatidiform mole-like structures in teratomata: a pathological and clinical study. J Obstet Gynaecol Br Emp 1903;4:1-64. 6. Vaughan HG, Howard RG. intracranial hemorrhage due to metastatic choriocarcinoma. Neurology 1962;12:771-7.