Ruptured cerebral aneurysm from choriocarcinoma

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Ruptured cerebral aneurysm from choriocarcinoma Jia Wang, Rong Wang, Jizong Zhao ⇑ Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, 6 Tiantan Xili, Dongcheng District, Beijing 100050, China

a r t i c l e

i n f o

Article history: Received 19 July 2012 Accepted 30 September 2012

Keywords: Aneurysm Brain metastasis Intracranial hemorrhage Choriocarcinoma

a b s t r a c t Gestational trophoblastic diseases include hydatidiform moles, invasive moles, choriocarcinoma, placental-site trophoblastic tumours and miscellaneous trophoblastic lesions. Choriocarcinoma is a rare disease that arises from the trophoblastic epithelium of the placenta at the beginning of pregnancy. Among the confirmed cases, 45% occur after molar pregnancy, 25% after normal pregnancy, 25% after an abortion and 5% after ectopic pregnancy. This tumour is usually diagnosed based on its histopathologic appearance and a high level of serum beta-human chorionic gonadotropin (b-HCG). Choriocarcinoma exhibits a good response to chemotherapy and radiation. However, it is also known for its tendency to spread rapidly to multiple organs, including the lungs, liver and brain. As one of the worst prognostic factors, brain metastasis complicates 3–28% of gestational choriocarcinoma cases. Increased b-HCG levels and a low serum cerebrospinal fluid b-HCG level can be strongly suggestive of intracranial choriocarcinoma, even in the absence of histopathologically proven disease. Reviewing the literature, there were 23 cases of intracerebral haemorrhage from an oncotic aneurysm as an initial presentation of choriocarcinoma. We report a further case of intracerebral haematoma secondary to oncotic aneurysm as the first presentation of metastatic choriocarcinoma with normal serum b-HCG. Ó 2013 Elsevier Ltd. All rights reserved.

1. Case report A 27-year-old woman, gravida 1, para 1, suffered from a sudden onset of headache 5 days prior to presentation. She had a history of a normal pregnancy 3 months prior to presentation. On admission, she was drowsy and had a stiff neck. A CT scan of the head without contrast showed a large intracerebral haematoma in the right temporoparietal area (Fig. 1a and b). Subsequent brain MRI revealed a distal aneurysm arising from the right middle cerebral artery (Fig. 1c and d). Digital subtraction (DS) angiography confirmed a distal middle cerebral artery aneurysm (Fig. 1e and f). She was taken for emergency decompression via a right frontotemporal craniectomy. Inspection under microscope magnification revealed a mulberry-like aneurysm on the middle cerebral artery which was clipped and removed. The aneurysm was 0.7 cm in diameter. The peripheral vasculature was relatively fragile. Examination of the tissue at high magnification showed a few tumour cells with abnormal nuclei and unclear borders. The histopathological immunohistochemical staining of the centre of the tissue showed epithelium, and it also revealed positive cytokeratin and beta-human chorionic gonadotropin (b–HCG) (Fig. 2). Her serum b–HCG was 2.92 mIU/mL (normal 8 Two or more drugs

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Table 2 Demographic and clinical data of previously reported cases of oncotic aneurysms from choriocarcinoma Case 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24

Year

Author 15

1962 1971

Vaughan and Howard Montaut et al.16

1972 1974 1975 1977 1978 1980 1985 1986 1987 1989 1990 1992 1992 1998 2007 2008 2008 2010

Stilp et al.17 Shuangshoti et al.18 Nakahara et al.19 Olmsted and Mcgee20 Weir et al.6 Weed and Hammond21 Pullar et al.22 Momma et al.23 Seigle et al.24 Noterman et al.25 Hove et al.26 Fujiwara et al.8 Giannakopoulos et al.12 Kalafut et al.3 Huang et al.9 Rocque and Baskkaya27 Chang et al.28 Zairi et al.29

2012

Present study

Age (years)

Location

Number

Treatment

Outcome

31 18 21 21 22 40 22 16 25 20 16 29 28 30 32 26 30 33 40 34 35 28 18 27

LMCA LMCA RMCA LMCA RMCA LMCA RMCA RMCA LMCA RMCA LMCA RMCA RMCA-LPCA RMCA MCA MCA MCA MCA-ACA ACA

1 1 1 1 2 1 1 1 1 1 1 1 4 1 1 1 1 Multiple 2 1 Multiple 1 3 1

autopsy excision excision excision excision excision excision operation excision

Dead (32 h) Good (4 years)

MCA-ACA MCA MCA MCA

excision excision excision chemotherapy excision excision autopsy excision excision excision excision excision

Good Dead Dead Dead (36 months) Good (12 months) Dead (1 month) Dead (72 h) Dead (23 months) Alive (7 years) Good (6 years) Dead (2 weeks) Dead Chemotherapy with EMACO Good Dead Dead Dead Chemotherapy

ACA = anterior cerebral artery, EMACO = etoposide, methotrexate, actinomycin D, cyclophosphamide, vincristine, LMCA = left middle cerebral artery, LPCA = left posterior cerebral artery, MCA = middle cerebral artery, RMCA = right middle cerebral artery.

patient with normal or abnormal pregnancy history strongly suggests the diagnosis.13 In addition, chest radiography or CT scans, PET scans and gynaecological examinations are useful in identifying the metastatic lesions. Central nervous system metastases have a good response to chemotherapy if the treatment is initiated as soon as possible. Combination chemotherapy is considered the most effective treatment regimen, producing long-term survival rates of 80%. However, cerebral metastasis remains the worst prognostic factor. Surgical treatment should only be performed in life-threatening haemorrhagic complications or to remove the tumour for patients who are resistant to chemotherapy. In this case, after surgery, the patient refused brain irradiation and received combination chemotherapy. References 1. Scully RE, Bonfiglio TA, Kurman RJ, et al. Histological typing of female genital tract tumors. WHO international histological classification of tumors. 2nd ed. New York, NY: Springer Verlag; 1994. pp. 31–8. 2. Redline RW, Abdul-Karim FW. Pathology of gestational trophoblastic disease. Semin Oncol 1995;22:96–108. 3. Kalafut M, Vinuela F, Saver JL, et al. Multiple cerebral pseudoaneurysms and hemorrhages: the expanding spectrum of metastatic cerebral choriocarcinoma. J Neuroimaging 1998;8:44–7. 4. Athanassiou A, Begent RH, Newlands ES, et al. Central nervous system metastases of choriocarcinoma. 23 years’ experience at Charing Cross Hospital. Cancer 1983;52:1728–35. 5. Lage JM, Young RH. Pathology of trophoblastic disease. In: Clement PB, Young RH, editors. Tumors and tumor-like conditions of the uterine corpus and cervix. New York, NY: Churchill Livingstone; 1993. p. 417–75. 6. Weir B, MacDonald N, Mielke B. Intracanial vascular complications of choriocarcinoma. Neurosurgery 1978;2:138–42. 7. Gurwitt LJ, Long JM, Clark RE. Cerebral metastatic choriocarcinoma: a postpartum cause of ‘‘stroke’’. Obstet Gynecol 1975;45:583–8. 8. Fujiwara T, Mino S, Nagao S, et al. Metastatic choriocarcinoma with neoplastic aneurysms cured by aneurysm resection and chemotherapy. Case report. J Neurosurg 1992;76:148–51. 9. Huang CY, Chen CA, Hsieh CY, et al. Intracerebral hemorrhage as initial presentation of gestational choriocarcinoma: a case report and literature review. Int J Gynecol Cancer 2007;17:1166–71. 10. Fadli M, Lmejjati M, Amarti A, et al. Metastatic and hemorrhagic brain arteriovenous fistulae due to a choriocarcinoma. Case report. Neurochirurgie 2002;48:39–43. doi:http://dx.doi.org/10.1016/j.jocn.2012.09.045

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