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CASE REPORT
Year : 2007  |  Volume : 55  |  Issue : 2  |  Page : 151-153

Late intrathoracic relapse of pineal germinoma connected to intraspinal canal


Department of Neurosurgery, Kitasato University School of Medicine, Sagamihara, Kanagaw, Japan

Date of Acceptance15-Jan-2007

Correspondence Address:
Satoshi Utsuki
Department of Neurosurgery, Kitasato University School of Medicine, 1-15- 1 Kitasato, Sagamihara, Kanagawa - 228-8555
Japan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.32788

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 » Abstract 

Extraneural metastases of intracranial germinoma are rarely reported. The authors describe the first case of metastatic lung germinoma of the thoracic spine. A 27-year-old man presented with right shoulder pain and right upper limb weakness. He had a history of repetitive radiation therapy - nine (whole-abdomen; 15Gy), 12 (whole brain; 30Gy, whole spine 42Gy) and 14 years ago (local; 32Gy) - for abdominal metastasis, temporal and fourth ventricle metastasis and spinal dissemination and metastatic pineal germinoma, respectively. Magnetic resonance imaging revealed a lung mass invading the thoracic spine that was diagnosed as a germinoma by tumor biopsy. He was treated by irradiation with 54Gy and two cycles of chemotherapy with cisplatin and etoposide. He did not have any sign of tumor eight years later.


Keywords: Extraneural metastasis, germinoma, lung, thoracic spine


How to cite this article:
Utsuki S, Oka H, Sagiuchi T, Fujii K. Late intrathoracic relapse of pineal germinoma connected to intraspinal canal. Neurol India 2007;55:151-3

How to cite this URL:
Utsuki S, Oka H, Sagiuchi T, Fujii K. Late intrathoracic relapse of pineal germinoma connected to intraspinal canal. Neurol India [serial online] 2007 [cited 2019 Nov 14];55:151-3. Available from: http://www.neurologyindia.com/text.asp?2007/55/2/151/32788



 » Introduction Top


Intracranial germinomas are highly radiosensitive and the 10-year survival rates of patients following radiotherapy have been reported to be 79 to 100%.[1] Although distant metastasis of intracranial germinoma is rare, metastases to bone,[2] spinal epidural space[3] and shunt-related peritoneal deposits from primary tumor were reported.[4],[5] These extraneural metastases of intracranial germinoma were controlled with adjuvant therapy.[4],[6] We describe a case of germinoma metastatic to the lung via thoracic spine and discuss the importance of long-term follow-up.


 » Case Report Top


First admission

In April 1983, a 13-year-old boy presented with a one-month history of double vision, headache, nausea and vomiting. Computed tomography (CT) revealed a calcified mass lesion in the pineal region with hydrocephalus [Figure - 1]. Ventricle-peritoneal (VP) shunt was performed for hydrocephalus. Tumor cells were not detected in cerebrospinal fluid (CSF) cytology. After shunt operation, it was diagnosed as nonpathological germinoma following tumor ablation with a 20Gy irradiation. The patient was treated with local irradiation using a dose of 32Gy, in daily fractions of 200cGy. No tumor was visible on CT after irradiation. During the hospital stay, the beta-human chorionic gonadotropin (beta-HCG), alpha-fetoprotein (AFP), carcinoembryonic antigen (CEA) titers in serum or cerebrospinal fluid (CSF) were not measured.

Second admission

In April 1985, the patient presented with a two-month-history of both lower-limbs hypoaesthesia, absence of deep tendon reflex and left shoulder pain. Head CT revealed enhanced mass lesion in the left temporal and para-fourth ventricle [Figure - 2]. Beta-HCG titer in serum was slightly elevated (2.2 ng/ml). The AFP and CEA titers in serum were normal, although they were not tested in CSF. Tumor cells were not detected in CSF cytology. Spinal magnetic resonance imaging (MRI) was not investigated. However, it was suspected as recurrent intracranial and spinal cord germinoma with syncytiotrophoblastic giant cells (STGC) based on neurological findings, CT findings and elevation of beta-HCG titer. The patient was treated by whole-brain and whole-spine irradiation with 30Gy and 42Gy, respectively, in 150cGy daily fractions. When irradiation approached 15Gy the pineal region was protected. No tumor was visible on head CT and beta-HCG titers in serum were undetectable.

Third admission

In July 1988, the patient visited the hospital for a tender abdominal mass. Abdominal MRI revealed a huge tumor in the intraperitoneal space [Figure - 3]. Beta-HCG titer in serum was slightly elevated at 4.8ng/ml. while AFP and CEA titer in serum were normal. It was diagnosed as metastasis of intracranial germinoma involving the VP shunt based on MRI finding and beta-HCG titer. The patient was treated by whole-abdomen irradiation with a total dose of 15Gy in 100cGy daily fractions and local irradiation with an additional 15Gy. No tumor was visible on abdominal CT and beta-HCG titer in serum was undetectable.

Fourth admission

In September 1997, the patient presented with a four-month-history of right shoulder pain, right upper limb dysesthesia and weakness. Chest MRI revealed a large right-sided intrathoracic mass lesion connected to the intraspinal canal lesion [Figure - 4]. Beta-HCG titer in serum was slightly elevated (3.3 ng/ml). The AFP and CEA titers in serum were normal. Biopsy was performed for intrathoracic mass lesion and diagnosed as germinoma [Figure - 5]. Immunohistochemical studies were performed and neoplastic cells were positive for placental alkaline phosphatase and negative for AFP, CEA and HCG. The patient was treated by local irradiation with total 34Gy, in 170cGy daily fractions and intravenous administration of cisplatin (dose 30 mg) and etoposide (dose 100 mg) for five consecutive days. It was repeated twice every three weeks in addition to local irradiation for right lung apex with 20Gy, in 200cGy daily fractions. Scar tissue of the right lung apex was apparent on chest CT although beta-HCG titer in serum became undetectable after irradiation. After eight years, he now runs a liquor shop and he is enjoying his life without recurrence.


 » Discussion Top


Some reports have demonstrated intraperitoneal metastasis through a VP-shunt,[5] it was not recommend the operation of VP-shunt before the intracranial germinoma treatment.[4] In fact, we should have treated the metastatic invasion in our case with chemotherapy or radiotherapy to follow the ventricular drainage temporally[6] or third ventriculostomy[7] for obstructive hydrocephalus.

Intracranial germinomas and germinomas with STGC recur in 10 to 17% of cases,[1],[4] within two years of the initial tumor, usually at nonirradiated sites.[4],[8] In the case of germinomas with STGC, some studies have reported tumor recurrence at the irradiated site.[8] Control of the tumors outside the irradiated area is possible only with radiation therapy[9] or a combination of radiation therapy and chemotherapy.[3] In this case, we used irradiation even for those recurring nine years after treatment since tumor site is different each time. Recurrences more than five years later are extremely rare,[4],[9],[10] and such cases often occur in the irradiated field. However, tumor control is not always possible even with additional irradiation in view of the brain damage.[10] Therefore, it is important to aggressively treat the intracranial germinoma the first time. In addition, a long-term follow-up is necessary as germinomas can recur even 23 years after treatment.[10]

The findings reveal that the mechanism of pulmonary metastasis of intracranial germinoma involves the spinal cord originating as a thoracic mass lesion. This explains the relationship between the tumors of the intraspinal canal and the intrapleural space [Figure - 4] and is the first such case of a metastasis. Given the frequent occurrence of systemic failure after therapy, we believe a careful follow-up of extraneural metastases is essential.

 
 » References Top

1.Shirato H, Nishio M, Sawamura Y, Myohjin M, Kitahara T, Nishioka T, et al . Analysis of long-term treatment of intracranial germinoma. Int J Radiat Oncol Biol Phys 1997;37:511-5.  Back to cited text no. 1    
2.Itami J, Kondo T, Niino H, Saito K, Uno T, Aruga M. Bone metastasis of intracranial germinoma. Acta Oncol 1999;38:267-8.  Back to cited text no. 2  [PUBMED]  
3.Tosaka M, Ogimi T, Itoh J, Itoh H, Hayashi S, Ono N, et al . Spinal epidural metastasis from pineal germinoma. Acta Neurochir (Wien) 2003;145:407-10.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Ono N, Isobe I, Uki J, Kurihara H, Shimizu T, Kohno K. Recurrence of primary intracranial germinomas after complete response with radiotherapy: Recurrence patterns and therapy. Neurosurgery 1994;35:615-21.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.Pallini R, Bozzini V, Scerrati M, Zuppi C, Zappacosta B, Rossi GF. Bone metastasis associated with shunt-related peritoneal deposits from a pineal germinoma: Case report and review of the literature. Acta Neurochir (Wien) 1991;109:78-83.  Back to cited text no. 5  [PUBMED]  
6.Buatti JM, Friedman WA. Temporary ventricular drainage and emergency radiotherapy in the management of hydrocephalus associated with germinoma. J Neurosurg 2002;96:1020-2.  Back to cited text no. 6  [PUBMED]  
7.Gangemi M, Maiuri F, Colella G, Buonamassa S. Endoscopic surgery for pineal region tumors. Minim Invasive Neurosurg 2001;44:70-3.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Utsuki S, Kawano N, Oka H, Tanaka T, Suwa T, Fujii K. Cerebral germinoma with syncytiotrophoblastic giant cells: Feasibility of predicting prognosis using the serum hCG level. Acta Neurochir (Wien) 1999;141:975-8.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Tokoro K, Chiba Y, Murase S, Yagishita S, Kyuma Y. Subarachnoid dissemination of pineal germinoma 9 years after radiation therapy without local relapse-case report. Neurol Med Chir (Tokyo) 1991;31:725-8.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]
10.Kiltie AE, Collins CD, Gattamaneni HR, Shalet SM. Relapse of intracranial germinoma 23 years postirradiation in a patient given growth hormone replacement. Med Pediatr Oncol 1997;29:41-4.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]


    Figures

  [Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5]



 

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