10.1007/s00381-007-0495-2</TITLE= > <META content=3D"text/html; charset=3DISO-8859-1" = http-equiv=3DContent-Type><LINK=20 rel=3DstyleSheet type=3Dtext/css=20 href=3D"http://www.springerlink.com/config/css/A++_common.css"><LINK=20 rel=3DstyleSheet type=3Dtext/css=20 href=3D"http://www.springerlink.com/config/css/content_A++_SpringerLink.c= ss"> <META name=3DGENERATOR content=3D"MSHTML 8.00.6001.18372"></HEAD> <BODY class=3Dfor-print> <TABLE border=3D1 cellPadding=3D2> <TBODY> <TR class=3Dheader> <TD>Child's Nervous System</TD></TR> <TR> <TD>=A9 Springer-Verlag 2007</TD></TR> <TR> <TD>10.1007/s00381-007-0495-2</TD></TR></TBODY></TABLE> <H2 class=3Drubric>Original Paper</H2> <DIV class=3DHeading1><A name=3Dtitle></A>Germinoma involving the basal = ganglia in=20 children </DIV> <P class=3DAuthorGroup>Tai-Tong Wong<SUP>1 <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#ContactOfAuthor1"><IMG=20 border=3D0 alt=3D"Contact Information"=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/contact.gif">= </A></SUP>,=20 Y. W. Chen<SUP>2</SUP>, Wan-Yuo Guo<SUP>3</SUP>,=20 Kai-Ping Chang<SUP>4</SUP>, Donald M. Ho<SUP>5</SUP> and=20 Sang-Hue Yen<SUP>2</SUP></P> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff1></A>(1) </SPAN></TD> <TD><SPAN class=3DAffiliation>Division of Pediatric Neurosurgery,=20 Neurological Institute, Taipei Veterans General Hospital and = National Yang=20 Ming University, School of Medicine, No. 210, Sec 2, Shih-Pai, = Taipei,=20 Taiwan, 11217, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff2></A>(2) </SPAN></TD> <TD><SPAN class=3DAffiliation>Cancer Center, School of Medicine, = Taipei=20 Veterans General Hospital and National Yang Ming University, = Taipei,=20 Taiwan, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff3></A>(3) </SPAN></TD> <TD><SPAN class=3DAffiliation>Department of Radiology, School of = Medicine,=20 Taipei Veterans General Hospital and National Yang Ming = University,=20 Taipei, Taiwan, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff4></A>(4) </SPAN></TD> <TD><SPAN class=3DAffiliation>Department of Pediatrics, School of = Medicine,=20 Taipei Veterans General Hospital and National Yang Ming = University,=20 Taipei, Taiwan, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff5></A>(5) </SPAN></TD> <TD><SPAN class=3DAffiliation>Division of Neuropathology, Department = of=20 Pathology, School of Medicine, Taipei Veterans General Hospital = and=20 National Yang Ming University, Taipei, Taiwan, Republic of=20 China</SPAN></TD></TR></TBODY></TABLE> <P><A name=3DContactOfAuthor1></A></P> <TABLE class=3DContact> <TBODY> <TR> <TD vAlign=3Dtop><IMG border=3D0 alt=3D"Contact Information"=20 = src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/contact.gif">= </TD> = <TD><STRONG>Tai-Tong </STRONG><STRONG>Wong</STRONG><STRONG></STRONG>= <BR><STRONG>Email:=20 </STRONG><A=20 = href=3D"mailto:ttwong@vghtpe.gov.tw">ttwong@vghtpe.gov.tw</A></TD></TR></= TBODY></TABLE> <P class=3DAffiliation><STRONG>Received:=20 </STRONG>12 July 2007 <STRONG>Published online:=20 </STRONG>29 September 2007 </P> <DIV class=3DAbstract><A name=3DAbs1></A><SPAN = class=3DAbstractHeading>Abstract</SPAN> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A=20 name=3DASec1></A>Background </SPAN>Germinoma originating in = the basal=20 ganglia is rare, and the majority of reported papers have been from = Japan. In a=20 collection of the first 500 cases of primary brain tumors in children in = Taipei=20 Veterans General Hospital, six pure germinomas with tissue diagnosis = situated in=20 this location. </DIV></DIV> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A name=3DASec2></A>Materials = and=20 methods </SPAN>We reviewed the clinical features, = neuroimaging=20 studies, tumor markers, management, and outcome of these six patients.=20 </DIV></DIV> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A=20 name=3DASec3></A>Results </SPAN>All of them were boys. The = median age=20 of onset of symptoms was 9.7 years. They uniformly presented with=20 hemiparesis. The average duration of symptoms before surgical management = was=20 1 year. One patient had bilateral basal ganglia tumors. Serum = β-human=20 chorionic gonadotropin levels was elevated (128 mIU/ml) in one = patient.=20 Longitudinal neuroimaging studies in four patients clearly showed that = the tumor=20 arose as a tiny lesion at the lenticular nucleus. Five patients had = cysts within=20 tumors. Five patients received partial, subtotal, to total resection. = One=20 patient had stereotactic biopsy of the tumors. Postoperative primary = adjuvant=20 therapies included radiotherapy, chemotherapy alone, and combined = chemotherapy=20 and radiotherapy. Five patients survived, and one patient died of=20 radiation-induced sarcoma with median follow-up period of = 13.7 years. Local=20 recurrence was observed in all of three patients after solitary = postoperative=20 chemotherapy. </DIV></DIV> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A=20 name=3DASec4></A>Conclusions </SPAN>The lenticular nucleus is = a=20 significant locus for germinomas and can be bilateral. Although rarely = reported=20 in Western countries, it does exist in Taiwan as well. Treatment of = germinomas=20 in this specific location is similar to germinoma in other intracranial=20 locations. </DIV></DIV></DIV> <P class=3DKeyword><SPAN=20 class=3DKeywordHeading>Keywords </SPAN>Germinoma - I= ntracranial - Basal=20 ganglia - Children </P> <DIV><A name=3DSec1></A> <HR> <DIV class=3Dheading2>Introduction</DIV> <P>In a review of 119 published reports, among 253 histologically = verified=20 intracranial germinomas, only five germinomas originated outside the = suprasellar=20 and pineal regions [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR1">1</A></CITE>].=20 Hoffman reviewed 51 cases of intracranial germ cell tumors in children = treated=20 at the Hospital for Sick Children, Toronto. Among 34 patients with pure=20 germinomas, there was no tumor situated in the basal ganglia and = thalamus=20 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR2">2</A></CITE>].=20 However, the same institute reported four cases collecting from two = hospitals in=20 2006 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR3">3</A></CITE>].=20 In Asia, Kobayashi in Japan reported the incidence of intracranial = germinoma=20 involving the basal ganglia and thalamus to be 10% in 1981 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>].=20 There was also a reported series of 13 patients in South Korea in 1998 = [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR5">5</A></CITE>].<SUP>31</SUP>=20 According to our experience of the first 32 intracranial germinomas in = children=20 with tissue diagnoses, six tumors arose from the basal ganglia. The = distribution=20 of primary intracranial germinoma in this location was 18.8%. We report = the=20 clinical features, neuroimaging studies, tumor marker studies, = management, and=20 outcome of these six patients. </P></DIV> <DIV><A name=3DSec2></A> <HR> <DIV class=3Dheading2>Materials and methods</DIV> <DIV><A name=3DSec3></A> <DIV class=3DHeading3>Clinical materials</DIV> <DIV class=3DPara> <DIV>During the period 1978 to 1994, 500 children of less than = 15 years old=20 were treated for primary brain tumors at the Taipei Veterans General = Hospital.=20 Among 432 tumors with tissue diagnosis, 59 cases were germ cell tumors, = and 32=20 cases were pure germinomas. Of these 32 cases of intracranial = germinomas, 14=20 were located in the suprasellar region, 11 in the pineal region, 1 in = both the=20 suprasellar and pineal regions, and 6 in the basal ganglia. Among these = six=20 children with germinomas involving the basal ganglia, five patients had=20 unilateral lesion, and one patient had bilateral lesions. They were all = boys.=20 The age range at diagnosis was from 7.9 to 13.6 years. The median = age was=20 10.9 years (Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab1">1</A>).=20 <A name=3DTab1></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 1 </SPAN> = Location, age=20 distribution, and sex ratio of 32 intracranial germinomas in children of = less=20 than 15 years old </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH align=3Dleft> <P>Location</P></TH> <TH align=3Dleft> <P>Case number</P></TH> <TH align=3Dleft> <P>Sex (M/F)</P></TH> <TH align=3Dleft> <P>Age range (years)</P></TH> <TH align=3Dleft> <P>Median</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>Suprasellar</P></TD> <TD align=3Dleft> <P>14</P></TD> <TD align=3Dleft> <P>6/8</P></TD> <TD align=3Dleft> <P>6=9615</P></TD> <TD align=3Dleft> <P>11.3</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>Pineal</P></TD> <TD align=3Dleft> <P>11</P></TD> <TD align=3Dleft> <P>10/1</P></TD> <TD align=3Dleft> <P>2=9614</P></TD> <TD align=3Dleft> <P>9.7</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>Suprasellar and pineal</P></TD> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>1/0</P></TD> <TD align=3Dleft> <P>13.5</P></TD> <TD align=3Dleft> <P>13.5</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>Basal ganglia</P></TD> <TD align=3Dleft> <P>6</P></TD> <TD rowSpan=3D3 align=3Dleft> <P>6/0</P></TD> <TD rowSpan=3D3 align=3Dleft> <P>8=9614</P></TD> <TD rowSpan=3D3 align=3Dleft> <P>10.9</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P> Unilateral</P></TD> <TD align=3Dleft> <P>5</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P> Bilateral</P></TD> <TD align=3Dleft> <P>1</P></TD></TR></TBODY></TABLE></DIV></DIV></DIV> <DIV><A name=3DSec4></A> <DIV class=3DHeading3>Materials and methods</DIV> <P>We reviewed the clinical features, neuroimaging studies, tumor = markers=20 (α-fetoprotein [AFP] and β-human chorionic gonadotropin = [HCG]), management, and=20 outcome of these six patients with pure germinomas involving the basal = ganglia.=20 Serum tumor markers were studied in six patients, cerebrospinal fluid = (CSF)=20 tumor markers were studied in three patients, and tumor markers in tumor = cystic=20 fluid were studied in four patients. Five patients underwent craniotomy = and=20 partial, subtotal, to near-total removal of tumors. One patient = underwent a=20 stereotaxic biopsy. Postoperative management included radiotherapy in = two=20 patients, chemotherapy alone in three patients, and combined = chemotherapy and=20 radiotherapy in one patient. </P></DIV></DIV> <DIV><A name=3DSec5></A> <HR> <DIV class=3Dheading2>Results</DIV> <DIV><A name=3DSec6></A> <DIV class=3DHeading3>Symptom and signs</DIV> <DIV class=3DPara> <DIV>Children with basal ganglia and thalamus germinomas presented with=20 different symptomatology from that of other intracranial germinomas in = the=20 suprasellar location or pineal region. The initial symptoms and signs = were=20 hemiparesis that progressed slowly. The average age of onset of = hemiparesis was=20 9.7 years. The duration of prediagnostic symptoms varied from = 1 month=20 to 2 years and 2 months, with an average of 1 year. There = was no=20 diabetes insipitus, hypopituitarism, or visual loss that characterized = the=20 symptomatology of suprasellar germinomas. There was no Parinaud=92s sign = that=20 might present in pineal region germinomas. Information of precocious = puberty was=20 not available for analysis. Hydrocephalus and raised intracranial = pressure were=20 noted only in one of six patients before surgical treatment. Generalized = convulsion on one occasion was noted in one case. In one patient who had = bilateral basal ganglia lesions presented progressive hemiparesis, = lethargy,=20 slurring speech, drooling, and dysphagia. In one patient, the tumor was=20 associated with familial neurofibromatosis type 1 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR6">6</A></CITE>].=20 Clinical data are summarized in Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab2">2</A>.=20 <A name=3DTab2></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 2 </SPAN> = Summarized clinical=20 data of patients </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH align=3Dleft> <P>Case number</P></TH> <TH align=3Dleft> <P>Age of diagnosis (years)</P></TH> <TH align=3Dleft> <P>Sex</P></TH> <TH align=3Dleft> <P>Duration of symptoms</P></TH> <TH align=3Dleft> <P>Symptoms/signs</P></TH> <TH align=3Dleft> <P>Location of tumor</P></TH> <TH align=3Dleft> <P>Treatment</P></TH> <TH align=3Dleft> <P>Recurrence</P></TH> <TH align=3Dleft> <P>Treatment of recurrence</P></TH> <TH align=3Dleft> <P>Follow-up /outcome</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>9.7</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>1 month</P></TD> <TD align=3Dleft> <P>Right hemiparesis</P></TD> <TD align=3Dleft> <P>Left basal ganglia</P></TD> <TD align=3Dleft> <P>Stereotactic biopsy, RT (LF, 5,000 cGy; CSI,=20 2,600 cGy)</P></TD> <TD align=3Dleft> <P>No</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>13.2 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>2</P></TD> <TD align=3Dleft> <P>13.6</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>11 months</P></TD> <TD align=3Dleft> <P>Right hemiparesis, seizures, headache, vomiting</P></TD> <TD align=3Dleft> <P>Left basal ganglia- thalamus- cerebral white matter</P></TD> <TD align=3Dleft> <P>Craniotomy, subtotal removal, RT (LF, 5,032 cGy; WB,=20 3,830 cGy; WS, 2,515 cGy)</P></TD> <TD align=3Dleft> <P>No</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>15.9 years, died of radiation-induced tumor</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>11.8</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>2 years 2 months</P></TD> <TD align=3Dleft> <P>Left hemiparesis, familial NF1</P></TD> <TD align=3Dleft> <P>Right basal ganglia- thalamus</P></TD> <TD align=3Dleft> <P>Craniotomy, near-total removal, CMT</P></TD> <TD align=3Dleft> <P>Recurrent, 14 mos post- CMT</P></TD> <TD align=3Dleft> <P>RT (LF, 3,000 cGy; WB 3,000 cGy)</P></TD> <TD align=3Dleft> <P>14.5 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>4</P></TD> <TD align=3Dleft> <P>9.3</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>10 months</P></TD> <TD align=3Dleft> <P>Left hemiparesis</P></TD> <TD align=3Dleft> <P>Right basal ganglia,</P></TD> <TD align=3Dleft> <P>Craniotomy, subtotal removal, CMT</P></TD> <TD align=3Dleft> <P>Recurrent, 6 months post-CMT</P></TD> <TD align=3Dleft> <P>RT (LF, 4,993 cGy; WB, 2,000 cGy)</P></TD> <TD align=3Dleft> <P>11.1 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>7.9</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>5 months</P></TD> <TD align=3Dleft> <P>Right hemiparesis</P></TD> <TD align=3Dleft> <P>Left basal ganglia</P></TD> <TD align=3Dleft> <P>Craniotomy, subtotal removal, CMT</P></TD> <TD align=3Dleft> <P>Recurrent, 13 months post-CMT</P></TD> <TD align=3Dleft> <P>RT (LF, 4,799 cGy; WB, 3,000 cGy)</P></TD> <TD align=3Dleft> <P>6.1 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>6</P></TD> <TD align=3Dleft> <P>13.6</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>1 year 8 months</P></TD> <TD align=3Dleft> <P>Left hemiparesis, lethargy, drooling, slurring speech, = dysphagia</P></TD> <TD align=3Dleft> <P>Left basal ganglia, right basal ganglia- thalamus</P></TD> <TD align=3Dleft> <P>Craniotomy, partial removal, CMT and RT (LF, 4,996 cGy; = WB,=20 2,791 cGy)</P></TD> <TD align=3Dleft> <P>No</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>12.8 years, tumor-free, hemiplegia, slurring=20 speech</P></TD></TR></TBODY></TABLE> <DIV class=3DCapt> <DIV class=3DCaptCont> <DIV><I>CSI</I> Craniospinal irradiation, <I>LF</I> local field = irradiation,=20 <I>WB</I> whole-brain irradiation, <I>WS</I> whole spine irradiation, = <I>RT</I>=20 radiotherapy, <I>CMT</I> chemotherapy = </DIV></DIV></DIV></DIV></DIV></DIV> <DIV><A name=3DSec7></A> <DIV class=3DHeading3>Neuroimaging</DIV> <DIV class=3DPara> <DIV>The tumors were diagnosed by computed tomography (CT) in three = patients and=20 by magnetic resonance imaging (MRI) in three patients. Four cases had=20 longitudinal neuroimaging studies before surgery. A novel finding was = that the=20 basal ganglia germinomas arose as a tiny lesion in the lenticular = nucleus=20 (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig1">1</A>a=96b).=20 The tumors enlarged during the period of slow progression of = hemiparesis. At a=20 later stage, the tumor occupied the basal ganglia and showed a large = irregular,=20 multicystic, enhancing mass (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig1">1</A>c=96d).=20 In this series, four patients had CT or MRI evidence of lenticular = nucleus=20 lesions in the initial neuroimaging studies. Small cysts were found in = two of=20 them. At a late stage, in the preoperative studies, large multicysts = were found=20 in association with the tumor in five of six patients. The association = of=20 ipsilateral hemicerebral atrophy was not found before treatment. = Definite to=20 marked ipsilateral hemicerebral atrophy was noted after treatment in = cases 1, 2,=20 5, and 6 (tumors of advanced stage) with disappearance of tumors. Case 6 = presented bilateral basal ganglia lesions in the first MRI study = (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig2">2</A>a).=20 After prolonged informal treatment for 20 months, before surgical=20 resection, the left basal ganglia lesion disappeared, but the right = basal=20 ganglia lesion progressed (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig2">2</A>b).=20 After postoperative radiotherapy and chemotherapy, there was no residue = tumor,=20 but a large empty space remained over the right basal ganglia and = thalamus=20 region with prominent right hemispheric atrophy. There was evidence of a = small=20 area of tissue loss at the right basal ganglia (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig2">2</A>c=96d).=20 The neuroimaging findings are listed in Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab3">3</A>.=20 <DIV class=3DFigure><A name=3DFig1></A><IMG=20 alt=3DMediaObjects/381_2007_495_Fig1_HTML.jpg=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/MediaObjects/= 381_2007_495_Fig1_HTML.jpg"></DIV> <DIV class=3DCapt><SPAN class=3DCaptNr>Fig. 1 </SPAN> Case 4. = MRI at onset=20 of hemiparesis (<B>a</B>, <B>b</B>), 10 months after hemiparesis = with tumor=20 progression (<B>c</B>, <B>d</B>), after operation (<B>e</B>, <B>f</B>), = and=20 after chemotherapy (<B>g</B>, <B>h</B>). <B>a</B>, <B>b</B> A tiny = cystic lesion=20 in right lenticular nucleus with enhancement. <B>c</B>, <B>d</B> The = lesion=20 enlarged with multiple large cysts involving the basal ganglion and = thalamus.=20 <B>e</B>, <B>f</B> Subtotal removal of the tumor through a frontal = craniotomy.=20 <B>g</B>, <B>h</B> Follow-up images 6 months later after six = courses of=20 chemotherapy (VBPE regimen) showed tumor regression. The caudate nucleus = and=20 internal capsule were preserved </DIV> <HR> <DIV class=3DFigure><A name=3DFig2></A><IMG=20 alt=3DMediaObjects/381_2007_495_Fig2_HTML.jpg=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/MediaObjects/= 381_2007_495_Fig2_HTML.jpg"></DIV> <DIV class=3DCapt><SPAN class=3DCaptNr>Fig. 2 </SPAN> Case 6. = MRI at=20 3 months after onset of left hemiparesis (<B>a</B>), 20 months = after=20 informal treatment with progressing clinical symptoms/signs (<B>b</B>), = and=20 10 years follow-up after radiotherapy and chemotherapy (<B>c</B>,=20 <B>d</B>). <B>a</B> Bilateral basal ganglia lesions. <B>b</B> = Progressing right=20 basal ganglia lesion and partial regression of the left-side lesion. = <B>c</B>,=20 <B>d</B> Prominent Wallerian degeneration and atrophy over the right = cerebral=20 hemisphere. Evidences of tissue loss of the left basal ganglion </DIV> <HR> <A name=3DTab3></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 3 </SPAN> = Neuroimaging=20 findings </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH align=3Dleft> <P>Case number</P></TH> <TH align=3Dleft> <P>Neuroimaging</P></TH> <TH align=3Dleft> <P>Size (cm)</P></TH> <TH align=3Dleft> <P>Initial study location</P></TH> <TH align=3Dleft> <P>Cyst</P></TH> <TH align=3Dleft> <P>Duration</P></TH> <TH align=3Dleft> <P>Size (cm)</P></TH> <TH align=3Dleft> <P>Preoperative Study Location</P></TH> <TH align=3Dleft> <P>Cyst</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>CT</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>2.5 =D7 2.5 =D7 2.5</P></TD> <TD align=3Dleft> <P>Left basal ganglia</P></TD> <TD align=3Dleft> <P>−</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>2</P></TD> <TD align=3Dleft> <P>CT</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>5 =D7 6 =D7 6</P></TD> <TD align=3Dleft> <P>Left basal ganglia, thalamus, deep hemisphere</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>CT</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>Right lenticular nucleus</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>13 mos</P></TD> <TD align=3Dleft> <P>4 =D7 4 =D7 5</P></TD> <TD align=3Dleft> <P>Right basal ganglia, thalamus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>4</P></TD> <TD align=3Dleft> <P>MRI</P></TD> <TD align=3Dleft> <P>1 =D7 1 =D7 1</P></TD> <TD align=3Dleft> <P>Right lenticular nucleus</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>10 mos</P></TD> <TD align=3Dleft> <P>3.5 =D7 3.5 =D7 3</P></TD> <TD align=3Dleft> <P>Right basal ganglia, thalamus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>MRI</P></TD> <TD align=3Dleft> <P>1 =D7 1 =D7 1</P></TD> <TD align=3Dleft> <P>Left lenticular nucleus</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>2 mos</P></TD> <TD align=3Dleft> <P>2.5 =D7 2.5 =D7 2.5</P></TD> <TD align=3Dleft> <P>It lenticular nucleus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>6</P></TD> <TD align=3Dleft> <P>MRI</P></TD> <TD align=3Dleft> <P>2 =D7 1 =D7 1</P></TD> <TD align=3Dleft> <P>Left and right lentricular nucleus</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>16 mos</P></TD> <TD align=3Dleft> <P>3 =D7 3 =D7 4</P></TD> <TD align=3Dleft> <P>Right basal ganglia, thalamus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR></TBODY></TABLE></DIV></DIV></DIV> <DIV><A name=3DSec8></A> <DIV class=3DHeading3>Surgical management, pathological findings,=20 immunohistochemical staining, and tumor markers (Serum, cerebrospinal = fluid, and=20 cystic fluid of tumors) </DIV> <DIV class=3DPara> <DIV>Cases 2, 3, 4, 5, and 6 underwent craniotomies and partial, = subtotal, to=20 near-total removal of the tumor. Postoperatively, there were no = additional=20 neurological deficits and no worsening of hemiparesis in particular. = Adequate=20 tumor tissues were obtained for pathological studies in all of these = patients.=20 Case 1 underwent a stereotaxic biopsy. Pathologically, they were all = germinomas=20 composed of sheets or lobules of germ cells admixed with variable amount = of=20 lymphocytes. The tumor cells were round with abundant clear or slightly=20 acidophilic cytoplasm. The tumor cell nuclei were large and vesicular, = some with=20 prominent nucleoli. Mitotic activity was not uncommonly found. Despite = multiple=20 sectioning of these tumors, there was no other germ cell tumor = components=20 identified (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig3">3</A>).=20 Immunohistochemical staining for placental alkaline phosphatase (PLAP) = was=20 positive in all cases and was mainly located on the cell membrane as = well as in=20 the cytoplasm of some tumor cells. Immunostaining for AFP and = cytokeratin were=20 all negative. Immunostaining for β-HCG was negative in cases 1 to = 5, and was=20 positive for syncytiotrophoblastic giant cells (STGCs) in case 6. All of = these=20 six patients had serum AFP and β-HCG studies. Serum β-HCG = levels were normal in=20 cases 1, 3, 4, 5 and 6. However, it was high in case 2 = (128 mIU/ml). Cases=20 4 and 5 had CSF tumor marker studies that were normal. Case 6 had an = elevated=20 level of CSF β-HCG (117 mIU/ml) and a normal level of CSF AFP. = Intraoperatively, cystic fluid was obtained for tumor marker studies in = cases 2,=20 4, 5, and 6. β-HCG titers were high in cases 2 = (17,614 mIU/ml), 5=20 (3,943 mIU/ml), and 6 (1,100 mIU/ml). Both AFP (84 ng/ml) = and=20 β-HCG levels (106 mIU/ml) were elevated in case 4. = Immunohistochemical=20 stains and tumor marker levels are summarized in Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab4">4</A>.=20 <DIV class=3DFigure><A name=3DFig3></A><IMG=20 alt=3DMediaObjects/381_2007_495_Fig3_HTML.jpg=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/MediaObjects/= 381_2007_495_Fig3_HTML.jpg"></DIV> <DIV class=3DCapt><SPAN class=3DCaptNr>Fig. 3 </SPAN> = Histopathology of=20 the right basal ganglia germinoma of case 4 </DIV> <HR> <A name=3DTab4></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 4 </SPAN> Results = of=20 Immunohistochemical stains of PLAP/HCG/AFP and serum/CSF/TCF (tumor = cystic=20 fluid) levels of HCG and AFT </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH rowSpan=3D2 align=3Dleft> <P>Case</P></TH> <TH rowSpan=3D2 align=3Dleft> <P>STGC</P></TH> <TH colSpan=3D3 align=3Dleft> <P>Immunohistochemical stain</P></TH> <TH colSpan=3D3 align=3Dleft> <P>HCG(mIU/ml)</P></TH> <TH align=3Dleft> <P>AFP(ng/ml)</P></TH> <TD class=3Dauto-generated> </TD> <TD class=3Dauto-generated> </TD> <TD class=3Dauto-generated> </TD></TR> <TR class=3Dheader> <TH align=3Dleft> <P>PLAP</P></TH> <TH align=3Dleft> <P>HCG</P></TH> <TH align=3Dleft> <P>AFP</P></TH> <TH align=3Dleft> <P>Serum</P></TH> <TH align=3Dleft> <P>CSF</P></TH> <TH align=3Dleft> <P>TCF</P></TH> <TH align=3Dleft> <P>Serum</P></TH> <TH align=3Dleft> <P>CSF</P></TH> <TH align=3Dleft> <P>TCF</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>8</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>2</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>128</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>17614</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>10</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>4</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>106</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>84</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>3943</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>7</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>6</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>117</P></TD> <TD align=3Dleft> <P>1100</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD></TR></TBODY></TABLE> <DIV class=3DCapt> <DIV class=3DCaptCont> <DIV><I>STGC</I> Syncytiotrophoblastic giant cells, <I>PLAP</I> = placental=20 alkaline phosphatase, <I>AFP</I> α-fetoprotein, <I>HCG</I> = β-human chorionic=20 gonadotropin, <I>TCF</I> cystic fluid of tumor, <I>ND</I> not done=20 </DIV></DIV></DIV></DIV></DIV></DIV> <DIV><A name=3DSec9></A> <DIV class=3DHeading3>Postoperative management and outcome</DIV> <P>Cytological examinations of CSF and myelographic studies were = performed in=20 all of these patients. There was neither abnormal cell in the CSF nor = CSF=20 seeding on myelography. Cases 1 and 2 were treated by craniospinal = irradiation.=20 Case 1 had tumor-free survival with a follow-up period of 18 years. = Case 2=20 developed tumor-induced soft tissue sarcoma of the right frontal region = and died=20 with follow-up period of 15.9 years. Cases 3, 4, and 5 received 5 = to 6=20 courses of VBPE (Vinblastine, Bleomycin, Cisplatin, Etoposide) alone = after tumor=20 resections with radiographic disappearance of the residual tumors = (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig1">1</A>g=96h).=20 All of them showed local recurrence at 14, 6, and 13 months after = cessation=20 of chemotherapy. They were then treated by salvage local field and whole = brain=20 irradiation. All of them achieved tumor-free survival. The follow-up = periods=20 were 14.5, 11.1, and 6.1 years, respectively. Case 6 was a case of=20 bilateral lesions, and the treatment was delayed for almost = 2 years. He was=20 treated by a combination of VBPE chemotherapy and local-field = whole-brain=20 radiotherapy. The follow-up period is 12.8 years. He was tumor-free = but=20 presented residual severe neurological deficits that included left = hemiplegia,=20 slurring speech, and poor ambulatory status (Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab2">2</A>).=20 </P></DIV></DIV> <DIV><A name=3DSec10></A> <HR> <DIV class=3Dheading2>Discussion</DIV> <P>Germinomas situated in the basal ganglia are rare. The majority of = reported=20 papers have been from Japan. In 1990, including two cases of their own, = Tamaki=20 et al. reviewed 42 cases germ cell tumors originating in the basal = ganglia.=20 Twenty cases of them were pure germinomas of the basal ganglia. Except = for two=20 cases, all were reported from Japan [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR7">7</A></CITE>].=20 However, there were also reported series from South Korea and China and = sporadic=20 case reports from India, Canada, USA, and Malaysia [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR8">8</A></CITE>=96<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR12">12</A></CITE>].=20 In our experience, among 432 pediatric brain tumors with pathological = diagnoses,=20 13 tumors involved the basal ganglia. Ten of these 13 tumors were germ = cell=20 tumors, and six were germinomas. We concluded that germ cell tumor, = particularly=20 germinomas, are the most common tumors in the basal ganglia of children. = </P> <P>CT and MRI findings of germinomas in the basal ganglia were reported = by=20 Soejima et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR13">13</A></CITE>],=20 Takano et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR14">14</A></CITE>],=20 and others [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR8">8</A></CITE>].=20 Similar findings were observed in this series. Longitudinal neuroimaging = studies=20 in four patients revealed their tumors arising from lenticular nucleus = of the=20 basal ganglia at an early stage in all of patients. The tumors extended = to the=20 regions of thalamus and deep hemispheres in later stages of the disease. = We=20 presume that all germinomas involving basal ganglia and thalamus arise = as a tiny=20 lesion in the lenticular nucleus. The close proximity of the lenticular = nucleus=20 and the internal capsule explains why hemiparesis is always the initial = clinical=20 presentation. The hemiparesis might occur before the demonstration of a = lesion=20 in the basal ganglia. In one case (case 3), the first CT scan after=20 3 months of hemiparesis showed no basal ganglia lesion. A second CT = scan=20 was performed 9 months later because the progression of the = hemiparesis=20 demonstrated a tiny enhancing lesion over the right lenticular nucleus. = The=20 tumor enlarged involving the basal ganglia and hypothalamus = 4 months later=20 before surgical intervention [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR6">6</A></CITE>].=20 In the report of Kobayashi et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>],=20 there were two cases that presented with involuntary movement and/or = hemiparesis=20 in whom the first CT scan studies were normal. In children who present = with=20 slowly progressing hemiparesis with or without involuntary movements,=20 contrast-enhanced CT or MRI should be performed to rule out a lesion in = the=20 basal ganglia. If the study is normal or if there is a tiny lesion in = the basal=20 ganglia, longitudinal neuroimaging studies should be performed. A = progressively=20 enlarging lesion in the basal ganglia of children implies a tumor, = especially a=20 germ cell tumor, or germinoma. Intratumoral cysts were characteristic = for=20 germinomas in this location. In five of six tumors in this series, = presurgical=20 neuroimages showed intratumoral cysts. The associated atrophy of the = cerebral=20 hemisphere and brainstem on the affected side was found as the result of = Wallerian degeneration extending from the corona radiata and internal = capsule to=20 the brainstem for a lesion involving the basal ganglia, internal = capsule, and=20 thalamus [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR15">15</A></CITE>].=20 In the present series, it was observed only in patients after treatment = with=20 tumors extending beyond basal ganglia. Bilateral basal ganglia germinoma = is=20 exceedingly rare. However, it has been reported [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR16">16</A></CITE>].=20 We also observed one case in six patients. The initial clinical = presentations=20 and neuroimaging findings might not definite, and the diagnosis is often = delayed. </P> <P>In germ cell tumors, AFP is identified in endodermal sinus tumor = (EST), mixed=20 germ cell tumor with component of EST, embryonal carcinoma, and immature = teratoma. The presence of β-HCG can be found in choriocarcinoma and = various=20 types of germ cell tumors that contain STGCs. It is well known that AFP = and=20 β-HCG in serum and CSF increase in germ cell tumors with components = that secrete=20 these markers [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR17">17</A></CITE>=96<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR19">19</A></CITE>].=20 In five children with large pure germinomas in the basal ganglia, we = achieved=20 partial to near-total removal of these tumors with adequate tumor = tissues for=20 pathological study and immunohistochemical staining. There were no = components of=20 other germ cell tumors. STGCs were found only in one of six children. = However,=20 there were increases in serum β-HCG levels in one of six patients, = CSF β-HCG=20 levels in one of three patients, tumor cystic fluid AFP in one of four = patients,=20 and tumor cystic fluid β-HCG in four of four patients. The presence = of elevated=20 tumor markers in serum, CSF, and especially tumor cystic fluid could not = be=20 explained by inadequate tumor tissue for pathological diagnosis. The = clinical=20 significance of elevated tumor markers, especially β-HCG, in pure = basal ganglia=20 germinomas is unknown. </P> <P>Jennings et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR1">1</A></CITE>]=20 reviewed 253 intracranial germinomas from the literature. He reported an = 11%=20 incidence of spinal metastasis. In four germinomas involving the basal = ganglia=20 and thalamus, Kobayashi et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>]=20 found tumor cells in the CSF of one patient. CSF cytology and = myelographic=20 studies were all negative for spinal metastasis in our six patients.=20 Intracranial germinomas respond very well to either to local = radiotherapy or=20 craniospinal irradiation [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR20">20</A></CITE>].=20 The 5-year survival rate was 85.1% in Hoffman=92s series of 25 = intracranial=20 germinomas (diagnosed between 1952 and 1989) that were receiving = radiotherapy=20 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR2">2</A></CITE>].=20 In a recent study of 180 intracranial germinomas in Japan, the patients = were=20 treated with radiation therapy and/or chemotherapy from 1980 to 2001 = with a mean=20 follow-up time of 89 months. Local field and/or whole brain = irradiation was=20 performed in 114 patients, and craniospinal irradiation was performed in = 66=20 patients. The 8-year overall and event-free survival rate was 91 and = 89%,=20 respectively. In this review, the conclusion was that spinal irradiation = did not=20 contribute to favorable event-free survival in patients with = intracranial=20 germinoma [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR21">21</A></CITE>].=20 Actually, for pure intracranial germinoma without tumor dissemination at = diagnosis, reduction dosage of focal radiotherapy alone or along with=20 chemotherapy are being conducted with good outcomes [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR22">22</A></CITE>=96<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR24">24</A></CITE>].=20 The result of radiotherapy for basal ganglia and thalamus germinomas in=20 Kobayashi=92s series was survival in five of six patient [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>].=20 In our series, five of six patients who received primary or salvage = radiotherapy=20 survived with median follow-up period of 13.7 years. One patient = died of=20 radiation-induced myxoliposarcoma at the skull vault involving the = scalp, skull=20 bone, and dura at 15.9 years after diagnosis and radiation therapy. = Intracranial germ cell tumors may respond to chemotherapy [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR25">25</A></CITE>].=20 Hoffman et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR2">2</A></CITE>]=20 reported that one young child with germinoma and five children with=20 leptomeningeal seeding survived 7 months to 12 years after = adjuvant=20 chemotherapy. To avoid radiotherapy in children, five to six courses of=20 postoperative chemotherapy with VBPE regimen was administered in cases = 3, 4, and=20 5 with initial clearance of the residual tumors. However, all of them = showed=20 focal recurrence of tumors at 6 to 14 months after cessation of=20 chemotherapy. It is clear that VBPE regimen alone could not cure these = three=20 patients. We conclude that VBPE regimen should not be used as a solitary = adjuvant treatment of pure germinomas in the basal ganglia. There was = also a=20 related report on recurrence of intracranial germinoma initially treated = with=20 chemotherapy only [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR26">26</A></CITE>].=20 Because of the uncertainty of elevated tumor markers (AFP and/or = β-HCG) in serum=20 or tumor cystic fluid in patients with pure nondisseminated germinomas = in the=20 basal ganglia and thalamus, we now suggest treating these types of = patients with=20 a combination of limited-field focal reduced-dosage radiotherapy and=20 chemotherapy. </P></DIV> <P></P> <HR> <H2><A name=3DBib1></A>References </H2> <TABLE> <TBODY class=3DCitation> <TR vAlign=3Dtop> <TD>1.</TD> <TD><A name=3DCR1></A>Jennings MT, Gelman R, Hochberg F (1985) = Intracranial=20 germ-cell tumors: natural history and pathogenesis. 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From: Subject: 10.1007/s00381-007-0495-2 Date: Mon, 2 Mar 2009 16:07:39 +0100 MIME-Version: 1.0 Content-Type: multipart/related; type="text/html"; boundary="----=_NextPart_000_0000_01C99B51.0285CFF0" X-MimeOLE: Produced By Microsoft MimeOLE V6.00.2900.5579 This is a multi-part message in MIME format. ------=_NextPart_000_0000_01C99B51.0285CFF0 Content-Type: text/html; charset="iso-8859-1" Content-Transfer-Encoding: quoted-printable Content-Location: http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.html 10.1007/s00381-007-0495-2</TITLE= > <META content=3D"text/html; charset=3DISO-8859-1" = http-equiv=3DContent-Type><LINK=20 rel=3DstyleSheet type=3Dtext/css=20 href=3D"http://www.springerlink.com/config/css/A++_common.css"><LINK=20 rel=3DstyleSheet type=3Dtext/css=20 href=3D"http://www.springerlink.com/config/css/content_A++_SpringerLink.c= ss"> <META name=3DGENERATOR content=3D"MSHTML 8.00.6001.18372"></HEAD> <BODY class=3Dfor-print> <TABLE border=3D1 cellPadding=3D2> <TBODY> <TR class=3Dheader> <TD>Child's Nervous System</TD></TR> <TR> <TD>=A9 Springer-Verlag 2007</TD></TR> <TR> <TD>10.1007/s00381-007-0495-2</TD></TR></TBODY></TABLE> <H2 class=3Drubric>Original Paper</H2> <DIV class=3DHeading1><A name=3Dtitle></A>Germinoma involving the basal = ganglia in=20 children </DIV> <P class=3DAuthorGroup>Tai-Tong Wong<SUP>1 <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#ContactOfAuthor1"><IMG=20 border=3D0 alt=3D"Contact Information"=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/contact.gif">= </A></SUP>,=20 Y. W. Chen<SUP>2</SUP>, Wan-Yuo Guo<SUP>3</SUP>,=20 Kai-Ping Chang<SUP>4</SUP>, Donald M. Ho<SUP>5</SUP> and=20 Sang-Hue Yen<SUP>2</SUP></P> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff1></A>(1) </SPAN></TD> <TD><SPAN class=3DAffiliation>Division of Pediatric Neurosurgery,=20 Neurological Institute, Taipei Veterans General Hospital and = National Yang=20 Ming University, School of Medicine, No. 210, Sec 2, Shih-Pai, = Taipei,=20 Taiwan, 11217, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff2></A>(2) </SPAN></TD> <TD><SPAN class=3DAffiliation>Cancer Center, School of Medicine, = Taipei=20 Veterans General Hospital and National Yang Ming University, = Taipei,=20 Taiwan, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff3></A>(3) </SPAN></TD> <TD><SPAN class=3DAffiliation>Department of Radiology, School of = Medicine,=20 Taipei Veterans General Hospital and National Yang Ming = University,=20 Taipei, Taiwan, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff4></A>(4) </SPAN></TD> <TD><SPAN class=3DAffiliation>Department of Pediatrics, School of = Medicine,=20 Taipei Veterans General Hospital and National Yang Ming = University,=20 Taipei, Taiwan, Republic of China</SPAN></TD></TR></TBODY></TABLE> <TABLE> <TBODY> <TR vAlign=3Dtop> <TD><SPAN class=3DAffiliation><A = name=3DAff5></A>(5) </SPAN></TD> <TD><SPAN class=3DAffiliation>Division of Neuropathology, Department = of=20 Pathology, School of Medicine, Taipei Veterans General Hospital = and=20 National Yang Ming University, Taipei, Taiwan, Republic of=20 China</SPAN></TD></TR></TBODY></TABLE> <P><A name=3DContactOfAuthor1></A></P> <TABLE class=3DContact> <TBODY> <TR> <TD vAlign=3Dtop><IMG border=3D0 alt=3D"Contact Information"=20 = src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/contact.gif">= </TD> = <TD><STRONG>Tai-Tong </STRONG><STRONG>Wong</STRONG><STRONG></STRONG>= <BR><STRONG>Email:=20 </STRONG><A=20 = href=3D"mailto:ttwong@vghtpe.gov.tw">ttwong@vghtpe.gov.tw</A></TD></TR></= TBODY></TABLE> <P class=3DAffiliation><STRONG>Received:=20 </STRONG>12 July 2007 <STRONG>Published online:=20 </STRONG>29 September 2007 </P> <DIV class=3DAbstract><A name=3DAbs1></A><SPAN = class=3DAbstractHeading>Abstract</SPAN> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A=20 name=3DASec1></A>Background </SPAN>Germinoma originating in = the basal=20 ganglia is rare, and the majority of reported papers have been from = Japan. In a=20 collection of the first 500 cases of primary brain tumors in children in = Taipei=20 Veterans General Hospital, six pure germinomas with tissue diagnosis = situated in=20 this location. </DIV></DIV> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A name=3DASec2></A>Materials = and=20 methods </SPAN>We reviewed the clinical features, = neuroimaging=20 studies, tumor markers, management, and outcome of these six patients.=20 </DIV></DIV> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A=20 name=3DASec3></A>Results </SPAN>All of them were boys. The = median age=20 of onset of symptoms was 9.7 years. They uniformly presented with=20 hemiparesis. The average duration of symptoms before surgical management = was=20 1 year. One patient had bilateral basal ganglia tumors. Serum = β-human=20 chorionic gonadotropin levels was elevated (128 mIU/ml) in one = patient.=20 Longitudinal neuroimaging studies in four patients clearly showed that = the tumor=20 arose as a tiny lesion at the lenticular nucleus. Five patients had = cysts within=20 tumors. Five patients received partial, subtotal, to total resection. = One=20 patient had stereotactic biopsy of the tumors. Postoperative primary = adjuvant=20 therapies included radiotherapy, chemotherapy alone, and combined = chemotherapy=20 and radiotherapy. Five patients survived, and one patient died of=20 radiation-induced sarcoma with median follow-up period of = 13.7 years. Local=20 recurrence was observed in all of three patients after solitary = postoperative=20 chemotherapy. </DIV></DIV> <DIV class=3DAbstractSection> <DIV><SPAN class=3DAbstractSectionHeading><A=20 name=3DASec4></A>Conclusions </SPAN>The lenticular nucleus is = a=20 significant locus for germinomas and can be bilateral. Although rarely = reported=20 in Western countries, it does exist in Taiwan as well. Treatment of = germinomas=20 in this specific location is similar to germinoma in other intracranial=20 locations. </DIV></DIV></DIV> <P class=3DKeyword><SPAN=20 class=3DKeywordHeading>Keywords </SPAN>Germinoma - I= ntracranial - Basal=20 ganglia - Children </P> <DIV><A name=3DSec1></A> <HR> <DIV class=3Dheading2>Introduction</DIV> <P>In a review of 119 published reports, among 253 histologically = verified=20 intracranial germinomas, only five germinomas originated outside the = suprasellar=20 and pineal regions [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR1">1</A></CITE>].=20 Hoffman reviewed 51 cases of intracranial germ cell tumors in children = treated=20 at the Hospital for Sick Children, Toronto. Among 34 patients with pure=20 germinomas, there was no tumor situated in the basal ganglia and = thalamus=20 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR2">2</A></CITE>].=20 However, the same institute reported four cases collecting from two = hospitals in=20 2006 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR3">3</A></CITE>].=20 In Asia, Kobayashi in Japan reported the incidence of intracranial = germinoma=20 involving the basal ganglia and thalamus to be 10% in 1981 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>].=20 There was also a reported series of 13 patients in South Korea in 1998 = [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR5">5</A></CITE>].<SUP>31</SUP>=20 According to our experience of the first 32 intracranial germinomas in = children=20 with tissue diagnoses, six tumors arose from the basal ganglia. The = distribution=20 of primary intracranial germinoma in this location was 18.8%. We report = the=20 clinical features, neuroimaging studies, tumor marker studies, = management, and=20 outcome of these six patients. </P></DIV> <DIV><A name=3DSec2></A> <HR> <DIV class=3Dheading2>Materials and methods</DIV> <DIV><A name=3DSec3></A> <DIV class=3DHeading3>Clinical materials</DIV> <DIV class=3DPara> <DIV>During the period 1978 to 1994, 500 children of less than = 15 years old=20 were treated for primary brain tumors at the Taipei Veterans General = Hospital.=20 Among 432 tumors with tissue diagnosis, 59 cases were germ cell tumors, = and 32=20 cases were pure germinomas. Of these 32 cases of intracranial = germinomas, 14=20 were located in the suprasellar region, 11 in the pineal region, 1 in = both the=20 suprasellar and pineal regions, and 6 in the basal ganglia. Among these = six=20 children with germinomas involving the basal ganglia, five patients had=20 unilateral lesion, and one patient had bilateral lesions. They were all = boys.=20 The age range at diagnosis was from 7.9 to 13.6 years. The median = age was=20 10.9 years (Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab1">1</A>).=20 <A name=3DTab1></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 1 </SPAN> = Location, age=20 distribution, and sex ratio of 32 intracranial germinomas in children of = less=20 than 15 years old </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH align=3Dleft> <P>Location</P></TH> <TH align=3Dleft> <P>Case number</P></TH> <TH align=3Dleft> <P>Sex (M/F)</P></TH> <TH align=3Dleft> <P>Age range (years)</P></TH> <TH align=3Dleft> <P>Median</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>Suprasellar</P></TD> <TD align=3Dleft> <P>14</P></TD> <TD align=3Dleft> <P>6/8</P></TD> <TD align=3Dleft> <P>6=9615</P></TD> <TD align=3Dleft> <P>11.3</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>Pineal</P></TD> <TD align=3Dleft> <P>11</P></TD> <TD align=3Dleft> <P>10/1</P></TD> <TD align=3Dleft> <P>2=9614</P></TD> <TD align=3Dleft> <P>9.7</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>Suprasellar and pineal</P></TD> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>1/0</P></TD> <TD align=3Dleft> <P>13.5</P></TD> <TD align=3Dleft> <P>13.5</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>Basal ganglia</P></TD> <TD align=3Dleft> <P>6</P></TD> <TD rowSpan=3D3 align=3Dleft> <P>6/0</P></TD> <TD rowSpan=3D3 align=3Dleft> <P>8=9614</P></TD> <TD rowSpan=3D3 align=3Dleft> <P>10.9</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P> Unilateral</P></TD> <TD align=3Dleft> <P>5</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P> Bilateral</P></TD> <TD align=3Dleft> <P>1</P></TD></TR></TBODY></TABLE></DIV></DIV></DIV> <DIV><A name=3DSec4></A> <DIV class=3DHeading3>Materials and methods</DIV> <P>We reviewed the clinical features, neuroimaging studies, tumor = markers=20 (α-fetoprotein [AFP] and β-human chorionic gonadotropin = [HCG]), management, and=20 outcome of these six patients with pure germinomas involving the basal = ganglia.=20 Serum tumor markers were studied in six patients, cerebrospinal fluid = (CSF)=20 tumor markers were studied in three patients, and tumor markers in tumor = cystic=20 fluid were studied in four patients. Five patients underwent craniotomy = and=20 partial, subtotal, to near-total removal of tumors. One patient = underwent a=20 stereotaxic biopsy. Postoperative management included radiotherapy in = two=20 patients, chemotherapy alone in three patients, and combined = chemotherapy and=20 radiotherapy in one patient. </P></DIV></DIV> <DIV><A name=3DSec5></A> <HR> <DIV class=3Dheading2>Results</DIV> <DIV><A name=3DSec6></A> <DIV class=3DHeading3>Symptom and signs</DIV> <DIV class=3DPara> <DIV>Children with basal ganglia and thalamus germinomas presented with=20 different symptomatology from that of other intracranial germinomas in = the=20 suprasellar location or pineal region. The initial symptoms and signs = were=20 hemiparesis that progressed slowly. The average age of onset of = hemiparesis was=20 9.7 years. The duration of prediagnostic symptoms varied from = 1 month=20 to 2 years and 2 months, with an average of 1 year. There = was no=20 diabetes insipitus, hypopituitarism, or visual loss that characterized = the=20 symptomatology of suprasellar germinomas. There was no Parinaud=92s sign = that=20 might present in pineal region germinomas. Information of precocious = puberty was=20 not available for analysis. Hydrocephalus and raised intracranial = pressure were=20 noted only in one of six patients before surgical treatment. Generalized = convulsion on one occasion was noted in one case. In one patient who had = bilateral basal ganglia lesions presented progressive hemiparesis, = lethargy,=20 slurring speech, drooling, and dysphagia. In one patient, the tumor was=20 associated with familial neurofibromatosis type 1 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR6">6</A></CITE>].=20 Clinical data are summarized in Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab2">2</A>.=20 <A name=3DTab2></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 2 </SPAN> = Summarized clinical=20 data of patients </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH align=3Dleft> <P>Case number</P></TH> <TH align=3Dleft> <P>Age of diagnosis (years)</P></TH> <TH align=3Dleft> <P>Sex</P></TH> <TH align=3Dleft> <P>Duration of symptoms</P></TH> <TH align=3Dleft> <P>Symptoms/signs</P></TH> <TH align=3Dleft> <P>Location of tumor</P></TH> <TH align=3Dleft> <P>Treatment</P></TH> <TH align=3Dleft> <P>Recurrence</P></TH> <TH align=3Dleft> <P>Treatment of recurrence</P></TH> <TH align=3Dleft> <P>Follow-up /outcome</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>9.7</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>1 month</P></TD> <TD align=3Dleft> <P>Right hemiparesis</P></TD> <TD align=3Dleft> <P>Left basal ganglia</P></TD> <TD align=3Dleft> <P>Stereotactic biopsy, RT (LF, 5,000 cGy; CSI,=20 2,600 cGy)</P></TD> <TD align=3Dleft> <P>No</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>13.2 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>2</P></TD> <TD align=3Dleft> <P>13.6</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>11 months</P></TD> <TD align=3Dleft> <P>Right hemiparesis, seizures, headache, vomiting</P></TD> <TD align=3Dleft> <P>Left basal ganglia- thalamus- cerebral white matter</P></TD> <TD align=3Dleft> <P>Craniotomy, subtotal removal, RT (LF, 5,032 cGy; WB,=20 3,830 cGy; WS, 2,515 cGy)</P></TD> <TD align=3Dleft> <P>No</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>15.9 years, died of radiation-induced tumor</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>11.8</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>2 years 2 months</P></TD> <TD align=3Dleft> <P>Left hemiparesis, familial NF1</P></TD> <TD align=3Dleft> <P>Right basal ganglia- thalamus</P></TD> <TD align=3Dleft> <P>Craniotomy, near-total removal, CMT</P></TD> <TD align=3Dleft> <P>Recurrent, 14 mos post- CMT</P></TD> <TD align=3Dleft> <P>RT (LF, 3,000 cGy; WB 3,000 cGy)</P></TD> <TD align=3Dleft> <P>14.5 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>4</P></TD> <TD align=3Dleft> <P>9.3</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>10 months</P></TD> <TD align=3Dleft> <P>Left hemiparesis</P></TD> <TD align=3Dleft> <P>Right basal ganglia,</P></TD> <TD align=3Dleft> <P>Craniotomy, subtotal removal, CMT</P></TD> <TD align=3Dleft> <P>Recurrent, 6 months post-CMT</P></TD> <TD align=3Dleft> <P>RT (LF, 4,993 cGy; WB, 2,000 cGy)</P></TD> <TD align=3Dleft> <P>11.1 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>7.9</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>5 months</P></TD> <TD align=3Dleft> <P>Right hemiparesis</P></TD> <TD align=3Dleft> <P>Left basal ganglia</P></TD> <TD align=3Dleft> <P>Craniotomy, subtotal removal, CMT</P></TD> <TD align=3Dleft> <P>Recurrent, 13 months post-CMT</P></TD> <TD align=3Dleft> <P>RT (LF, 4,799 cGy; WB, 3,000 cGy)</P></TD> <TD align=3Dleft> <P>6.1 years, tumor-free, hemiparesis</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>6</P></TD> <TD align=3Dleft> <P>13.6</P></TD> <TD align=3Dleft> <P>Male</P></TD> <TD align=3Dleft> <P>1 year 8 months</P></TD> <TD align=3Dleft> <P>Left hemiparesis, lethargy, drooling, slurring speech, = dysphagia</P></TD> <TD align=3Dleft> <P>Left basal ganglia, right basal ganglia- thalamus</P></TD> <TD align=3Dleft> <P>Craniotomy, partial removal, CMT and RT (LF, 4,996 cGy; = WB,=20 2,791 cGy)</P></TD> <TD align=3Dleft> <P>No</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>12.8 years, tumor-free, hemiplegia, slurring=20 speech</P></TD></TR></TBODY></TABLE> <DIV class=3DCapt> <DIV class=3DCaptCont> <DIV><I>CSI</I> Craniospinal irradiation, <I>LF</I> local field = irradiation,=20 <I>WB</I> whole-brain irradiation, <I>WS</I> whole spine irradiation, = <I>RT</I>=20 radiotherapy, <I>CMT</I> chemotherapy = </DIV></DIV></DIV></DIV></DIV></DIV> <DIV><A name=3DSec7></A> <DIV class=3DHeading3>Neuroimaging</DIV> <DIV class=3DPara> <DIV>The tumors were diagnosed by computed tomography (CT) in three = patients and=20 by magnetic resonance imaging (MRI) in three patients. Four cases had=20 longitudinal neuroimaging studies before surgery. A novel finding was = that the=20 basal ganglia germinomas arose as a tiny lesion in the lenticular = nucleus=20 (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig1">1</A>a=96b).=20 The tumors enlarged during the period of slow progression of = hemiparesis. At a=20 later stage, the tumor occupied the basal ganglia and showed a large = irregular,=20 multicystic, enhancing mass (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig1">1</A>c=96d).=20 In this series, four patients had CT or MRI evidence of lenticular = nucleus=20 lesions in the initial neuroimaging studies. Small cysts were found in = two of=20 them. At a late stage, in the preoperative studies, large multicysts = were found=20 in association with the tumor in five of six patients. The association = of=20 ipsilateral hemicerebral atrophy was not found before treatment. = Definite to=20 marked ipsilateral hemicerebral atrophy was noted after treatment in = cases 1, 2,=20 5, and 6 (tumors of advanced stage) with disappearance of tumors. Case 6 = presented bilateral basal ganglia lesions in the first MRI study = (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig2">2</A>a).=20 After prolonged informal treatment for 20 months, before surgical=20 resection, the left basal ganglia lesion disappeared, but the right = basal=20 ganglia lesion progressed (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig2">2</A>b).=20 After postoperative radiotherapy and chemotherapy, there was no residue = tumor,=20 but a large empty space remained over the right basal ganglia and = thalamus=20 region with prominent right hemispheric atrophy. There was evidence of a = small=20 area of tissue loss at the right basal ganglia (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig2">2</A>c=96d).=20 The neuroimaging findings are listed in Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab3">3</A>.=20 <DIV class=3DFigure><A name=3DFig1></A><IMG=20 alt=3DMediaObjects/381_2007_495_Fig1_HTML.jpg=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/MediaObjects/= 381_2007_495_Fig1_HTML.jpg"></DIV> <DIV class=3DCapt><SPAN class=3DCaptNr>Fig. 1 </SPAN> Case 4. = MRI at onset=20 of hemiparesis (<B>a</B>, <B>b</B>), 10 months after hemiparesis = with tumor=20 progression (<B>c</B>, <B>d</B>), after operation (<B>e</B>, <B>f</B>), = and=20 after chemotherapy (<B>g</B>, <B>h</B>). <B>a</B>, <B>b</B> A tiny = cystic lesion=20 in right lenticular nucleus with enhancement. <B>c</B>, <B>d</B> The = lesion=20 enlarged with multiple large cysts involving the basal ganglion and = thalamus.=20 <B>e</B>, <B>f</B> Subtotal removal of the tumor through a frontal = craniotomy.=20 <B>g</B>, <B>h</B> Follow-up images 6 months later after six = courses of=20 chemotherapy (VBPE regimen) showed tumor regression. The caudate nucleus = and=20 internal capsule were preserved </DIV> <HR> <DIV class=3DFigure><A name=3DFig2></A><IMG=20 alt=3DMediaObjects/381_2007_495_Fig2_HTML.jpg=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/MediaObjects/= 381_2007_495_Fig2_HTML.jpg"></DIV> <DIV class=3DCapt><SPAN class=3DCaptNr>Fig. 2 </SPAN> Case 6. = MRI at=20 3 months after onset of left hemiparesis (<B>a</B>), 20 months = after=20 informal treatment with progressing clinical symptoms/signs (<B>b</B>), = and=20 10 years follow-up after radiotherapy and chemotherapy (<B>c</B>,=20 <B>d</B>). <B>a</B> Bilateral basal ganglia lesions. <B>b</B> = Progressing right=20 basal ganglia lesion and partial regression of the left-side lesion. = <B>c</B>,=20 <B>d</B> Prominent Wallerian degeneration and atrophy over the right = cerebral=20 hemisphere. Evidences of tissue loss of the left basal ganglion </DIV> <HR> <A name=3DTab3></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 3 </SPAN> = Neuroimaging=20 findings </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH align=3Dleft> <P>Case number</P></TH> <TH align=3Dleft> <P>Neuroimaging</P></TH> <TH align=3Dleft> <P>Size (cm)</P></TH> <TH align=3Dleft> <P>Initial study location</P></TH> <TH align=3Dleft> <P>Cyst</P></TH> <TH align=3Dleft> <P>Duration</P></TH> <TH align=3Dleft> <P>Size (cm)</P></TH> <TH align=3Dleft> <P>Preoperative Study Location</P></TH> <TH align=3Dleft> <P>Cyst</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>CT</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>2.5 =D7 2.5 =D7 2.5</P></TD> <TD align=3Dleft> <P>Left basal ganglia</P></TD> <TD align=3Dleft> <P>−</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>2</P></TD> <TD align=3Dleft> <P>CT</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>5 =D7 6 =D7 6</P></TD> <TD align=3Dleft> <P>Left basal ganglia, thalamus, deep hemisphere</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>CT</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>Right lenticular nucleus</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>13 mos</P></TD> <TD align=3Dleft> <P>4 =D7 4 =D7 5</P></TD> <TD align=3Dleft> <P>Right basal ganglia, thalamus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>4</P></TD> <TD align=3Dleft> <P>MRI</P></TD> <TD align=3Dleft> <P>1 =D7 1 =D7 1</P></TD> <TD align=3Dleft> <P>Right lenticular nucleus</P></TD> <TD align=3Dleft> <P> </P></TD> <TD align=3Dleft> <P>10 mos</P></TD> <TD align=3Dleft> <P>3.5 =D7 3.5 =D7 3</P></TD> <TD align=3Dleft> <P>Right basal ganglia, thalamus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>MRI</P></TD> <TD align=3Dleft> <P>1 =D7 1 =D7 1</P></TD> <TD align=3Dleft> <P>Left lenticular nucleus</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>2 mos</P></TD> <TD align=3Dleft> <P>2.5 =D7 2.5 =D7 2.5</P></TD> <TD align=3Dleft> <P>It lenticular nucleus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>6</P></TD> <TD align=3Dleft> <P>MRI</P></TD> <TD align=3Dleft> <P>2 =D7 1 =D7 1</P></TD> <TD align=3Dleft> <P>Left and right lentricular nucleus</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>16 mos</P></TD> <TD align=3Dleft> <P>3 =D7 3 =D7 4</P></TD> <TD align=3Dleft> <P>Right basal ganglia, thalamus</P></TD> <TD align=3Dleft> <P>+</P></TD></TR></TBODY></TABLE></DIV></DIV></DIV> <DIV><A name=3DSec8></A> <DIV class=3DHeading3>Surgical management, pathological findings,=20 immunohistochemical staining, and tumor markers (Serum, cerebrospinal = fluid, and=20 cystic fluid of tumors) </DIV> <DIV class=3DPara> <DIV>Cases 2, 3, 4, 5, and 6 underwent craniotomies and partial, = subtotal, to=20 near-total removal of the tumor. Postoperatively, there were no = additional=20 neurological deficits and no worsening of hemiparesis in particular. = Adequate=20 tumor tissues were obtained for pathological studies in all of these = patients.=20 Case 1 underwent a stereotaxic biopsy. Pathologically, they were all = germinomas=20 composed of sheets or lobules of germ cells admixed with variable amount = of=20 lymphocytes. The tumor cells were round with abundant clear or slightly=20 acidophilic cytoplasm. The tumor cell nuclei were large and vesicular, = some with=20 prominent nucleoli. Mitotic activity was not uncommonly found. Despite = multiple=20 sectioning of these tumors, there was no other germ cell tumor = components=20 identified (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig3">3</A>).=20 Immunohistochemical staining for placental alkaline phosphatase (PLAP) = was=20 positive in all cases and was mainly located on the cell membrane as = well as in=20 the cytoplasm of some tumor cells. Immunostaining for AFP and = cytokeratin were=20 all negative. Immunostaining for β-HCG was negative in cases 1 to = 5, and was=20 positive for syncytiotrophoblastic giant cells (STGCs) in case 6. All of = these=20 six patients had serum AFP and β-HCG studies. Serum β-HCG = levels were normal in=20 cases 1, 3, 4, 5 and 6. However, it was high in case 2 = (128 mIU/ml). Cases=20 4 and 5 had CSF tumor marker studies that were normal. Case 6 had an = elevated=20 level of CSF β-HCG (117 mIU/ml) and a normal level of CSF AFP. = Intraoperatively, cystic fluid was obtained for tumor marker studies in = cases 2,=20 4, 5, and 6. β-HCG titers were high in cases 2 = (17,614 mIU/ml), 5=20 (3,943 mIU/ml), and 6 (1,100 mIU/ml). Both AFP (84 ng/ml) = and=20 β-HCG levels (106 mIU/ml) were elevated in case 4. = Immunohistochemical=20 stains and tumor marker levels are summarized in Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab4">4</A>.=20 <DIV class=3DFigure><A name=3DFig3></A><IMG=20 alt=3DMediaObjects/381_2007_495_Fig3_HTML.jpg=20 src=3D"http://www.springerlink.com/content/l74wt3pm821611x1/MediaObjects/= 381_2007_495_Fig3_HTML.jpg"></DIV> <DIV class=3DCapt><SPAN class=3DCaptNr>Fig. 3 </SPAN> = Histopathology of=20 the right basal ganglia germinoma of case 4 </DIV> <HR> <A name=3DTab4></A> <DIV class=3DCapt><SPAN class=3DCaptNr>Table 4 </SPAN> Results = of=20 Immunohistochemical stains of PLAP/HCG/AFP and serum/CSF/TCF (tumor = cystic=20 fluid) levels of HCG and AFT </DIV> <TABLE border=3D1> <COLGROUP> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft> <COL align=3Dleft></COLGROUP> <THEAD> <TR class=3Dheader> <TH rowSpan=3D2 align=3Dleft> <P>Case</P></TH> <TH rowSpan=3D2 align=3Dleft> <P>STGC</P></TH> <TH colSpan=3D3 align=3Dleft> <P>Immunohistochemical stain</P></TH> <TH colSpan=3D3 align=3Dleft> <P>HCG(mIU/ml)</P></TH> <TH align=3Dleft> <P>AFP(ng/ml)</P></TH> <TD class=3Dauto-generated> </TD> <TD class=3Dauto-generated> </TD> <TD class=3Dauto-generated> </TD></TR> <TR class=3Dheader> <TH align=3Dleft> <P>PLAP</P></TH> <TH align=3Dleft> <P>HCG</P></TH> <TH align=3Dleft> <P>AFP</P></TH> <TH align=3Dleft> <P>Serum</P></TH> <TH align=3Dleft> <P>CSF</P></TH> <TH align=3Dleft> <P>TCF</P></TH> <TH align=3Dleft> <P>Serum</P></TH> <TH align=3Dleft> <P>CSF</P></TH> <TH align=3Dleft> <P>TCF</P></TH></TR></THEAD> <TBODY> <TR class=3Dnoclass> <TD align=3Dleft> <P>1</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>8</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>2</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>128</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>17614</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>10</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>ND</P></TD> <TD align=3Dleft> <P>ND</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>4</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>106</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>84</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>3943</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>7</P></TD></TR> <TR class=3Dnoclass> <TD align=3Dleft> <P>6</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>+</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>−</P></TD> <TD align=3Dleft> <P>5</P></TD> <TD align=3Dleft> <P>117</P></TD> <TD align=3Dleft> <P>1100</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD> <TD align=3Dleft> <P>3</P></TD></TR></TBODY></TABLE> <DIV class=3DCapt> <DIV class=3DCaptCont> <DIV><I>STGC</I> Syncytiotrophoblastic giant cells, <I>PLAP</I> = placental=20 alkaline phosphatase, <I>AFP</I> α-fetoprotein, <I>HCG</I> = β-human chorionic=20 gonadotropin, <I>TCF</I> cystic fluid of tumor, <I>ND</I> not done=20 </DIV></DIV></DIV></DIV></DIV></DIV> <DIV><A name=3DSec9></A> <DIV class=3DHeading3>Postoperative management and outcome</DIV> <P>Cytological examinations of CSF and myelographic studies were = performed in=20 all of these patients. There was neither abnormal cell in the CSF nor = CSF=20 seeding on myelography. Cases 1 and 2 were treated by craniospinal = irradiation.=20 Case 1 had tumor-free survival with a follow-up period of 18 years. = Case 2=20 developed tumor-induced soft tissue sarcoma of the right frontal region = and died=20 with follow-up period of 15.9 years. Cases 3, 4, and 5 received 5 = to 6=20 courses of VBPE (Vinblastine, Bleomycin, Cisplatin, Etoposide) alone = after tumor=20 resections with radiographic disappearance of the residual tumors = (Fig. <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Fig1">1</A>g=96h).=20 All of them showed local recurrence at 14, 6, and 13 months after = cessation=20 of chemotherapy. They were then treated by salvage local field and whole = brain=20 irradiation. All of them achieved tumor-free survival. The follow-up = periods=20 were 14.5, 11.1, and 6.1 years, respectively. Case 6 was a case of=20 bilateral lesions, and the treatment was delayed for almost = 2 years. He was=20 treated by a combination of VBPE chemotherapy and local-field = whole-brain=20 radiotherapy. The follow-up period is 12.8 years. He was tumor-free = but=20 presented residual severe neurological deficits that included left = hemiplegia,=20 slurring speech, and poor ambulatory status (Table <A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#Tab2">2</A>).=20 </P></DIV></DIV> <DIV><A name=3DSec10></A> <HR> <DIV class=3Dheading2>Discussion</DIV> <P>Germinomas situated in the basal ganglia are rare. The majority of = reported=20 papers have been from Japan. In 1990, including two cases of their own, = Tamaki=20 et al. reviewed 42 cases germ cell tumors originating in the basal = ganglia.=20 Twenty cases of them were pure germinomas of the basal ganglia. Except = for two=20 cases, all were reported from Japan [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR7">7</A></CITE>].=20 However, there were also reported series from South Korea and China and = sporadic=20 case reports from India, Canada, USA, and Malaysia [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR8">8</A></CITE>=96<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR12">12</A></CITE>].=20 In our experience, among 432 pediatric brain tumors with pathological = diagnoses,=20 13 tumors involved the basal ganglia. Ten of these 13 tumors were germ = cell=20 tumors, and six were germinomas. We concluded that germ cell tumor, = particularly=20 germinomas, are the most common tumors in the basal ganglia of children. = </P> <P>CT and MRI findings of germinomas in the basal ganglia were reported = by=20 Soejima et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR13">13</A></CITE>],=20 Takano et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR14">14</A></CITE>],=20 and others [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR8">8</A></CITE>].=20 Similar findings were observed in this series. Longitudinal neuroimaging = studies=20 in four patients revealed their tumors arising from lenticular nucleus = of the=20 basal ganglia at an early stage in all of patients. The tumors extended = to the=20 regions of thalamus and deep hemispheres in later stages of the disease. = We=20 presume that all germinomas involving basal ganglia and thalamus arise = as a tiny=20 lesion in the lenticular nucleus. The close proximity of the lenticular = nucleus=20 and the internal capsule explains why hemiparesis is always the initial = clinical=20 presentation. The hemiparesis might occur before the demonstration of a = lesion=20 in the basal ganglia. In one case (case 3), the first CT scan after=20 3 months of hemiparesis showed no basal ganglia lesion. A second CT = scan=20 was performed 9 months later because the progression of the = hemiparesis=20 demonstrated a tiny enhancing lesion over the right lenticular nucleus. = The=20 tumor enlarged involving the basal ganglia and hypothalamus = 4 months later=20 before surgical intervention [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR6">6</A></CITE>].=20 In the report of Kobayashi et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>],=20 there were two cases that presented with involuntary movement and/or = hemiparesis=20 in whom the first CT scan studies were normal. In children who present = with=20 slowly progressing hemiparesis with or without involuntary movements,=20 contrast-enhanced CT or MRI should be performed to rule out a lesion in = the=20 basal ganglia. If the study is normal or if there is a tiny lesion in = the basal=20 ganglia, longitudinal neuroimaging studies should be performed. A = progressively=20 enlarging lesion in the basal ganglia of children implies a tumor, = especially a=20 germ cell tumor, or germinoma. Intratumoral cysts were characteristic = for=20 germinomas in this location. In five of six tumors in this series, = presurgical=20 neuroimages showed intratumoral cysts. The associated atrophy of the = cerebral=20 hemisphere and brainstem on the affected side was found as the result of = Wallerian degeneration extending from the corona radiata and internal = capsule to=20 the brainstem for a lesion involving the basal ganglia, internal = capsule, and=20 thalamus [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR15">15</A></CITE>].=20 In the present series, it was observed only in patients after treatment = with=20 tumors extending beyond basal ganglia. Bilateral basal ganglia germinoma = is=20 exceedingly rare. However, it has been reported [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR16">16</A></CITE>].=20 We also observed one case in six patients. The initial clinical = presentations=20 and neuroimaging findings might not definite, and the diagnosis is often = delayed. </P> <P>In germ cell tumors, AFP is identified in endodermal sinus tumor = (EST), mixed=20 germ cell tumor with component of EST, embryonal carcinoma, and immature = teratoma. The presence of β-HCG can be found in choriocarcinoma and = various=20 types of germ cell tumors that contain STGCs. It is well known that AFP = and=20 β-HCG in serum and CSF increase in germ cell tumors with components = that secrete=20 these markers [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR17">17</A></CITE>=96<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR19">19</A></CITE>].=20 In five children with large pure germinomas in the basal ganglia, we = achieved=20 partial to near-total removal of these tumors with adequate tumor = tissues for=20 pathological study and immunohistochemical staining. There were no = components of=20 other germ cell tumors. STGCs were found only in one of six children. = However,=20 there were increases in serum β-HCG levels in one of six patients, = CSF β-HCG=20 levels in one of three patients, tumor cystic fluid AFP in one of four = patients,=20 and tumor cystic fluid β-HCG in four of four patients. The presence = of elevated=20 tumor markers in serum, CSF, and especially tumor cystic fluid could not = be=20 explained by inadequate tumor tissue for pathological diagnosis. The = clinical=20 significance of elevated tumor markers, especially β-HCG, in pure = basal ganglia=20 germinomas is unknown. </P> <P>Jennings et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR1">1</A></CITE>]=20 reviewed 253 intracranial germinomas from the literature. He reported an = 11%=20 incidence of spinal metastasis. In four germinomas involving the basal = ganglia=20 and thalamus, Kobayashi et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>]=20 found tumor cells in the CSF of one patient. CSF cytology and = myelographic=20 studies were all negative for spinal metastasis in our six patients.=20 Intracranial germinomas respond very well to either to local = radiotherapy or=20 craniospinal irradiation [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR20">20</A></CITE>].=20 The 5-year survival rate was 85.1% in Hoffman=92s series of 25 = intracranial=20 germinomas (diagnosed between 1952 and 1989) that were receiving = radiotherapy=20 [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR2">2</A></CITE>].=20 In a recent study of 180 intracranial germinomas in Japan, the patients = were=20 treated with radiation therapy and/or chemotherapy from 1980 to 2001 = with a mean=20 follow-up time of 89 months. Local field and/or whole brain = irradiation was=20 performed in 114 patients, and craniospinal irradiation was performed in = 66=20 patients. The 8-year overall and event-free survival rate was 91 and = 89%,=20 respectively. In this review, the conclusion was that spinal irradiation = did not=20 contribute to favorable event-free survival in patients with = intracranial=20 germinoma [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR21">21</A></CITE>].=20 Actually, for pure intracranial germinoma without tumor dissemination at = diagnosis, reduction dosage of focal radiotherapy alone or along with=20 chemotherapy are being conducted with good outcomes [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR22">22</A></CITE>=96<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR24">24</A></CITE>].=20 The result of radiotherapy for basal ganglia and thalamus germinomas in=20 Kobayashi=92s series was survival in five of six patient [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR4">4</A></CITE>].=20 In our series, five of six patients who received primary or salvage = radiotherapy=20 survived with median follow-up period of 13.7 years. One patient = died of=20 radiation-induced myxoliposarcoma at the skull vault involving the = scalp, skull=20 bone, and dura at 15.9 years after diagnosis and radiation therapy. = Intracranial germ cell tumors may respond to chemotherapy [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR25">25</A></CITE>].=20 Hoffman et al. [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR2">2</A></CITE>]=20 reported that one young child with germinoma and five children with=20 leptomeningeal seeding survived 7 months to 12 years after = adjuvant=20 chemotherapy. To avoid radiotherapy in children, five to six courses of=20 postoperative chemotherapy with VBPE regimen was administered in cases = 3, 4, and=20 5 with initial clearance of the residual tumors. However, all of them = showed=20 focal recurrence of tumors at 6 to 14 months after cessation of=20 chemotherapy. It is clear that VBPE regimen alone could not cure these = three=20 patients. We conclude that VBPE regimen should not be used as a solitary = adjuvant treatment of pure germinomas in the basal ganglia. There was = also a=20 related report on recurrence of intracranial germinoma initially treated = with=20 chemotherapy only [<CITE><A=20 href=3D"http://www.springerlink.com/content/l74wt3pm821611x1/fulltext.htm= l#CR26">26</A></CITE>].=20 Because of the uncertainty of elevated tumor markers (AFP and/or = β-HCG) in serum=20 or tumor cystic fluid in patients with pure nondisseminated germinomas = in the=20 basal ganglia and thalamus, we now suggest treating these types of = patients with=20 a combination of limited-field focal reduced-dosage radiotherapy and=20 chemotherapy. </P></DIV> <P></P> <HR> <H2><A name=3DBib1></A>References </H2> <TABLE> <TBODY class=3DCitation> <TR vAlign=3Dtop> <TD>1.</TD> <TD><A name=3DCR1></A>Jennings MT, Gelman R, Hochberg F (1985) = Intracranial=20 germ-cell tumors: natural history and pathogenesis. 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