Chordoid Glioma Originating in the Intrasellar and Suprasellar Regions: Case Report
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1 pissn eissn imri 2015;19: Chordoid Gliom Originting in the Intrsellr nd Suprsellr Regions: Cse Report Jisun Hwng 1, Aleum Lee 1, Kee-Hyun Chng 1, Ah Rim Moon 2, Sun-Chul Hwng 3, Hyun Sook Hong 1 1 Deprtment of Rdiology, Soonchunhyng University Bucheon Hospitl, Bucheon, Kore 2 Deprtment of Pthology, Soonchunhyng University Bucheon Hospitl, Bucheon, Kore 3 Deprtment of Neurosurgery, Soonchunhyng University Bucheon Hospitl, Bucheon, Kore Cse Report Received: April 2, 2015 Revised: April 22, 2015 Accepted: My 10, 2015 Correspondence to: Aleum Lee, M.D. Deprtment of Rdiology, Soonchunhyng University Bucheon Hospitl, Soonchunhyng University College of Medicine, 170 Jomruro, Wonmi-gu, Bucheon , Kore. Tel Fx Emil: leerd@schmc.c.kr This is n Open Access rticle distriuted under the terms of the Cretive Commons Attriution Non-Commercil License ( y-nc/3.0/) which permits unrestricted non-commercil use, distriution, nd reproduction in ny medium, provided the originl work is properly cited. Copyright 2015 Koren Society of Mgnetic Resonnce in Medicine (KSMRM) Chordoid gliom is rre, low-grde rin neoplsm typiclly locted in the third ventricle. Herein, we report n unusul cse of histologiclly confirmed chordoid gliom locted in the pituitry foss nd suprsellr region, not ttched to the third ventricle. A 57-yer-old womn presented with 2-month history of hedche nd visul disturnce. Mgnetic resonnce imging reveled n ovoid mss in the pituitry foss nd suprsellr region, compressing the optic chism without involvement of the third ventricle. The tumor showed low signl intensity on T1- weighted imges nd iso- to high signl intensity on T2-weighted imges, with strong nd homogenous contrst enhncement. Sutotl resection ws performed vi the trnscrnil pproch, nd the ptient susequently received djuvnt gmm knife rdiosurgery. However, the residul mss showed disese progression 5 months fter the initil surgery. Keywords: Chordoid gliom; Third ventricle; Rdiologicl feture; Mgnetic resonnce imging INTRODUCTION Chordoid gliom (CG) is n uncommon, slow-growing primry rin neoplsm, first descried s distinct clinicopthologic entity y Brt et l. (1) in CG ws nmed for its histologic fetures, which resemle those of chordoms expressing glil firillry cidic protein (GFAP) (1). It ws incorported into the World Helth Orgniztion clssifiction s grde 2 lesion under the ctegory of glil tumor of uncertin origin. CG exclusively involves res of the nterior portion of the third ventricle ner the hypothlmus (2). The tumor is termed s CG of the third ventricle ecuse of this stereotypicl loction, nd only few cses of extrventriculr CG hve een reported (3, 4). To the est of our knowledge, 23 cses of suprsellr CG hve een reported to dte, including the current cse. Of these, only few cses of CG were loclized to the suprsellr region without ventriculr involvement (4). Although few previous reports hve descried the typicl rdiologic nd histopthologic fetures of CG, none of the reports focused on the rdio-pthologicl correltion. This is the first report providing comprehensive rdio-pthologicl correltion of pituitry mcrodenom-like 117
2 Chordoid Gliom in the Intr- nd Supr-sellr Regions Jisun Hwng, et l. extrventriculr CG unusully locted in the sellr nd suprsellr regions. CASE REPORT A 57-yer-old womn presented with 2-month history of hedches nd visul disturnce. Neurologicl exmintion reveled itemporl heminopsi. Hed computed tomogrphy (CT) demonstrted well-circumscried ovl mss without clcifiction or cystic components in the sellr nd suprsellr regions. It ppered hyperdense nd showed strong homogenous enhncement (Fig. 1, ). Mgnetic resonnce imging (MRI) reveled cm mss in the pituitry foss nd suprsellr region, which ws elevting nd compressing the optic chism nd the floor of the third ventricle superiorly without tumor extension. The norml pituitry glnd nd stlk were not discernile. The tumor ws hypointense on T1-weighted imges, iso- to hyperintense on T2-weighted imges, nd hyperintense fter gdolinium dministrtion (Fig. 2d). Presumptive dignoses were pituitry mcrodenom, meningiom, or crniophryngiom. The ptient underwent surgery vi the trnscllosl pproch. During surgery, the mss ppered to originte from the diphrgm selle ehind the optic chism, nd it ws not utting the third ventricle. Moreover, the pituitry stlk ws encsed y the tumor. The mss ws prtilly resected ecuse of the difficult surgicl pproch to the posterior portion of the mss. Histologiclly, the tumor ws composed of epithelioid-to-spindle cells rrnged in clusters nd cords, emedded in myxoid mtrix (Fig. 3). The neoplstic cells showed strong diffuse immunorectivity for GFAP (Fig. 3), CD34, nd vimentin. A smll suset of neoplstic cells stined positive for S100, cytokertin, nd epithelil memrne ntigen; however, stining for synptophysin ws negtive. The Ki-67 index ws pproximtely 15%. The differentil dignoses sed on tumor histology were CG, chordoid meningiom, nd chordom. Meningiom nd chordom were excluded sed on the strong GFAP expression; CD34 immunorectivity lso supported the dignosis of CG. The Ki-67 index reflected reltively high prolifertion rte, compred to previously reported rtes. Prtil improvement in visul function ws noted postopertively; however, the ptient developed memory nd git disturnce. Postopertive CT reveled residul mss nd hydrocephlus. The ptient underwent ventriculoperitonel shunt plcement nd received djuvnt gmm knife rdiosurgery. The hydrocephlus stilized, ut the lst ville follow-up MRI scn otined 5 months fter the initil resection reveled disese progression with tumor growth. DISCUSSION First, Brt et l. (1) identified tumor of the third ventricle presenting with oth glil nd chordoid fetures in 1998, nd new rre clinicopthologicl entity ws nmed Fig. 1. An xil precontrst () nd contrst-enhnced () computed tomogrphy scn show n ovl, well-defined, nd hyperdense mss with strong nd homogenous enhncement in the sellr nd suprsellr regions. 118
3 c d s "chordoid gliom of the third ventricle" (3). Since then, to our knowledge, pproximtely 84 cses hve een reported. The tumor develops minly in dults, with 1.7:1 femle predominnce, nd men ge t presenttion is 44.9 yers (5). The most common symptom is hedche; other symptoms include visul disturnce, memory disturnce, nd endocrine dysfunction. CGs exhiit remrkly similr chrcteristics on rin imging (2); the tumor exclusively involves res of the nterior portion of the third ventricle ner the hypothlmus. A few exceptionl cses with unusul loctions such s the juxtventriculr region, left temporoprietl cortex, nd thlmic pulvinr re (3) hve een reported. On CT, the lesions re well circumscried nd hyperttenuting, with uniform contrst enhncement. On MRI, CGs re isointense on T1-weighted imges, with vid nd uniform contrst enhncement fter gdolinium dministrtion, nd iso- to hyperintense on T2-weighted Fig. 2. Mgnetic resonnce imges of the tumor. () Sgittl T1weighted imge shows low signl intensity of the mss. () Coronl T2-weighted imge shows isoto high signl intensity of the mss. Coronl (c) nd sgittl (d) contrst-enhnced T1-weighted imges of the rin revel strong, homogeneously enhncing solid sellr nd suprsellr mss, not connected to the third ventricle. The norml pituitry glnd nd stlk re not discernile. The epicenter of the mss ppers to originte in the sell, nd not in the lmin terminlis of the nterior third ventricle. imges. This significnt contrst enhncement of CG is thought to rise from lood-rin rrier rekdown nd the ensuing extrvstion of contrst medium, rther thn tumor ngiogenesis, which is confirmed y the lck of vsculr prolifertion on histologicl exmintion (6). On sgittl imges, infundiulum shows posterior displcement y the tumor nd gretest dimeter of this tumor is usully oserved in the superoinferior orienttion (2). Intrlesionl clcifiction is rre. Cystic chnges re occsionlly oserved (2). CGs re typiclly well-circumscried nd noninfiltrtive tumors tht re confined to the ventricle, ut rre cse of tumor invsion into the optic chism hs een reported (7). Typicl histopthologic findings of CG include the following: 1) clusters nd cords of ovl-to-polygonl epithelioid tumor cells with undnt eosinophilic cytoplsm, emedded in PAS-positive mucinous mtrix; 2) prominent lymphoplsmcytic infiltrtes nd Russell 119
4 Chordoid Gliom in the Intr- nd Supr-sellr Regions Jisun Hwng, et l. Fig. 3. Histologicl fetures of the tumor. () The tumor shows clusters nd cords of epithelioid-to-spindle cells emedded in myxoid mtrix nd inflmmtory cells (Hemtoxylin & Eosin stining, originl mgnifiction 100). () The positive immunorectivity for glil firillry cidic protein expression re visulized s rown (originl mgnifiction 200). odies throughout the tumor; 3) mitotic ctivity nd nplstic fetures (nucler polymorphism, endothelil prolifertion, necrosis) re sent or rre; nd 4) minor tendency to infiltrte the surrounding rin prenchym; however, rective strocytes, Rosenthl fiers, nd chronic inflmmtory cells re present in djcent rin tissue (1). Although mny pulished cses hve shown consistent histopthologic nd rdiologicl fetures of CG, no report hs specificlly discussed the correltion etween typicl imging fetures nd the histologicl fetures of CG. The CGs re usully hyperdense on non-enhnced CT imges. We elieve tht the hyperdensity is proly relted to the incresed cellulrity of these lesions, owing to prominent lymphoplsmcytic infiltrtes. Moreover, the reltive iso-to hyperintensity of CGs on T2-weighted imges my reflect the presence of undnt eosinophilic cytoplsm in tumor cells nd the undnt mucinous mtrix. The precise pthogenesis of CGs remins uncler. However, severl recent ultrstructurl studies hve proposed n ependyml origin for these tumors (3, 5, 8). In the 3 cses studied y Cencchi et l. (8), the uthors suggest tht CGs my e n ependymom sutype, sed on the ultrstructurl similrities etween CGs nd the specilized ependyml cells of the sucommissurl orgn, which is prominent during emryologic life ut undergoes progressive involution. Psquier et l. (5) proposed the term chordoid ependymom of the lmin terminlis re, sed on the ultrstructurl findings nd remrkly consistent ntomic loction of this tumor in the nterior prt of the third ventricle, closely ssocited with the lmin terminlis, ner severl circumventriculr orgns. Of note, in the current cse, neuroimging studies reveled tht the tumor ppered s solid sellr nd suprsellr mss, independent of the third ventricle, nd ppered to mimic pituitry mcrodenoms. Moreover, the epicenter of the mss s ppered to originte in the sell, nd not from the lmin terminlis in the nterior third ventricle. To the est of our knowledge, 23 cses of suprsellr CG hve een reported. Of these, only few cses of CG were loclized to the suprsellr region without ventriculr involvement (4), s seen in this cse. Furthermore, only one cse ws reported to e loclized to the sellr nd suprsellr regions without ventriculr involvement, mimicking pituitry mcrodenom, s oserved in our cse (4). On MRI, clssic CG cn e clerly distinguished from the pituitry glnd nd stlk, excluding the possiility of pituitry denom (6). However, in the current cse, the tumor could not e distinguished from the pituitry glnd nd stlk, prompting the preopertive dignosis of pituitry mcrodenom. This unique loction of current tumor leds to confusion with other sell nd suprsell pthologies. Vrious pthologic entities cn rise in the sellr or suprsellr region, nd common etiologies of msses involving oth the sellr nd suprsellr spces include pituitry mcrodenom, crniophryngiom, Rthke cleft 120
5 cyst, nd meningiom (9). Pituitry denoms re the most common lesion involving the suprsellr region. Although their chrcteristics of signl intensity or enhncement re not distinctive, fetures such s sellr enlrgement nd erosion, cvernous sinus invsion, nd loulted mrgins re helpful in dignosing pituitry denoms (9). Crniophryngioms often hve degenertive clcifiction nd prominent cystic component. In ddition, In ddition, one-third of crniophryngioms re heterogeneously hyperintense on T1-weighted MRI scns nd show heterogeneous contrst enhncement in the solid portions nd cyst wll. In contrst, CGs rrely show clcifiction, nd stereotypiclly show homogenous contrst enhncement (9). On CT, CG my e confused with meningiom owing to its hyperdensity, nd homogenous strong enhncement fter contrst dministrtion. Moreover, the reltive isointensity of CG is similr to tht of meningiom in T2- weighted imges. However, the ilterlly symmetric posterior extension of vsogenic edem into the optic trcts my fcilitte the differentil dignosis (2). Moreover, meningioms frequently crete n otuse ngle with the tuerculum or dorsum selle (67%), show the durl til sign or meningel enhncement (68%), nd re commonly ssocited with hyperostosis when the sell is involved (34%) (9). CGs re reported to hve poor prognosis. It ws reported tht 74% of ptients with CG died from postopertive complictions or tumor recurrence; the most common cuse of deth ws pulmonry emolism (10). Sutotl resection is ssocited with the risk of recurrence. Adjuvnt rdition therpy hs een performed in limited numer of cses; Koyshi et l. (10) reported effective residul tumor control with gmm knife rdiosurgery in three ptients. We present n unusul cse of CG locted in the pituitry foss nd suprsellr region without involvement of the third ventricle, which ppered to mimic pituitry mcrodenom. We recommend tht the differentil dignoses for lesions in the sellr nd suprsellr regions should e extended to include CGs in the list of possile etiologies. Acknowledgments This work ws supported in prt y the Soonchunhyng University Reserch Fund. REFERENCES 1. Brt DJ, Scheithuer BW, Stugitis SM, Cortez SC, Brecher K, Burger PC. Third ventriculr chordoid gliom: distinct clinicopthologic entity. J Neuropthol Exp Neurol 1998;57: Pomper MG, Psse TJ, Burger PC, Scheithuer BW, Brt DJ. Chordoid gliom: neoplsm unique to the hypothlmus nd nterior third ventricle. AJNR Am J Neurordiol 2001;22: Kim JW, Kim JH, Choe G, Kim CY. Chordoid gliom: cse report of unusul loction nd neurordiologicl chrcteristics. J Koren Neurosurg Soc 2010;48: Suh YL, Kim NR, Kim JH, Prk SH. Suprsellr chordoid gliom comined with Rthke's cleft cyst. Pthol Int 2003;53: Psquier B, Peoc'h M, Morrison AL, et l. Chordoid gliom of the third ventricle: report of two new cses, with further evidence supporting n ependyml differentition, nd review of the literture. Am J Surg Pthol 2002;26: Grnd S, Psquier B, Gy E, Kremer S, Remy C, Le Bs JF. Chordoid gliom of the third ventricle: CT nd MRI, including perfusion dt. Neurordiology 2002;44: Al Hini QS, Petrecc K. Rrest of the rre: chordoid gliom infiltrting the optic chism. Surg Neurol Int 2011;2:53 8. Cencchi G, Roncroli F, Cersoli S, Ficrr G, Merli GA, Gingspero F. Chordoid gliom of the third ventricle: n ultrstructurl study of three cses with histogenetic hypothesis. Am J Surg Pthol 2001;25: Donovn JL, Nesit GM. Distinction of msses involving the sell nd suprsellr spce: specificity of imging fetures. AJR Am J Roentgenol 1996;167: Koyshi T, Tsugw T, Hshizume C, et l. Therpeutic pproch to chordoid gliom of the third ventricle. Neurol Med Chir (Tokyo) 2013;53:
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