Ambiguity in the Dural Tail Sign on MRI

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1 SNI: Unique Cse Oservtions OPEN ACCESS For entire Editoril Bord visit : Editor: Ather Enm, M.D., Ag Khn University, Krchi, Sindh, Pkistn Cse Report Amiguity in the Durl Til Sign on MRI Rmesh S. Doddmni, Rjesh K. Meen, Dttrj Swrkr Deprtment of Neurosurgery, All Indi Institute of Medicl Sciences, New Delhi, Indi E mil: *Rmesh S. Doddmni drsdrmesh@gmil.com; Rjesh K. Meen drrjeshmeen165@gmil.com; Dttrj Swrkr dttrj@gmil.com *Corresponding uthor Received: 02 Septemer 17 Accepted: 30 Jnury 18 Pulished: 19 Mrch 18 Astrct Bckground: Meningioms give rise to the durl til sign (DTS) on contrst enhnced mgnetic resonnce imging (CEMRI). The presence of DTS does not lwys qulify for meningiom, s it is seen in only 60 72% of cses. This sign hs een descried in vrious other lesions like lymphoms, metstsis, hemngiopericytoms, schwnnoms nd very rrely gliolstom multiforme (). The chrcteristics of durl sed s re discussed here, s only eleven such cses re reported in the literture till dte. Here we discuss the unique fetures of this rre presenttion. Cse Description: A 17 yer old mle presented to the emergency deprtment (ED) with, complints of hedche, recurrent vomiting, vision loss in right eye nd ltered sensorium. On exmintion ptient ws drowsy with right hemipresis, secondry optic trophy in the right eye nd ppilledem in the left eye. MRI rin showed, heterogeneous predominntly solid cystic lesion with centrl hypo intense core suggestive of necrosis with heterogeneous enhncement nd positive DTS. Ptient underwent emergency left prsgittl prieto occipitl crniotomy nd gross totl tumor excision including the involved dur nd the flx. On opening the dur, tumor ws surfcing, invding the superior sgittl sinus nd the flx, greyish, soft to firm in consistency with centrl necrosis nd highly vsculr suggesting high grde lesion. Postopertive computed tomogrphy (CT) of the rin showed evidence of gross totl tumor (GTR) excision. The postopertive course of the ptient ws uneventful. Histopthologicl nlysis reveled with PNET like components. The dur s well s the flx were involved y the tumor. Conclusion: s cn rise in typicl loctions long with DTS mimicking meningioms. Excision of the involved dur nd the flx ecomes importnt in this scenrio, so s to chieve GTR. Hence high index of suspicion preopertively ided y Mgnetic Resonnce Imging (MRS) cn help distinguish s from meningiom, therey impcting upon the prognosis. Access this rticle online Wesite: DOI: /sni.sni_328_17 Quick Response Code: Key Words: Durl til sign, gliolstom multiforme, meningiom, posterior third prsgittl This is n open ccess journl, nd rticles re distriuted under the terms of the Cretive Commons Attriution-NonCommercil-ShreAlike 4.0 License, which llows others to remix, twek, nd uild upon the work non-commercilly, s long s pproprite credit is given nd the new cretions re licensed under the identicl terms. For reprints contct: reprints@medknow.com How to cite this rticle: Doddmni RS, Meen RK, Swrkr D. Amiguity in the Durl Til Sign on MRI. Surg Neurol Int 2018;9: Surgicl Neurology Interntionl Pulished y Wolters Kluwer - Medknow

2 INTRODUCTION Durl til sign (DTS) is considered the hllmrk for the rdiologicl dignosis of meningiom. It is seen in 60 72% cses of meningioms nd would represent either direct tumor invsion or rective chnges surrounding the tumor itself. [1,2,4,6,10 12] Durl til hs een reported in the literture in nonmeningiomtous pthologies such s lymphoms/chloroms, durl sed metstsis, hemngiopericytoms, schwnnom, chordoms, pleomorphic xntho strocytoms, nd very rrely gliolstom multiforme (). [3] Literture regrding s presenting with durl til mimicking meningioms is sprse. Here, we report rre cse of with durl til mimicking posterior one third prsgittl meningiom nd review the relevnt literture. CASE REPORT A 17-yer-old mle with no comoridities presented to the emergency deprtment (ED) with complints of hedche nd recurrent vomiting for 2 weeks, vision loss in right eye for 1 week, nd ltered sensorium for 2 dys. On exmintion, the ptient ws drowsy ut rousle, right hemipresis grde 4/5, right-sided secondry optic trophy, nd left-sided ppilledem (pseudofoster Kennedy syndrome). Mgnetic resonnce imging (MRI) of the rin showed, T1-weighted imges heterogeneous predominntly solid (iso-intense) cystic with centrl hypo-intense core suggestive of necrosis [Figure 1]. T2-weighted imges showed, solid (iso-intense) cystic (hyper-intense) with hyper-intense centrl core suggestive of necrosis [Figure 1]. On contrst dministrtion, the lesion demonstrted heterogeneous enhncement with centrl necrosis with positive DTS [Figure 2]. Ptient ws tken up for emergency surgery, nd left prsgittl prieto-occipitl crniotomy fshioned nd gross totl tumor excision ws done. On opening the dur, tumor ws seen surfcing nd invding the superior sgittl sinus s well s the flx, with infiltrtion into the djcent rin prenchym. Tumor ws greyish soft to firm in consistency with centrl necrosis nd highly vsculr suggestive of high-grde lesion. Per-opertively ptient hd trnsient episode of hypotension, which ws mnged. Approximte lood loss ws 2 liters. Postopertive computed tomogrphy scns showed complete tumor removl [Figure 3]. Postopertive recovery ws uneventful nd ptient ws dischrged in stle condition. Finl iopsy reveled with primitive neuro-ectoderml (PNET) like components. DISCUSSION The presence of durl til sign on MRI is highly suggestive of meningiom ut not pthognomonic sign. The presence of durl til in is very rre, nd thorough review of English literture reveled 10 cses of exhiiting DTS mimicking meningiom. The demogrphic nd clinicl dt re listed in Tle 1. All except 2 ptients including the index cse were elderly, suggesting its common occurrence in tht ge group. Meningioms re extr xil tumors, rising from rchnoid cp cells nd prsitize on the durl lood supply with susequent invsion. Approximtely 60 72% Tle 1: Demogrphic nd clinicl fetures of ll the cses reported in literture Authors, yer [ref.] Age/sex Signs/symptoms Tretment Follow up Wilms et l [11] Gupt et l [4] 48/M Seizures NA NA Left hemipresis Left 7 th plsy Hsieh et l [5] 85/F Memory disturnces Agnosi Concurrent WBRT + TMZ Died t 3 months of septic shock following spirtion Wu et l [12] 60/M Left 5 th, 7 th, 8 th nd lower crnil nerves involvement, git disturnce, nd cchexi Ptient refused further tretment Died fter 2 months due to severe cchexi Kyci et l [6] 19/M Hedche nd seizure Not mentioned Not mentioned Ptel et l [8] 57/M Hedche, txi, nd memory Undergoing concurrent Alive disturnces WBRT nd TMZ therpy 60/M Right sided wekness Concurrent WBRT nd TMZ + Bevcizum Died t 28 months of initil dignosis Present 17/M Hedche nd visul prolems with right optic trophy nd left ppilledem Undergoing concurrent WBRT nd TMZ therpy Alive t 3 months TMZ: Temozolomide, WBRT: Whole rin rdiotherpy

3 of meningioms show clssicl DTS. [2] Controversy exists regrding the nture of the dur showing the til sign, with mjority of the pulished studies climing it to e rective chnges, wheres few studies hve shown it to e due to ctul tumorl involvement. [1,2,4,6,10 12] s re intr xil lesions exhiiting ring like contrst enhncement with res of centrl necrosis nd gross perilesionl edem. s presenting s extr xil mss nd DTS is unusul, therey leding to dignostic dilemm in the preopertive period. The criteri for the dignosis of durl til ws given y Aoki et l. [1] which included:. Liner enhncement ws present long the durmter originting from nd extending outwrd from the tumor mrgin. Enhncement ws greter thn elsewhere long the dur c Figure 1: MRI rin. () T1-weighted imges: Posterior one-third iso-to-hypointense lesion utting the flx nd the convexity dur. () T2-weighted imges: iso-to-hyperintense lesion with perilesionl edem. (c) MRS showing choline nd lipid lctte pek c. Findings were present in the two different imging plnes d. There ws greement mong three oservers. All the criteri were fulfilled in our cse, therey confirming DTS leding to the provisionl dignosis of meningiom. Wilms et l. [11] first reported the significnce of the DTS in s through histopthologicl confirmtion of the involved dur. None of the five ptients with the finl iopsy of reported in their series showed invsion of the durl til y the tumor. Hence, they concluded tht the durl til to e just rective chnge rther thn ctul infiltrtion of the tumor. Ten cses of s with DTS hve een reported in the literture till dte, except Wilms et l. [11] none of the other reports included durl iopsy. In the index cse reported here, we hve histologicl confirmtion of the involvement of oth the flx nd the dur y the tumor eyond ttchment. There were lytic chnges on the inner tle of the overlying one, which were drilled wy. Unlike meningioms, s re highly vsculr nd ggressive lesions invding norml rin, deriving their lood supply from pil vsculture. The vessels of durmter rrely feed s, the enhnced durl til sign is likely to develop from vsculr congestion or prolifertion. [3] On the other hnd, meningioms derive their lood supply from durl vessels, mostly externl crotid circultion (ECA) with few exceptions. Ptel et l. [8] demonstrted tumor lush nd ECA supply in oth their cses on ngiogrphy, nd therey misleding the preopertive dignosis s meningiom. They sujected oth the ptients for ngio emoliztion followed y surgery. Blood loss ws less thn 500 ml in oth their cses. Similrly, in one of the cses reported y c Figure 2: Contrst-enhnced MRI in ll three plnes. () Axil imges exhiiting heterogeneous enhncement with DTS (open rrow). () Coronl imges: prsgittl loction with DTS (open rrow). (c) Sgittl imges demonstrting DTS (open rrow)

4 Tle 2: Imging, opertive, nd pthologicl findings of ll the cses reported in the literture Authors/yer [ref.] CT MRI Angiogrm Opertive findings Biopsy Wilms et l. Not specified DTS + Norml dur nd LM, no durl involvement 1991 [11] Not specified DTS + Norml dur nd LM, no durl involvement Not specified DTS + Norml dur ut with, no durl involvement Not specified DTS + Autting dur ut with, no durl involvement Not specified DTS + ECA supply from MMA Autting dur ut with, no durl involvement Gupt et l [4] Not specified T1W: HE T2W: Gd: HE++ DTS + Hsieh et l. Left. temporl T1W: HE 2009 [5] density T2W: HE, cleft sign+, Hypodense Gd: HE++ core (necrosis) MRS lipid/lctte pek+dts + Wu et l. Left CPA T1W 2011 [12] predominntly T2W ± hyper dense lesion Gd HE++ with mixed density DTS + Extending nd enlrging IAC Kyci et l. Not specified Mid 1/3 rd flx 2014 [6] T1W T2W ± Gd HE++ Ptel et l. Not specified Right Temporo prietl mss with 2016 [8] rod contct long rt. tentorium DTS+ CSF cleft sign + Present Not specified Hyper dense lesion, predominntly solid with cystic components in Right Posterior 1/3 rd prsgittl loction Heterogeneous enhncing left prsgittl, mss utting the flx. DTS + T1W HE predominntly solid (isointense) cystic with centrl hypointense core. T2W solid (isointense) cystic (hyperintense) with hyperintense centrl core s/o necrosis. Gd HE++with centrl necrosis, DTS+MRS choline nd lipid lctte peks + Lrge intr xil mss Gr III Astrocytom, durl coming up to rin surfce specimen not sent without durl ttchment, with res of necrosis. Dur externl surfce - norml Inner surfce - invded Hypervsculrity present Grey white geltinous moderte vsculr lesion, dherent to petrous dur, invding proximl 7 th 8 th nerves Petrosqumous rnch of the right middle meningel rtery (MMA) nd Right Occipitl rtery Tumor dherent to flx nd sgittl sinus which ws cogulted, sutotl excision done. Necrotic tumor, sttus of dur not mentioned Left MMA supply Necrotic tumor, sttus of dur not mentioned Inner tle of the overlying one lytic chnges, overlying dur hypervsculr, Gryish tumor with res of necrosis invding the inner surfce of dur nd the sinodurl ngle nd SSS, long with proximl flx. Highly vsculr CPA: Cereellopontine ngle, DTS: Durl til sign, Gd: Gdolinium, : Gliolstom multiforme, LM: Leptomeninges, SSS: Superior sgittl Ssinus, durl involvement not specified, durl specimen not sent MIB 10% with PNET like components MIB % Dur nd flx involved y tumor Wilms et l. [11] ngiogrm ws performed demonstrting feeders from middle meningel rtery (MMA) similr to meningioms [Tle 2]. Angiogrm ws not performed in our cse in view of the emergency setting, ut the tumor hd prsitized the flx nd the convexity dur for its nutrition, s noted during the surgery. The mssive lood loss encountered during the surgery would hve een reduced, y prior ngio emoliztion, s ws the cse with Ptel et l., [8] ut for the emergency sitution. Mgnetic resonnce spectroscopy (MRS) is useful djunct in differentiting preopertively meningioms from s. Mjos et l. [7] studied the role of proton MRS in differentiting vrious tumors nd found lrge lipid/lctte resonnce to e chrcteristic of s nd lrge lnine peks to e chrcteristiclly seen in meningioms. Heish et l. [5] reported cse of mimicking meningiom with the clssicl durl til, where MRS reveled chrcteristic lipid/lctte pek strongly

5 Figure 3: CT rin. () Preopertive contrst CT rin showing heterogeneously enhncing prsgittl lesion. () Postopertive plin CT rin showing gross totl tumor excision suggesting, which ws susequently confirmed on histopthology. The origin of extr xil s hs een mtter of dete. Vrious uthors hve proposed two mechnisms y which these lesions develop. One hypothesis involving of the crnil nerves (CN) sttes tht, rise primrily from the CNS tissue tht ly within the proximl prts of the CN itself. CNS tissue my extend well into the CN, nd isolted islnds of CNS tissue my even e found within the CN t considerle distnce from its exit point. The second hypothesis is tht the tumor originted s primry in the heterotopic neuroglil cell nests in the leptomeninges of the djcent rin. [9] In the index cse, considering the durl nd flcine invsion cn e possily explined y the second hypothesis. CONCLUSION Lesions rising in typicl loctions for meningioms ut with typicl ppernces, should e considered in the differentil dignosis. MRS is very vlule method of differentiting s from meningioms. Preopertive ngiogrphy ppers to hve role in reducing the lood loss lthough not performed in the present cse. High index of suspicion prior to surgery nd excision of the involved durl elements would led to etter outcome. Declrtion of ptient consent The uthors certify tht they hve otined ll pproprite ptient consent forms. In the form the ptient(s) hs/hve given his/her/their consent for his/her/their imges nd other clinicl informtion to e reported in the journl. The ptients understnd tht their nmes nd initils will not e pulished nd due efforts will e mde to concel their identity, ut nonymity cnnot e gurnteed. Finncil support nd sponsorship Nil. Conflicts of interest There re no conflicts of interest. REFERENCES 1. Aoki S, Sski Y, Mchid T, Tniok H. Contrst enhnced MR imges in ptients with meningiom: Importnce of enhncement of the dur djcent to the tumor. AJNR Am J Neurordiol 1990;11: Goldsher D, Litt AW, Pinto RS, Bnnon KR, Kricheff II. Durl til ssocited with meningioms on Gd DTPA enhnced MR imges: Chrcteristics, differentil dignostic vlue, nd possile implictions for tretment. Rdiology 1990;176: Guermzi A, Lfitte F, Miux Y, Adem C, Bonneville JF, Chirs J. The durl til sign eyond meningiom. Clin Rdiol 2005;60: Gupt S, Gupt RK, Bnerjee D, Gujrl RB. Prolems with the durl til sign. Neurordiology 1993;35: Hsieh CT, Liu MY, Tng CT, Sun JM, Tsi WC, Hsi CC. Prolem of durl til sign in gliolstom multiforme? Act Neurol Belg 2009;109: Kyci S, Şengöz A, Köksl V, Gunver F, Kiliç K. Gliolstom multiforme mimicking flx meningiom with chondroplsi. Neurosurgery Qurterly 2014;24: Mjos C, Alonso J, Aguiler C, Serrllong M, Perez Mrtin J, et l. Proton mgnetic resonnce spectroscopy ((1) H MRS) of humn rin tumours: Assessment of differences etween tumour types nd its pplicility in rin tumour ctegoriztion. Eur Rdiol 2003;13: Ptel M, Nguyen HS, Don N, Gelsomino M, Shni S, Mueller W. Gliolstom Mimicking Meningiom: Report of 2 Cses. World Neurosurg 2016;95:624.e9 624.e Reifenerger G, Boström J, Bettg M, Bock WJ, Wechsler W, Kepes J. Primry gliolstom multiforme of the oculomotor nerve. Cse report. J Neurosurg 1996;84: Tokumru A, O uchi T, Eguchi T, Kwmoti S, Kokuo T, Suzuki M, et l. Prominent meningel enhncement djcent to meningiom on Gd DTPA enhnced MR imges: Histopthologic correltion. Rdiology 1990;175: Wilms G, Lmmens M, Mrchl G, Vn Clenergh F, Plets C, Vn Freyenhoven L, et l. Thickening of dur surrounding meningioms: MR fetures. J Comput Assist Tomogr 1989;13: Wu B, Liu W, Zhu H, Feng H, Liu J. Primry gliolstom of the cereellopontine ngle in dults. J Neurosurg 2011;114: