Dorsal extradural meningioma: Case report and literature review

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OPEN ACCESS For entire Editoril Bord visit : http://www.surgiclneurologyint.com Editor: Jmes I.

1 OPEN ACCESS For entire Editoril Bord visit : Editor: Jmes I. Ausmn, MD, PhD University of Cliforni, Los Angeles, CA, USA Cse Report Dorsl extrdurl meningiom: Cse report nd literture review Soheil Rysi Dehcordi, Alessndro Ricci, Alessndro Chiominto 1, Dnilo De Pulis, Hmr Di Vitntonio 2, Rento J. Glzio Opertive Unit of Neurosurgery, 1 Deprtment of Pthology, Sn Slvtore Hospitl, University of L Aquil, L Aquil, Itly, 2 MESVA Deprtment, University of L Aquil, L Aquil, Itly E mil: *Soheil Rysi Dehcordi soheil.rysi@lice.it; Alessndro Ricci lex.ricci@emil.it; Alessndro Chiominto lessndro.chiominto@univq.it; Dnilo De Pulis d.depulis@lice.it; Hmr Di Vitntonio hmr.divitntonio@gmil.com; Rento J. Glzio rento.glzio@cc.univq.it *Corresponding uthor Received: 29 Mrch 16 Accepted: 23 June 16 Pulished: 23 August 16 Astrct Bckground: Extrdurl spinl mss lesions re most commonly metsttic tumors. Extrdurl meningioms re rre, ccounting for pproximtely % of spinl meningioms; intropertively, they re esily mistken for mlignnt tumors, especilly in the en plque vriety, resulting in indequte surgicl tretment. Cse Description: Our cse is one of the first to descrie ptient with two purely extrdurl meningioms, one ech etween D3 D4 nd etween D5 D6 verterl levels. Surgicl resection ws rdicl, nd pthologiclly oth lesions were meningothelilmeningioms. Conclusions: Reviewing the literture, we discuss the pthogenesis, tretment strtegies, nd long term ehvior of these uncommon lesions. Access this rticle online Wesite: DOI: / Quick Response Code: Key Words: Extrdurl extrmedullry neoplsms, extrdurl meningiom, extrdurl spinl tumors INTRODUCTION Verteromedullry tumors re clssified s either extrdurl or intrdurl. Intrdurl tumors re further divided into intrmedullry or extrmedullry. The most common intrdurl extrmedullry neoplsms re schwnnoms, neurofiroms, nd meningioms. Extrdurl tumors re most commonly metsttic lesions. [38] Meningioms ccount for 25 46% of primry spinl tumors, [14] with pek ge of yers, femle rtio = 4:1 overll, nd the most frequent loction in the thorcic spine. [24] Extrdurl spinl meningioms re very rre nd ccount for only % of ll spinl meningioms. [40,51,66] Multiple spinl meningioms lso occur rrely. We report rre cse of two purely extrdurl thorcic spine meningioms in young womn suffering from meningiomtosis. We discuss the fetures nd clinicl mngement of entirely extrdurl meningioms. CASE REPORT A 39 yer old womn, suffering from meningiomtosis, ws operted in our institute severl times. The first opertion ws performed in 2006, when she underwent exeresis of prmedin meningiom (meningothelilmeningiom) rising in the right prmedin region. In 2009, she presented with grn ml seizure nd left hemipresis; mgnetic resonnce imging (MRI) reveled multicentric meningioms in the right prietl prmedin nd ilterl flcine This is n open ccess rticle distriuted under the terms of the Cretive Commons Attriution NonCommercil ShreAlike 3.0 License, which llows others to remix, twek, nd uild upon the work non commercilly, s long s the uthor is credited nd the new cretions re licensed under the identicl terms. For reprints contct: reprints@medknow.com How to cite this rticle: Dehcordi SR, Ricci A, Chiominto A, De Pulis D, Di Vitntonio H, Glzio RJ. Dorsl extrdurl meningiom: Cse report nd literture review. Surg Neurol Int 2016;7: Surgicl Neurology Interntionl Pulished y Wolters Kluwer - Medknow

Surgicl Neurology Interntionl 2016, 7:76 regions. These lesions were prtilly removed nd the postopertive course ws good, with n improvement in her seizures nd wekness.

Since Ferury 2015, she noted progressive numness nd wekness in oth lower extremities with git disturnce. She denied sphincter disturnce.

Another extrmedullry mss ws locted etween D5 D6 verterl levels, extending to the right D5 D6 formen [Figure 2].

2 Surgicl Neurology Interntionl 2016, 7:76 regions. These lesions were prtilly removed nd the postopertive course ws good, with n improvement in her seizures nd wekness. In ddition, in this cse, the histologicl dignosis ws meningothelilmeningiom. Therefore, fter opertion, the ptient ws sujected to rdiotherpy nd close neurordiologic follow up. Since Ferury 2015, she noted progressive numness nd wekness in oth lower extremities with git disturnce. She denied sphincter disturnce. Spine mgnetic resonnce imging (MRI) showed n extrmedullry mss, locted etween D3 D4 verterl levels, extending to the left D3 D4 formen [Figure 1]. Another extrmedullry mss ws locted etween D5 D6 verterl levels, extending to the right D5 D6 formen [Figure 2]. Both lesions were hypointense to the spinl cord on T1 weighted imges, T2 hyperintense, displyed importnt enhncement fter Gdolinium injection, nd compressed the spinl cord dorslly. Neurologicl exmintion reveled severe prpresis with motor strength of 2/5 in oth legs (left more thn right) nd T6 sensory level ilterlly. A mild wekness in her rm ws lso noted (outcome of previous intervention). The following deep tendon reflexes were exggerted: Bilterl knee jerk, ilterl nkle jerk, nd ilterl medil hmstrings. Left sided Binski reflex ws noted. Generl physicl exmintion ws norml. Opertion Using n opertive microscope, the ptient underwent rdicl surgicl excision of oth lesions y D4 lminectomy nd D5 left prtil hemilminectomy. Two purely extrdurl tumors were found intropertively; lesions were found to e gry, with hrd consistency, long with clcifictions nd signs of one erosion. Intropertive findings were suggestive of metsttic extrdurl nd verterl secondry loction (even if no primry lesion ws found in the preopertive lortory nd rdiologicl evlution). Extrdurl msses were completely removed nd durl sements were cuterized [Figures 3 nd 4]. Pthologicl exmintion of msses reveled, in oth cses, meningothelilmeningiom [Figure 5]. Ki67 ws expressed in 2% of neoplstic cells. Postopertively, there ws mrked improvement in the muscle strength of the lower extremities; follow up neuroimging reveled complete removl of lesions nd no spinl instility. No pprent tumor recurrence or regrowth ws detected in follow up study 6 months lter. Figure 1: () Sgittl mgnetic resonnce (MR) imge showing n enhncing extrmedullry mss locted etween D3 D4 verterl levels; () xil MR imge showing n enhncing extrmedullry mss with spinl cord displcement nd compression, nd extending to the left D3 D4 formen Figure 2: () Sgittl mgnetic resonnce (MR) imge showing n enhncing extrmedullry mss locted etween D5 D6 verterl levels; () xil MR imge showing n enhncing extrmedullry mss with spinl cord displcement nd compression, nd extending to the right D5-D6 formen DISCUSSION Exclusively extrdurl meningioms re very rre, ccounting for % of spinl meningioms. [40,45,51,66] Reviewing the literture, 44 studies hve descried 100 ptients with extrdurl spinl meningiom [Tle 1]. Figure 3: After D4 lminectomy, the mss ws removed (); the lesion ws completely extrdurl (); the underlying dur mter ws intct ()

Figure 4: After D5 left prtil hemilminectomy, the mss ws removed, it ws completely extrdurl (); the surgicl field t the end of meningioms exeresis: the dur mter ws comletely intct () However, ecuse

3 Surgicl Neurology Interntionl 2016, 7:76 c Figure 5: Histology. Hemtoxylin nd eosin stining () shows the histologic pttern of meningothelil meningioms nd polygonl cells with ill-defined cytoplsm, immunohistochemiclly EMA+ (), with low Ki67 prolifertion index (c) Figure 4: After D5 left prtil hemilminectomy, the mss ws removed, it ws completely extrdurl (); the surgicl field t the end of meningioms exeresis: the dur mter ws comletely intct () However, ecuse mny reports used different criteri to differentite etween prtilly nd completely extrdurl lesions, it is difficult to get their rel frequency, which my e lower thn tht lredy presented.[45,64] In our literture review, ptients with extrdurl meningioms, rnged in ge from 8 to 76 yers old (men, 41 yers); the mjority of ptients were femle (60%), nd the lesions occurred most commonly in the thorcic spine.[9,10,22,35,36,58] These chrcteristics, s well s histology nd clinicl ehvior, seem to e no different from their intrdurl counterprts.[45,52] Severl theories hve een postulted for the pthogenesis of purely extrdurl meningioms. They re elieved to rise from ectopic rchnoid cells round the perirdiculr nerve root sleeve, where the spinl meninx merges directly into the dur,[55,68] s seen with other extrcrnil meningioms such s the nose or skin.[20,54] Other uthors hve postulted tht the perirdiculr dur, eing less thick, my contin vestigil remnnts of the superficil lyers of the emryonl rchnoid mter nd villi, explining the extrdurl loction nd root proximity of some meningioms.[55,64] It hs lso een suggested tht islnd of rchnoidl tissue might migrte into the extrdurl spce, s seen with introritl meningioms tht hve no ssocition to the sheth of the optic nerve.[20,54,55,64] Concerning neuroimging, MRI is the technique of choice for the dignosis of spinl meningioms; it clerly defines the mss nd its reltion to the spinl cord.[37,65] Mostly, the lesion ppers iso or hypointense on T1 MRI imges, hyperintense on T2 sequences, with homogeneous enhncement fter Gdolinium injection. Klekmp nd Smii sustined tht MRI imging led to erlier dignosis of spinl meningioms y 6 months.[29] Prior to the dvent of MRI imging, mielogrphy ws the est imging technique for the dignosis of spinl meningioms.[23,30] Bone window computed tomogrphy (CT) nd/or Rdiogrphy my show clcifictions, one destruction or erosion, increse in interpediculr distnce, etc.[64] These rdiogrphic chrcteristics, ssocited with extrdurl loction, gross intropertive ppernce, nd rpid progression of symptoms my induce mny investigtors to confuse extrdurl meningioms with extrdurl spinl metstses, especilly in the cse of the en plque vriety.[64] In our cse, we performed gross totl resection, however, this is not lwys possile nd the erroneous ide of metsttic/mlignnt lesion my lter surgicl pproch, leding some surgeons to perform two surgicl opertions; t first pllitivesurgery for presumed metsttic lesion, nd lter, second opertion for complete resection of the tumor.[64] Therefore, intropertive histology is mndtory for choice of the tretment strtegy; if the intropertive dignosis is unequivocl for meningiom, the surgeon should consider gross totl exeresis of the tumor. Bsed on the pthogenetic hypothesis tht extrdurl meningioms rise from the durl sleeve nd not from the externl surfce of the dur, these tumors cn e stripped off from the spinl dur, without the need to excise the dur, s demonstrted y Svrdekr.[55]

4 Surgicl Neurology Interntionl 2016, 7:76 Tle 1: Review of cses of epidurl meningioms descried in literture Article Author Epidurl with n intrdurl component/totl Exclusively epidurl Age Gender Tumor loction Bony chnges on Neuroimging Tissier, 1898 [62] 1 14 M C2-C5 Soderergh nd Sunderg, 1916 [56] 1 59 M C3-C4 Mss, 1918 [36] 1 54 F D4-D6 Nffziger nd H, 1933 [42] 4 54 M D8 60 F D9 55 M C6-D2 34 F C7-D2 Enderle, 1934 [18] 1 44 M L3-L4 Noisi, 1936 [43] 1 Ingrhm, 1938 [27] 1 10 M C3-C5 Rsmussen, 1940 [48] 10/140 Elserg, 1941 [17] 4/73 Oddsson, 1947 [45] 1 32 M L/S Bull, 1953 [9] 6/59 Pedicle destruction (1 cse) Henschen, 1955 [26] 3 42 F C5 17 F D1 31 M D5 Arseni, 1958 [3] 9/114 Rnd, 1960 [47] F dorsl Destruction of left D6-D7 hemilmin nd erosion of pedicle Lomrdi, 1961 [35] 3/71 3 Widened interverterl formen Hft, 1963 [25] M D5-D7 Norml Erly, 1966 [16] M D3-D5 Erosion of D4 ody Soo, 1966 [58] F D4-D6 Norml 0 7 M C1-D7 Widened pedicles Vkili, 1967 [64] 1 61 M D1 Pecker, 1967 [46] 3 25 F C2-C5 Norml 43 F C6-D1 Enlrgement of C7-D1 formen 65 F D4-D6 Erosion of D3-D5 pedicles Rth, 1967 [49] F C3-C6 Norml Aott, 1968 [1] M D4 Hllpike, 1968 [24] M D6-D7 Erosion of the right D6 pedicle Singh, 1968 [55] 1 35 F D4 Fortun, 1969 [19] F D7-D F D7-D F D4-D M D1-D2 Mittl, 1960 [40] 1 40 F D12 Blprmeswrro, 1970 [5] 3/14 40 F L3-L5 54 F L4-L5 30 F D3-D4 Clogero, 1972 [10] F D M L F D9-D M D7 Borghi, 1973 [7] 5 28 F C7-D2 Erosion of C7 nd D1 pedicles 0 23 F D1 Norml 32 F C5-D1 Norml 0 28 F D5-D6 D6 pedicle thinned Contd...

5 Surgicl Neurology Interntionl 2016, 7:76 Tle 1: Contd... Article Author Epidurl with n intrdurl component/totl Exclusively epidurl Age Gender Tumor loction Bony chnges on Neuroimging 60 F D3-D4 Erosion of pedicle Bret, 1976 [8] 2/60 Srtor, 1977 [52] M C1-C4 Roux, 1996 [50] 7/54 5 King, 1998 [31] 4/78 2 Kumr, 1980 [32] M D6 Motomochi, 1980 [41] 1 14 M C4-C7 Stern, 1980 [60] F D3-D M D2-D6 56 F D2-D3 Ky, 1982 [28] 1 11 M C4-C7 Levy, 1982 [34] 7/97 0 In 2 cses, verterl posterior rc nd pedicle disruption Milz, 1983 [39] F D4-D6 Norml 1 45 F D8 Norml Kyoushim, 1987 [33] 1 Stechison, 1987 [59] F D5-D6 Clificrchnoiditis 63 F D12-L1 Solero, 1989 [57] 9/174 9 Chen, 1992 [11] 1 14 F C2-C6 Di Rocco, 1994 [15] 1 14 F D6-D7 Christopherson, 1997 [12] 1 13 F D2-D3 Sto, 1997 [53] M C1-C3 Yoshiur, 1998 [67] F C2-C4 Achri, 2000 [2] 1 Gmche, 2001 [21] 1 Messori, 2002 [38] F C5-C7 Clcifictions Zevgridis, 2002 [68] F D11-D12 Cohen Gdol, 2003 [13] 7/40 0 Tkeuchi, 2006 [61] M C1-C4 Enlrgement of the C3-C4 formen Ymd, 2007 [66] F C1-C5 Clcifiction in the spinl cnl; tumor infiltrtion over oth the sides of the trnsvers processes Brner, 2007 [6] F C7-D1 C6-C7 verterl ony destruction Frnk, 2007 [20] F C5-C7 Enlrgement of C6-C7 formen Sntigo, 2009 [51] M D2-D3 Bone remodelling of the left posterior segment of the D3 ody Tuli, 2012 [63] F D4-D6 Norml Svrdekr, 2014 [54] F C3-C6 C4 nd C5 lterl spinous processes infiltrtion 23 F D4-D5 Norml Nsir, 2014 [44] M D5 T5 ody signls chnges M: Mle, F: Femle. Blnk cells in column 3 indicte tht we hd no informtion if the lesion ws only epidurl or intr nd epidurl; Blnk cells in column 7 indicte tht there ws no mention of ony chnges in the text Vrious studies hve provided contrdicting reports for the long term prognosis of ptients with extrdurl spinl meningioms. Some uthors hve sserted the ggressive ehvior of these tumors, others hve demonstrted these meningioms to e enign. [64] However, we suggest tht their ehvior is relted to their pthologicl World Helth Orgniztion (WHO) grde, nd d prognosis my e cused y difficulty in gross totl resection of the tumor ecuse of its ony involvement nd/or prspinl extension, [55] s well s y the genetic predisposition to form neoplsms. A complete or prtil loss of chromosome 22 ws noted in more thn 50% of ptients with spinl meningioms. [4,23,31] Arslnts sustined reltionship etween some normlities of cncer relted genes locted on 1p, 9p, 10q, nd 17q nd the etiology of spinl meningioms. [4]

6 Surgicl Neurology Interntionl 2016, 7:76 CONCLUSION Purely extrdurl meningioms re very infrequent ut they should e included in the differentil dignosis of extrdurl lesions. They re esily mistken pre nd intropertively for metsttic tumors, with possile consequences on the proposed surgicl tretment, nd therefore, on the moridity. Becuse prognosis is relted to the extent of resection, we elieve it would e etter to completely remove the lesion, if considered sfe. In cse of douts, n intropertive pthology support could e useful. Finlly, ecuse of opposing views in the long term prognosis nd rte of surgicl cure, it is very importnt to e wre of these lesions, undergoing ptients to very close follow ups. Finncil support nd sponsorship Nil. Conflicts of interest There re no conflicts of interest. REFERENCES 1. Aott M, Killeffer FA, Crndll PH. Melnotic meningiom: Cse report. J Neurosurg 1968;29: Achri G, Behri S, Mishr A, Pndey R, Jin VK. Extrdurl meningiom en plque of the cervicl cord. 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Springfield: Chrles C Thoms; Rsmussen TB, Kernohn JW, Adson AW. Pthologic clssifiction, with surgicl considertion, of intrspinl tumors. Ann Surg 1940;111: Rth S, Mthi KV, Chndy J. Multiple meningioms of the spinl cnl. Cse report. J Neurosurg 1967;26: Roux FX, Ntf F, Pinudeu M, Borne G, Devux B, Meder JF. Intrspinl meningioms: Review of 54 cses with discussion of poor prognosis fctors nd modern therpeutic mngement. Surg Neurol 1996;46: Sntigo BM, Rodei P, Cunh E S M. Extrdurl thorcic spinl meningiom. Neurol Indi 2009;57:98.

7 Surgicl Neurology Interntionl 2016, 7: Srtor K, Fliedner E, Pfingst E. Angiogrphic demonstrtion of cervicl extrdurl meningiom. Neurordiology 1977;14: Sto N, Sze G. Extrdurl spinl meningiom: MRI. Neurordiology 1997;39, Svrdekr A, Chtterjee D, Chtterjee D, Dhndpni S, Mohindr S, Slunke P. Totlly extrdurl spinl en plque meningioms Dignostic dilemms nd tretment strtegies. Surg Neurol Int 2014;5(Suppl 7): Singh R, Coerkmp G, Luyendijk W. Spinl epidurl meningioms. Act Neurochir 1968;18: Soderergh G, Sunderg C. Atrophy of the smll muscle of the hnd through compression of upper cervicl cord. Hygie 1916;78: Solero CL, Fornri M, Giomini S, Lsio G, Oliveri G, Cimino C, et l. Spinl meningioms: Review of 174 operted cses. Neurosurgery 1989;25: Soo LY. Spinl epidurl meningiom. South Med J 1966;59: Stechison MT, Tsker RR, Wortzmn G. Spinl meningiom en plque. Report of two cses. J Neurosurg 1987;67: Stern J, Wheln MA, Correll JW. Spinl extrdurl meningioms. Surg Neurol 1980;14: Tkeuchi H, Kuot T, Sto K, Hirose S. Cervicl extrdurl meningiom with rpidly progressive myelopthy. J Clin Neurosci 2006;13: Tissier H. Compression lente de l moelle. Bull Soc Ant Pris 1898;73: Tuli J, Drzymlski DM, Lidov H, Tuli S. Extrdurl En Plque Spinl Meningiom with Intrneurl Invsion. World Neurosurg 2012;77:e Vkili H. The Spinl Cord. New York: Interntionl Medicl Book Co; Vrgs MI, Au Eid M, Bogorin A, Beltechi R, Boyer P, Jvier RM, et l. Spinl extrdurl meningioms: MRI findings in two cses. J Neurordiol 2004;31: Ymd S, Kwi S, Yonezw T, Msui K, Nishi N, Fujiwr K. Cervicl extrdurl en plque meningiom. Neurol Med Chir 2007;47: Yoshiur T, Shrier DA, Pilcher WH, Ruio A. Cervicl spinl meningiom with unusul MR contrst enhncement. AJNR Am J Neurordiol 1998;19: Zevgridis D, Thomé C. Purely epidurl spinl meningiom mimicking metsttic tumor: Cse report nd review of literture. Spine 2002;27:E403 5.

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