Surgical Neurology International

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1 Surgil Neurology Interntionl SNI: Unique Cse Oservtions, supplement to Surgil Neurology Interntionl OPEN ACCESS For entire Editoril Bord visit : Editor: S. A. Enm, MD Ag Khn University; Krhi, Sindh, Pkistn Comined supr trnsoritl keyhole pproh for tretment of delyed introritl enephloele: A minimlly invsive pproh for n unusul omplition of deompressive rnietomy Lui di Somm, Murizio Iongeli, Dvide Nsi, Polo Bleri 1, Ettore Lupi 1, Rirdo Girotto 1, Griele Polonr 2, Mssimo Serrti Deprtments of Neurosurgery, nd 1 Orl nd Hed Nek Surgery, Umerto I Generl Hospitl, Polytehni University of Mrhe, 2 Deprtment of Rdiology, Setion of Neurordiology, Umerto I Generl Hospitl, Polytehni University of Mrhe, Anon, Itly E mil: Lui di Somm luxgmneuro@liero.it; *Murizio Iongeli neurotr@tisli.it; Dvide Nsi dvidensi83@gmil.om; Polo Bleri pleri@liero.it; Ettore Lupi ettore.lupi@ospedliriuniti.mrhe.it; Rirdo Girotto rirdogirotto@me.om; Griele Polonr g.polonr@univpm.it; Mssimo Serrti mserrti@virgilio.it *Corresponding uthor Reeived: 04 Septemer 15 Aepted: 20 Otoer 15 Pulished: 07 Jnury 16 Astrt Bkground: Introritl enephloele is rre entity hrterized y the hernition of ererl tissue inside the oritl vity through defet of the oritl roof. In ptients who hve experiened hed trum, introritl enephloele is usully seondry to oritl roof frture. Cse Desription: We desrie here se of ptient who presented n introritl enephloele 2 yers fter severe trumti rin injury, treted y deompressive rnietomy nd susequent utologous rnioplsty, without ny evidene of oritl roof frture. The enephloele removl nd the susequent oritl roof reonstrution were performed y using modifition of the suproritl keyhole pproh, in whih we omine n oritl osteotomy with suproritl minirniotomy to filitte view nd ess to oth the nterior rnil foss nd oritl omprtment nd to preserve the lredy osseointegrted utologous rnioplsty. Conlusions: The peulirities of this se re the oritl enephloele without n oritl roof trumti frture, nd the omined minimlly invsive pproh used to fix oth the enephloele nd the oritl roof defet. Delyed introritl enephloele is proly omplition relted to n unintentionl opening of the orit during deompressive rnietomy through whih the rin hernited following the restortion of physiologil intrrnil pressure grdients fter the one flp repositioning. The reonstrution of the oritl roof ws performed y using omined supr trnsoritl minimlly invsive pproh iming t hieving dequte surgil exposure while preserving the utologous rnioplsty, lredy osteointegrted. To the est of our knowledge, this pproh hs not een previously used to ddress introritl enephloele. Aess this rtile online Wesite: DOI: / Quik Response Code: Key Words: Deompessive rnietomy, introritl enephloele, minimlly invsive surgery, piezosurgery This is n open ess rtile distriuted under the terms of the Cretive Commons Attriution-NonCommeril-ShreAlike 3.0 Liense, whih llows others to remix, twek, nd uild upon the work non-ommerilly, s long s the uthor is redited nd the new retions re liensed under the identil terms. For reprints ontt: reprints@medknow.om How to ite this rtile: di Somm L, Iongeli M, Nsi D, Bleri P, Lupi E, Girotto R, Polonr G, Serrti M. Comined supr-trnsoritl keyhole pproh for tretment of delyed introritl enephloele: A minimlly invsive pproh for n unusul omplition of deompressive rnietomy. Surg Neurol Int 2016;7:S trnsoritl-keyhole-pproh-for-tretment-of-delyed-introritl-enephloele:-a-minimlly-invsive-pproh-for-nunusul-omplition-of-deompressive-rnietomy/ S Surgil Neurology Interntionl Pulished y Wolters Kluwer - Medknow

2 INTRODUCTION Introritl enephloele is rre entity hrterized y the hernition of ererl tissue inside the oritl vity through defet of the oritl roof. [1,3,11] The most ommon uses reported in literture re trum, skull se mlformtions, nd tumors. [3,4] In ptients who hve experiened hed trum, introritl enephloele is usully seondry to oritl roof frture. [1 4,8 12] Erly reognition nd tretment re very importnt sine the rised introritl pressure my irreversily dmge the opti nerve nd uses pulsting exophthlmos. [8] Exision of the hernited rin tissue, durplsty nd reonstrution of the oritl roof re the key steps of surgery. [10 12] In lmost ll the reported ses, the fronto sl pproh with wide rniotomy is the preferred pproh. [1 4,8 12] We desrie here se of ptient who presented n introritl enephloele 2 yers fter severe trumti rin injury, treted y deompressive rnietomy nd susequent utologous rnioplsty, without ny evidene of oritl roof frture. The enephloele removl nd the susequent oritl roof reonstrution were performed y using modifition of the suproritl keyhole pproh, in whih we omine n oritl osteotomy with suproritl minirniotomy to filitte view nd ess to oth the nterior rnil foss nd oritl omprtment. The peulirities of this se re the oritl enephloele without n oritl roof trumti frture, nd the omined minimlly invsive pproh used to fix oth the enephloele nd the oritl roof defet. CASE REPORT A 40 yer old femle ws dmitted t our institution following motor vehile ident. The ptient ws unresponsive with Glsgow Com Sle (GCS) sore of 6 (E1V1M4) nd ws put on mehnil ventiltion. The pupils were ilterlly equl nd retive. She hd no perioritl hemtom nd no proptosis. Visul uity nd extroulr musle motility of oth eyes were not ssessed euse of the redued level of onsiousness of the ptient. The immedite postinjury omputed tomogrphy (CT) sn showed only mild, diffuse rin swelling [Figure 1]. Thin slie CT sn with three dimensionl (3D) reonstrution nd oronl nd sgittl setions did not show skull se frtures, in prtiulr, posttrumti d e f Figure 1: ( nd ) The immedite postinjury thin-slie omputed tomogrphy sn with oronl reonstrution showed generlized rin edem without left oritl roof frtures. () Computed tomogrphy sn 24 h fter trum reveled left-sided frontl ontusion using midline shift nd inresed diffuse rin swelling. (d nd e) Postopertive omputed tomogrphy sn showing left deompressive rniotomy nd evution of frontl ontusion with smll ony defet in the roof of the orit (see rrow) proly relted to n unintentionl opening of the orit during keyhole urr hole without evidene of enephloele. (f) Computed tomogrphy sn fter rnioplsty onfirming the smll one opening on the oritl roof (see rrow) S13

3 ony defets on the left oritl roof [Figure 1]. The ptient ws then trnsferred to the Intensive Cre Unit, nd n intrrnil pressure (ICP) proe ws implnted for ICP monitoring. After 24 h, despite mximl medil tretment, the ptient developed intrtle ICP with stle vlues up to 30 mmhg. Hene, she repeted immeditely CT sn tht reveled mssive inrement of the rin swelling, nd left sided frontl ontusion using ontrlterl midline shift [Figure 1]. She underwent left deompressive rniotomy nd evution of frontl ontusion. Immeditely fter deompressive surgery, the ICP deresed signifintly. The immedite postopertive CT sn onfirmed evution of left posttrumti hemorrhge nd resolution of the mss effet [Figure 1d]. At deeper exmintion, this exm lso showed smll ony defet on the oritl roof, undeteted until the ourrene of the oritl enephloele (see the rrow in Figure 1e). During the following dys, the ICP remined stle within norml limits. The ptient reovered well nd ws dishrged from the hospitl on the 25 th posttrumti dy when she ws onsious with GCS sore of 15 nd with only mild right hemipresis. After 2 months, the ptient ws sumitted to utologous one flp repositioning. There were no omplitions during the rnioplsty proedure. Wound heling ws uneventful, nd postopertive neuroimging showed urte fitting of rnioplsty, long with the undeteted ony defet of the oritl roof [Figure 1f]. After rnioplsty, the ptient presented full neurologil reovery, inluded the hemipresis, nd returned to the previous ouption. Two yers following the lst opertion, the ptient omplined left fronto oritl hedhe without ny others oulr symptoms nd/or signs, suh s visul disturne, proptosis, or pulstile exophthlmos. 3D CT sn demonstrted the omplete one fusion of utologous rnioplsty, nd no others intrrnil omplition. However, thin slie oritl CT setions with oronl reonstrution reveled the enlrgement of the supero lterl oritl roof defet with introritl enephloele [Figure 2 ]. Mgneti resonne imging (MRI) onfirmed the hernition of rin prenhym into the left orit through tht ony defet [Figure 2d]. Aurte nmnesti investigtion exluded history of ongenitl nomlies, one dysplsi, or neurofiromtosis (tht ould e ssoited with ongenitl skull se enephloele). Hene, the ptient underwent omined left supr trnsoritl minimlly invsive pproh iming t hieving dequte surgil exposure without ompromising the previously implnted utologous rnioplsty, lredy osteointegrted. A left superior lephroplsty skin inision ws performed to expose the oritl rim. The piezoeletri one slpel (piezosurgery) ws used to perform smll suproritl Figure 2: (-) Thin-slie oritl omputed tomogrphy setions with three-dimensionl nd oronl reonstrution (2 yers fter the rnioplsty) reveled the enlrgement of the lterl oritl roof ony defet ssoited with introritl enephloele. The rrow indites the urr hole, filled y utologous one dust mixed with one sustitute, too lose to the oritl wll. (d) Mgneti resonne imging imge onfirming the hernition of rin mtter into the left orit keyhole rniotomy inluding the supero lterl oritl rim in one piee using the defet of the oritl roof s the posterior edge of the oritotomy [Figure 3 e]. This omined minimlly invsive pproh gve ess to oth the superior oritl vity nd the intrrnil suproritl nterior skull se without the need for rnioplsty to e removed [Figure 3e]. The perioritl nd oritl ontents were identified nd seprted from the hernited ererl tissue. Hernited rin prenhym ws reseted with mirosurgil tehnique reveling the ler mrgin of durl nd ony defet of the oritl roof. The dur ws re pproximted with loked running stith, nd wter tight losure ws hieved. Then, the oritl roof ws reonstruted with utologous one, otined from the outer one prietl lyer ontrlterl to the rnioplsty through smll liner skin inision nd fixed y titnium low profile miniplte nd srews [Figure 3, d, nd f h]. The thin one utting line, performed y piezoeletri slpel, llowed perfet relignment of one stumps [Figure 3]. The postopertive period ws uneventful, nd she ws dishrged from the hospitl 5 dys fter surgery. The estheti results were exellent nd ptient ws le to hieve omplete eye losure within 2 weeks. Postopertive thin slie 3D CT sn showed the stle reonstrution of the oritl roof nd good restortion of roof ontour nd norml oritl volume, exluding intrrnil omplitions [Figure 3f h]. MRI imges 2 months fter surgery onfirmed the omplete redution of enephloele [Figure 3i]. At the lst follow up fter 3 yers, the ptient ws in good helth with no neurologil defiits. d S14

4 d e f g h i Figure 3: Adefghi: Intropertive imges nd postopertive thin-slie omputed tomogrphy sn with three-dimensionl reonstrution. () Left superior lephroplsty inision. ( nd ) Supr-trnsoritl keyhole pproh with the omintion of n oritl osteotomy with suproritl minirniotomy. The use of piezoeletri slpel llows to relize preise nd thin osteotomies, for etter future one heling nd etter estheti result. ( nd d) The one-piee one flp inludes the frontl one, nd the oritl rim using the defet of the oritl roof s the posterior edge of the oritotomy. The one-piee one flp ws then fixed y titnium low-profile miniplte nd srews. (e) Intropertive working re with the exposure of oth oritl nd intrrnil omprtments. (f-i) Postopertive thin-slie omputed tomogrphy sn with three-dimensionl reonstrution nd mgneti resonne imging showing left oritl roof reonstrution with utologous one, otined from the split lvril prietl one ontrlterl to the rnioplsty DISCUSSION Oritl enephloele is rre entity, nd it is hrterized y the hernition of prt of the rin through ongenitl, trumti, neoplsti, or itrogeni oritl roof defet. [1 4,11] The hernition of rin prenhym n lso our through nturl formin suh s the superior oritl fissure or the opti formen. Oritl enephloeles my e ongenitl or quired with ongenitl enephloeles more likely to our in erly hildhood. [3] In the dult popultion, quired enephloeles re reported in ptients who hve experiened hed trum with ssoited oritl roof frture. [3,9,11] However, even trumti oritl enephloeles re quite rre eveniene. There re <25 reported ses in literture thus fr, inluding oth peditri nd dult popultion. [1 4,8 12] In ll these ses, there ws rdiologil or diret intropertive evidene of n oritl roof frture ssoited with the enephloele. The inidene of oritl roof frtures is higher in hildren euse n impt to the oritl rim efore the ge of 7 yers, nnot e dissipted y the frontl sinus (not yet ompletely pneumtized), nd the trumti fore is trnsmitted diretly to the oritl roof. In peditri series, oritl roof frtures were found in 7%, 1% of ptients who suffered hed injuries, nd 13% of those ptients developed introritl enephloele. [3] An oritl enephloele due to trum my develop utely or my develop mny yers lter s the rhnoid nd tissue grdully hernite, or s the frture in the oritl wll grows over time. [1 4,7] Growing frtures of the oritl roof hve een reported severl times, espeilly in peditri popultion. [3] Grdul hernition of the rhnoid into the frture, the physiologi growth of the rnium nd the rin (in hildren), ontinuous pulstion of the CSF, nd the sene of ony ounter ompression re the epted mehnisms of growth nd expnsion of the frture. [1] Our ptient developed the introritl enephloele without ny evidene of oritl roof frture t the initil thin slie CT sn with 3D nd oronl reonstrutions. The first evidene of ony defet in the oritl roof ws undeteted ut present t the immedite CT sn fter deompressive rnietomy, proly relted to n unintentionl opening of the S15

5 orit y the keyhole urr hole. The development of the introritl enephloele ould e relted to the restortion of positive pressure grdient etween the intrrnil omprtment, nd the oritl omprtment fter rnioplsty similr to the etiopthologil mehnism of the growing frture in hildren. To the est of our knowledge, this is the first se of introritl enephloele seondry to deompressive rnietomy nd susequent one replement. The min symptoms nd signs of introritl enephloele re diplopi, exophthlmos, oritl edem, suonjuntivl hemorrhge, restrited movements of the eye, nd loss of vision. Among these, pulstile exophthlmos is the most reported symptom fter trumti introritl enephloele in the literture. [8] Our ptient omplined only left fronto oritl hedhe. Erly reognition nd tretment re very importnt sine the rised introritl pressure my irreversily dmge the opti nerve. [8,11] Thin slie CT sn with 3D reonstrution is the gold stndrd imging modlity to ssess oritl roof frtures or ony defets. However, MRI imging is essentil to detet nd evlute the hernited rin tissue nd for differentil dignosis with others introritl lesions suh s tumors or vsulr mlformtions. [1 3] Exision of the hernited rin tissue, durplsty, nd reonstrution of the oritl roof re the key steps of surgery. [10,12] There re two min routes to the oritl roof: The trnsrnil nd the extrrnil trnsoritl route. [10] The trnsrnil pproh is ommonly performed through ioronl inision nd uni or i frontl rniotomy. This pproh n lso llow to ddress onomitnt intrrnil lesions in the sme session. The extrrnil pprohes to the orit re generlly performed through superior lephroplsty skin inision or through preexisting skin lertion. In ll ut one ses desried to dte in the literture, the hernited rin prenhym into the orit ws essed through wide frontl rniotomy followed y the removl of frtured ony frgments inside the orit. [1 4,8 12] The min limittion of this pproh is the extensive rin exposure nd retrtion tht ould result in ererl prenhym ontusion, infetion, nd epilepsy. [10,12] In our se, to preserve the lredy osteointegrted utologous rnioplsty, we dopted modifition of the suproritl keyhole pproh, y omining n oritl osteotomy with suproritl minirniotomy. This omined minimlly invsive pproh ws used to filitte oth views nd ess to the nterior rnil foss nd oritl omprtment. The one piee one flp inludes the frontl one, nd the oritl rim using the defet of the oritl roof s the posterior edge of the oritotomy. The use of piezoeletri slpel enhne sfety y preventing injuries to nonosseous strutures nd llows to relize preise, thin osteotomies, so filitting the susequent one heling with etter estheti result. [5 7] CONCLUSIONS The peulirities of this se re the development of introritl enephloele without evidene of trumti oritl roof frture nd the omined supr trnsoritl minimlly invsive pproh. The delyed introritl enephloele is proly omplition relted to n unintentionl opening of the orit during deompressive rnietomy through whih the rin hernited following the restortion of physiologil ICP grdients fter the one flp repositioning. The reonstrution of the oritl roof ws performed y using omined supr trnsoritl minimlly invsive pproh iming t hieving dequte surgil exposure while preserving the utologous rnioplsty, lredy osteointegrted. To the est of our knowledge, this pproh hs not een previously used to ddress introritl enephloele. Finnil support nd sponsorship Nil. Conflits of interest There re no onflits of interest. REFERENCES 1. Antonelli V, Cremonini AM, Cmpossi A, Psrell R, Zofre G, Servdei F. Trumti enephloele relted to oritl roof frtures: Report of six ses nd literture review. Surg Neurol 2002;57: Bruzek A, Shepherd D, Vn Gompel J, Jentoft M. Piloyti stroytom presenting s n oritl enephloele: A se report. 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