Intrachiasmatic craniopharyngioma: Assessment of visual outcome with optical coherence tomography after complete surgical removal

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1 SNI: Unique Cse Observtions OPEN ACCESS For entire Editoril Bord visit : Editor: S. Ather Enm, M.D., Ph.D., Ag Khn University, Krchi, Sindh, Pkistn Cse Report Intrchismtic crniophryngiom: Assessment of visul outcome with opticl coherence tomogrphy fter complete surgicl removl Ricrdo Gil-Simoes, José M. Pscul, Andrés P. Css 1, Rfel G. de Sol Deprtments of Neurosurgery nd 1 Ophthlmology, L Princes University Hospitl, Mdrid, Spin E mil: *Ricrdo Gil-Simoes rgs1984rgs@gmil.com; José M. Pscul jmpsncj@hotmil.com; Andrés P. Css.perezcss@correo.sludmdrid.org; Rfel G. de Sol neurorgs1@gmil.com *Corresponding uthor Received: 23 August 18 Accepted: 29 October 18 Published: 21 Jnury 19 Abstrct Bckground: Optic chism invsion by crniophryngiom (CP) is exceptionl. Surgicl tretment of intrchismtic CPs ssocites high risk of chism injury, which should be properly ddressed before surgery. Cse Description: We present 46 yer old womn dmitted to the hospitl with low visul cuity (0.1 in the right eye nd 0.5 in the left) nd severe defect in her visul fields, in ddition to hedches, dibetes insipidus, nd long term depressive disorder. Her visul deficit progressed from right homonymous temporl inferior qudrntnopi to n lmost complete loss of vision in both eyes tht only spred the upper nsl qudrnts. Brin MRI showed rounded third ventricle tumor with potbelly expnsion of the optic chism, suggesting chism invsion by the tumor. Opticl coherence tomogrphy (OCT) showed the thinning of the retinl nerve fiber lyer (RNFL) in the superior nd temporl wedges of the right eye nd in the temporl wedge of the left one. The tumor ws completely removed by employing frontotemporl crniotomy nd trnslmin terminlis pproch. Histologicl nlysis showed squmous ppillry CP. Postopertively, significnt worsening of the visul defect ws evidenced on the perimetry, which ws relted to mrked RNFL trophy mesured with OCT, s compred to the preopertive study. The poor long term visul outcome in this ptient correlted well with the results of postopertive OCT. Conclusions: Preopertive nlysis of retinl trophy with optic coherence tomogrphy llows relible ssessment of the ptient s visul outcome in CPs involving the optic chism. Access this rticle online Website: DOI: /sni.sni_292_18 Quick Response Code: Key Words: Crniophryngiom, optic chism, opticl coherence tomogrphy, third ventricle, visul outcome This is n open ccess journl, nd rticles re distributed under the terms of the Cretive Commons Attribution-NonCommercil-ShreAlike 4.0 License, which llows others to remix, twek, nd build 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: Gil-Simoes R, Pscul JM, Css AP, de Sol RG. Intrchismtic crniophryngiom: Assessment of visul outcome with opticl coherence tomogrphy fter complete surgicl removl. Surg Neurol Int 2019;10: Surgicl Neurology Interntionl Published by Wolters Kluwer - Medknow

2 INTRODUCTION Crniophryngioms (CPs) represent complex group of epithelil benign (WHO I) tumors presumbly originted from cell remnnts of the Rthke s pouch. [6,16] CPs my develop t ny point long the pituitry hypothlmus xis, from the sell turcic to the third ventricle (3V). Very often, CPs cuse progressive decrese of visul cuity nd/or constriction of the visul fields relted to the ntomicl distortions these lesions cuse on the optic chism. [21] The primry development of CP within the optic chism or the invsion of this structure by the tumor is, however, n exceedingly rre event. Only few verified intrchismtic CPs hve been reported in medicl literture to dte. [2,5,11] This subgroup of CPs my cuse severe swelling of the optic chism similr to tht observed in optic chism glioms. In the present report, we describe new cse of n intrchismtic CP dignosed in n dult womn who showed progressive worsening of her vision. The tumor ws totlly removed using pterionl trnslmin terminlis pproch. This study presents, in comprehensive wy, the correltion between the ptient s postopertive visul outcome nd the structurl injury cused to her optic pprtus by this intrchismtic CP, evluted through opticl coherence tomogrphy (OCT), useful tool for quntittive ssessment of the structurl dmge of the retinl nerve fiber lyer (RNFL). CASE DESCRIPTION This 46 yer old womn ws initilly dmitted to our hospitl with hedche, progressive decrese in visul cuity, nd fever bove 38.5 C for the lst month, in ddition to polyuri nd polydipsi for the lst 2 weeks. Upon dmission, her neurologicl explortion ws unremrkble. Visul explortion disclosed reduced visul cuity of both eyes, 20/200 in the right eye nd 20/40 in the left one. The initil perimetry evidenced homonymous left inferior qudrntnopi, wheres the funduscopic exm did not revel ny bnormlity. All her bseline endocrine tests were lso norml t this time. The brin MRI displyed 1.5 cm mixed solid cystic lesion occupying the lower 3V nd djcent hypothlmic chismtic region [Figure 1 c]. As the ptient ws seropositive for HIV infection nd hd suffered from lung tuberculosis severl yers before, this lesion ws presumed to correspond to cerebrl tuberculom relted to the HIV infection, nd specific therpy with isonizid nd rifmpicin ws initited. Six months lter, the ptient ws dmitted gin due to visul deteriortion nd symptoms of pnhypopituitrism nd dibetes insipidus. She ws treted with hormone replcement therpy nd lso received psychitric d b e Figure 1: Preopertive mgnetic resonnce imging (MRI) studies. (, b) Initil MRI study, xil () nd coronl (b) T1 weighted scns (1/25/2012), showing heterogeneous solid cystic lesion centered t the chismtic recess of the third ventricle. Note the nterior tongue like tumor protrusion into the optic chism (white rrow). In the coronl imge, there is round hypointense re of similr low intensity s the tumor is visible over the trjectory of the right optic trct (white rrow). (c-f) Preopertive MRI study obtined 6 months lter, just prior to the surgicl procedure (7/13/2012), showing enlrgement of the tumor. The finger like tumor tissue projecting into the optic chism right optic nerve cn be identified (white rrow). (d) Axil T2 weighted preopertive MRI scn, showing hyperintense symmetricl signls following the course of both optic trcts, sign known s moustche sign, which is believed to correspond to optic trct edem (white rrows). (e, f) Coronl nd midsgittl T1 weighted scns demonstrting the strictly third ventricle topogrphy of this crniophryngiom, which hs developed bove n intct third ventricle floor/pituitry stlk (white rrows) ssessment for depressive disorder which hd worsened in the lst yer. Preopertive neuro ophthlmologicl exmintion At this point, thorough neuro ophthlmologicl nd neurordiologicl evlution ws performed. The ptient could only see light with her right eye nd could distinguish hnd movements from 2 m distnce with the left one. Visul fields were impired severely in both eyes with generlized loss of sensitivity in the right one nd spring of the upper nsl qudrnt of the left eye (Figure 2, perimetry on 07/16/2012). A mild blurred temporl border in the left mcul ws observed on funduscopic exm. OCT (SD OCT SPECTRALIS, Heidelberg Engineering, Heidelberg, Germny, Softwre 5.3) ws used to revel slight thinning of the RNFL in the superior nd temporl wedges of the right eye (90 µm on verge) nd trophy of the temporl wedge in the left eye (Figure 3, preopertive OCT). An MRI study showed significnt enlrgement of the 3V lesion, which hd bsl culiflower like nodule nd cystic upper component [Figure 1d f]. The chismtic (suprsellr) cistern ws tumor free, c f

3 b Figure 2: Visul field exm. Correltion between visul field defects nd the ntomicl injury to the visul pthwys cused by the tumor nd surgicl procedure. () Chronologicl evolution of the visul field defects identified in the ptient through consecutive utomted perimetry exms. (b) Illustrtive schemes showing the sequence of ntomicl injuries to the optic chism nd optic trcts, presumbly cused by the tumor nd the surgicl procedure, consistent with the visul defects shown in the corresponding perimetry studies on the left pnel. (c). Artistic illustrtion showing the sptil ntero posterior segregtion of retinl fibers within the optic chism tht ccounts for the visul defects ssocited with intrchismtic crniophryngioms (CPs). The crossed fibers from the retin s upper qudrnts follow their course t more cudl (posterior) position within the chism, wheres those from the retin s lower qudrnts tend to cross t more rostrl or nterior position within the chism. At the initil exm, the ptient presented homonymous left inferior qudrntnopi with worse visul cuity in the right eye (perimetry on 1/26/2012), result congruent with the invsion of the chism s posterior spect nd right optic trct s medil spect (top scheme in B). As the tumor enlrges, it encroches upon wider re of the posterior nd centrl chism, including both optic trcts, thus dmge resulting in severe visul deficit (second perimetry on 07/16/2012) tht only spred the uncrossed temporl inferior fibers from the left retin long the chism s left edge (second scheme from the top in (b) nd brown trct of fibers in (c)). After removl of the CP, the persistence of the visul deficit in the lower qudrnts of the right eye nd in the nsl qudrnts of the left eye (immedite postopertive perimetry on 8/6/2012) ws congruent with irreversible injury to the chism s posterior spect nd the medil spect of the right optic trct (third scheme from the top in B nd blue trct of fibers in C). The lte postopertive perimetry exm (postopertive perimetry on 01/21/2013) showed significntly worsening of the visul defect, probbly due to degenertive chnges in retinl fibers ssocited with gliotic scrring (bottom scheme in B) c nd both the pituitry stlk nd the pituitry glnd were ntomiclly intct. The optic chism ws grossly distorted, showing potbelly downwrd deformtion on coronl sections, due to tumor infiltrtion [Figure 1e]. In ddition, on T2 weighted xil imges, the optic trcts displyed moustche like ppernce, chrcterized by hyperintense signl of edem extending long both optic trcts nd into the djcent hypothlmus [Figure 1d]. Surgicl procedure The tumor ws pproched through right frontotemporl crniotomy. After opening the Sylvin fissure, the optic chism ws exposed. It ws displced ginst the tuberculum selle nd presented quite swollen nd shortened ppernce [Figure 4]. The tumor occupying the 3V ws pproched through the lmin terminlis, which ws found to hve bllooned nd thinned spect protruding bove the optic chism. The lesion could be dissected out from the 3V wlls, except t its solid bsl portion where solid tongue of the tumor infiltrted the right posterolterl region of the chism nd extended into the swollen proximl portion of the right optic nerve [Figure 4]. After severing the intrchism tumor ttchment with creful shrp dissection under microscope mgnifiction, the tumor ws totlly removed. Pthologicl dignosis identified CP of the squmous ppillry vriety [Figure 4b1 nd b2]. Postopertive course nd visul outcome The ptient recovered successfully from the surgicl procedure, lthough her pnhypopituitrism, dibetes insipidus, nd subjective visul disturbnce remined unchnged. Complete removl of the lesion ws confirmed on postopertive MRI [Figure 5], with smll defect or brech t the 3V floor tht extended to the junction of the optic chism nd the right optic trct, just t the site where the tumor infiltrted the chism [Figure 5b nd c]. Follow up neuro ophthlmologicl ssessment 1 week fter surgery showed some improvement in the ptient s visul cuity nd visul fields. Visul cuity returned to its prior initil vlues, from only light perception to 20/60 in the right eye nd 20/40 in the left one. A mrked improvement of vision in the nsl hemi field in the left eye ws observed s well, lthough only prtil recovery t the temporl superior qudrnt of the right

4 Figure 3: Opticl coherence tomogrphy (OCT) exms.anlysis of the chnges in the retinl nerve fiber lyer (RNFL) thickness tht occurred s the result of the chisml dmge ssocited with this intrchismtic crniophryngiom nd the surgicl procedure for its removl. Left pnel: Preopertive OCT confirmed the presence of RNFL thinning involving the superior nd temporl wedges (or qudrnts) of both eyes, more severe in the upper retin of the right eye (yellow wedge). Right pnel: Postopertive OCT demonstrted further thinning of the RNFL in the upper retin of the right eye s well s in the temporl wedge of the left retin (ornge wedges). Such thinning reflected the irreversible dmge of the xonl fibers within the re of the optic chism nd optic trcts infiltrted by the tumor, result indictive of worse prognosis for recovery following the surgery eye occurred (Figure 2, third perimetry on 8/6/2012). Postopertive OCT conducted t this time, however, showed reduction in the RNFL thickness in both eyes, with trophy of the superior wedge in the right eye nd greter degree of trophy in the temporl wedge in the left eye. At the lst visul ssessment, six months fter surgery, significnt worsening of the visul defect ws evidenced on the perimetry (Figure 2, fourth perimetry on 01/21/2013), probbly due to degenertive chnges of retinl fibers ssocited with gliotic scrring. This dmge ws congruent with the postopertive retinl trophy disclosed on the OCT. No tumor recurrence ws demonstrted on the lst postopertive MRI, performed 1 yer fter surgery. DISCUSSION Intrchismtic CPs: An intrventriculr topogrphicl vrint CPs with either primry intrchismtic development or true ntomicl invsion of the optic chism represent quite exceptionl topogrphicl vrint, with very few cses recorded in the literture.[2,5,11,19] In ll these cses, the tumor lrgely occupied the 3V nd presented the tightest nd widest dhesions to the posterior edge of the optic chism, which ws infiltrted by tongue like tumor protrusion. b1 b2 Figure 4: () Intropertive view nd histologicl dignosis. Surgicl view through the right frontl temporl (pterionl) pproch showing the ntomicl deformtion of the optic chism under microscopic mgnifiction. A prefixed, swollen optic chism is observed. Note the bulging of the lmin terminlis (yellow rrow) nd the whitish expnsion of the right optic trct, sign of tumor infiltrtion. A1: A1 segment of the right nterior cerebrl rtery; ACoA: nterior communicting rtery; CA: crotid rtery; M1: M1 proximl segment of the right medin rtery; LON: left optic nerve; RON: right optic nerve; b1 b2: histopthologicl sections showing the distinctive fetures of this squmous ppillry crniophryngiom. Solid res of well differentited strtified squmous epithelium with bsl plisding nd pseudoppille formtion round cores of loose fibrovsculr strom re typicl of this vrint. H nd E, originl mgnifiction 16 (b1) nd 40 (b2) Therefore, ll these lesions corresponded to strictly 3V CPs, which were developed bove n ntomiclly intct 3V floor.[14,15] CPs with primry intr 3V topogrphy usully push the optic chism downwrd ginst the tuberculum selle, cusing the chism to displce to prefixed position, presenting much shorter nd wider ppernce.[21] In our cse, the right hemi chism nd the right optic trct

5 b Figure 5: Postopertive MRI studies. () Midsgittl T1 weighted MRI scn fter totl removl of this intrchismtic crniophryngiom. The outline of the prefixed, downwrd displced optic chism (white rrow) is observed next to the defective or breched third ventricle floor, s the trophic tuber cinereum ws removed long with the tumor. (b, c) Axil nd coronl T1 weighted scns through the level of the optic pprtus. Notice the ntomicl injury to the optic chism right optic trct junction fter removing the lesion (white rrows) were the res observed to be swollen during the surgicl procedure, wheres the tumor spred the left hlf of the optic pprtus [Figure 4]. The intrchismtic development of CP cn be explined following the embryonic theory for the origin of CPs, originlly proposed by Jkob Erdheim in [6,18] He considered these lesions derived from noninvoluted cell remnnts of the crniophryngel duct nd/or Rthke s pouch, cells which cn come into contct with the infundibulr recess before the pi mter is developed to give rise to CP embedded within the neurl tissue of the infundibulum. [3] These lesions usully do not infiltrte the optic chism, owing to the compct consistency of the ltter, but in exceptionl cses, subpil inclusion of Rthke s pouch remnnts long the posterior chism edge could led to the development of n intrchismtic CP. [10,16] MRI findings defining the intrchismtic occuption by CP The optic chism displys n ovl or ellipticl morphology on midsgittl MRI scns in helthy individuls. In contrst, downwrd displcement of this structure with crescent moon shpe on midsgittl MRI represents the fundmentl deformtion produced by CPs developed within the 3V. [21] In the cse of intrchismtic CPs, n extremely thin crescent deformtion of the optic chism is visible on sgittl MRI scns [Figure 1f], lthough on coronl sections, the chism shows bilobed expnsion filled with T1 hypointense tissue, sign termed by Brodsky et l., potbelly deformtion of the optic chism [Figure 1e]. [6] Intrchismtic CPs my lso cuse bitemporl hyperintense liner signls following the trjectory of both optic trcts on xil T2 weighted MRI scns [Figure 1d]. These hyperintensities, known s the moustche sign, my correspond to the edem produced in the hypothlmus nd djcent optic pthwys by CPs expnding ginst the 3V wlls. [7,25] c Chism pthwys injured by intrchismtic CPs: Correltion with visul field deficits Over 2 million nerve fibers pss through the optic chism, in which sptil segregtion of crossed nd uncrossed fibers cn be evidenced. [10,20] The mjority of fibers tht form the uncrossed pthwy rise from the ipsilterl temporl hemiretin nd the crossed pthwy from the contrlterl nsl hemiretin. In the erly 1960s, Hoyt et l. trced the retinl projections in the chism of primtes nd reveled rostrl cudl (ntero posterior) sptil segregtion of retinl fibers within the chism, in which the crossed fibers from the dorsl retin (upper qudrnts) cross the chism t cudl (or posterior) position, wheres those from the ventrl retin (lower qudrnts) tend to cross t more rostrl (or nterior) position [Figure 2c]. [8] Fibers from the mcul represent the mjor bulk of fibers within the chism nd re found more centrlly nd cudlly (within the centrl posterior portion of the chism) thn the rest of fibers. [13] Visul disturbnces ssocited with CPs re minly relted to the prticulr type of ntomicl distortion to the optic chism cused by the tumor. [16,21] In the cse of intrchismtic CPs, topogrphy centered t the chism infundibulum junction, the tumor first encroches upon the posterosuperior nd centrl portion of the optic chism, the re where the crossed fibers from mculr nd upper nsl retinl qudrnts follow their course to the opposite optic trct. [8] Consequently, the chism injury ssocited with this topogrphicl vrint produces the worst visul field defect in the temporl lower qudrnts. The sequentil stges of visul field impirment in our ptient were congruent with the topogrphy nd the ntomicl region of the chism injured by the tumor. At the initil perimetry, the ptient presented homonymous left inferior qudrntnopi, worse in the right eye, deficit explined by dmge to the posterior edge of the optic chism, nd the medil spect of the right optic trct [Figure 2]. This is the region of the chism initilly infiltrted by the tumor [Figure 1], in which the tongue like intrchism tumor extension dmged the xonl bundles coming from the upper nsl qudrnt of the left retin nd the upper temporl qudrnt of the right one [Figure 2c]. The right eye is obviously the one suffering the most severe loss of vision, s the dmge involves significnt mount of the bundles from the fovel nd perimculr res of the right retin, including the uncrossed fibers from the temporl qudrnts. The rpid worsening of ptient s visul deficit over the following 6 months [Figure 2, perimetry on 07/16/2012] ws in correltion with encrochment or invsion of the centrl chism nd right optic trct by the tumor, which spred the uncrossed temporl inferior fibers from the left retin. [Figure 2b, c] At this time, the OCT

6 proved n overll thinning of the RNFL tht involved minly the superior wedges in both retins, result in greement with the tumor invsion of the posterior upper chism region [Figure 3, preopertive OCT]. The OCT lso showed trophy in the left eye s temporl wedge which is not well explined by the tumor extension into the chism, lthough this finding hs been frequently reported for chism compressions by mcrodenoms. [9] Surgicl removl nd visul outcome of intrchismtic CPs: The predictive vlue of OCT Intrchismtic CPs essentilly represent prticulr vrint of 3V CPs which re infiltrting the optic chism. [17,21 23] Consequently, the surgicl removl of these lesions requires the use of n pproch to the 3V providing n optiml view of the tumor ttchment to the optic chism. The frontotemporl or pterionl pproch, besides providing n optiml view of the optic chism nd djcent cisternl spces, lso permits the opening of the lmin terminlis for the removl of the intr 3V component of the tumor. [16,17] The use of trnslmin terminlis pproch llowed us to remove the intrchismtic CP described here, through the relese of the tumor ttchment to the chism under microscopy mgnifiction. One week fter the procedure, the first postopertive perimetry exmintion showed significnt improvement in visul cuity in the nsl field of the left eye but only prtil recovery of vision in the temporl superior qudrnt of the right eye (Figure 2, perimetry on 08/06/2012). These findings re concordnt with n irreversible lesion of the xonl pthwys t the chism right optic trct junction, the re infiltrted by the tumor, where residul ntomicl defect is evident on the postopertive MRI [Figure 5]. Apprently, the inferior nsl fibers from the right retin tht cross the chism through its nterior edge, in ddition to the temporl uncrossed fibers of the left eye, seemed to hve been spred following CP removl [Figure 2b]. The results of the postopertive OCT performed round this time showed, however, tht the degree of trophy in the RNFL hd progressed s compred to the prior study, especilly t the superior re of the right retin nd the temporl one in the left eye [Figure 5]. Six months lter, mrked deteriortion of the visul sttus ws confirmed in the perimetry, with preservtion of islnds of vision only in the upper temporl qudrnts of both eyes, result congruent with severe bilterl RNFL trophy (Figure 2, perimetry on 01/21/2013). Progressive degenertion of the retinl pthwys occurred within few months fter the surgicl procedure, process probbly linked to the development of glil scr t the site of the intrchismtic wound. No conclusive explntions regrding the initil incongruence between the erly postopertive visul improvement but worsening OCT results in this ptient cn be stted. The greter trophy in the RNFL immeditely following the surgicl procedure would logiclly reflect the injury to the chism ssocited with the surgicl mneuvers employed to dissect the intrchismtic tumor ttchment. RNFL thinning reflects irreversible retinl gnglion cell dmge, which usully occurs fter prolonged compression of the visul pthwys. [4,9,24] Hence, ny structurl dmge of the retin detectble with OCT predicts the existence of irreversible injury t the optic chism/optic trcts tht will ssocite visul loss to be confirmed on stndrd utomted perimetry [1,12,24] Despite the pprent prtil visul recovery observed on our ptient s initil postopertive perimetry, the OCT lredy reflected structurl dmge in the retinl gnglion cells, which hd dvnced with respect to the preopertive study nd nticipted long term worsening of visul function. The finl question to consider is the degree of removl to be ttempted when deling with CP tht infiltrtes the optic chism. A generlized surgicl ttitude cnnot be recommended, nd the most pproprite, judicious decision should be estblished on n individul bsis. The high risk of irreversible chism injury ssocited with rdicl removl of CP must be pondered with the rte of tumor recurrence, tking into considertion the preopertive visul sttus, the ptient s ge, nd, bove ll, the extent nd strength of tumor ttchment. The impossibility of pplying ny type of rdiosurgery on the optic chism for the tretment of CP remnnts dds n extr level of complexity to this issue. Preopertive OCT ssessment of the structurl dmge to the visul pthwys my help to predict the potentil reversibility of visul impirment nd provides helpful insight to guide the intropertive surgicl decisions. CONCLUSIONS Intrchismtic CPs represent n exceptionl vrint of 3V CPs chrcterized by the invsion of the posterocentrl region of the optic chism. A chrcteristic potbelly deformtion of the optic chism visible on preopertive MRI points to CP infiltrtion of this structure. Tumor encrochment of the chism s posterior spect nd djcent optic trcts usully cuse visul deficit involving initilly the temporl lower qudrnts. The pterionl pproch combined with the opening of the lmin terminlis provides n optiml exposure of ny CP extension into the optic chism. Preopertive nlysis of retinl trophy with OCT llows relible ssessment of the ptient s visul outcome nd it my help to decide bout the degree of CP removl to be performed. Acknowledgments The uthors wish to express their grtitude to George Hmilton for his criticl review of the lnguge nd style of the mnuscript.

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