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Show f. (//11. Nt'UrtJ-ilJ'"tllllIIl/IJ/. 5: 31-35, 1CJ85. Tuberculosis Granuloma in the Midbrain Causing Wall-Eyed Bilateral Internuclear Ophthalmoplegia (Webino)* FROILAN P. INOCENCIO, ~1.D. RX't ~tOND BALLECER, M.D. Abstract A 24-year-old female developed bilateral internuclear ophthalmoplegia with exotropia and loss of convergence, in association with a tuberculous granuloma situated in the midline midbrain at the level of the third ventricle. We labeled this case Webino lafter Martin Lubow), and to our knowledge, this is the first reported case occurring in association with an intraparenchymaltuberculous granuloma. Introduction Bilateral internuclear ophthalmoplegia associated with exotropia and loss of convergence most Iil..el\· indicates a midbrain lesion involving both med'ial rectus subnuclei. Dr. Martin Lubow has coined the term 'Webino" (wall-eyed bilateral internuclear ophthalmoplegia) to describe patients with bimedial rectus palsy and exotropia.' \\'e describe a case \-\'hich presents with this rare neuro-ophthalmologic finding, and an attempt to localize the lesion and the mechanism to explain it is brief]~, presented. Case Report A 24-year-old, right-handed female was admitted to the hospital in August 19R2 fLlf generalized body weakness and double vision. History reveals that 5 months prior to admission, she developed left-sided body weakness associated with numbness and paresthesias of the left lower Climcal 1\",,,I,,nl 1'",ll·""r (FI'I) .lnJ Ch,d '~","lc-nl I'h\'" Clan (RB), Dep.ulmenl "I (lphlh.,lm",,,gl·, "..", I,"n "' N."ur" ophlhalm"J"g\', I'htl'ppllll' Cl',wr.,( 11,,'p,I.,1. Un'\',''''I\' "I Ihe I'h'hppone' '-,1"lem . .\1.1ntl.1. I'h,hpponl". • P""I'n!l'J al Ihe I'h'hpponl' <'",WII' "f Ophlh.,ln",l"g\' '-,(, ~nt"lC .\11'l'long'. I'htl'pponl' Inll'rn.,I,,,n.,1 C"'Wl'nthlll Cl'nll'r, N"\'l'mt",r 2K, 1LlH2. r-L,ntla, ('htlip!,""". March 1985 e:-.trl'mity. There was no consultation done until after 11h~ months, when she noted progression of the left-sided weakness together with the appearance of mild drooping of the right upper eyelid, diplopia, and mild dysarthria The rightsided ptosis disappeared spontaneously after 2 weeks. Family history was noncontributory. Past medical history revealed a 3-month treatment of INH for mild pulmonary tuberculosis by a general practitioner. On examination, the patient's vital signs were normal. She was coherent, conscious, oriented, and ambulatory with slight support. There was note of slight dysarthria. Neuro-ophthalmologic examination showed exotropia in primary position (Fig. 10), bimedial rectus palsy on attempted horizontal gaze on either direction with noted variable abducting horizontal jerk nystagmus (Figs. 1/1 and 1e). Upward gaze was normal (Fig. 1tt). There was loss of convergence (Fig, 1e), although pupils were 3 mm in size, equal, reacted to light, and with minimal visible pupillary constriction noted on attempted accommodation for near gaze. Visual acuity was 20/20, J2 with correction, for both eyes. Forced ductilm test was essentially negativ~ Fundi were normal. Visual iield examination showed bibteral mild constrictilm of the peripheral iields. Cllior testing and light intensity examin'ltion I·vere unremarbble. Except for slight shallowing IJi the right nasolabi, ll iold and mild part'sis lli the leit lower l'xlremitv, the rl'mainder oi the neumlogical examinatil; n was unremarkable. Intravenous edrophllnium chloride (Tl'nsilon 10 mg) did nl)t alter thl' p,ltil'nt's ophth'llnlllplegia. Thyroid and diabdic wllrl..ups were normal. Chest x-ray shl)wed apic,ll densities in both lungs (llmpatible with minim'll tll l1111der,ltl.'ly active pulmonary tubercuillsis. Lumbar puncture showl'd clear CSF with nllrmal pressure, mildly elev,lted protein (330 mg/dl), ,md mild cellular Cllunt (60) with lymphocytic predominance and decreased glucose. 31 III} Figures la-leo (a) Ey", in prim,lry posill,)n showin~ ,'wtropl,l (/!l Ll'lt m,'diJI rectus paralysis on attempted right gaze. (c) Right m,'d'JI n't'lu, pJI,y on Jt"'mp"'d Il'lt ~,l/". (d) Norm,ll upwMd ~.lI". (c) Loss of convergence on attempted near gaze. 32 Journal of Clinical Neuro-ophthalmo)ogy \io malignant cells were present. A search for malignant focus elsewhere was negative. Plain skull x-ray (AP lateral) and bilateral carotid angiography were normal. Computerized tomography, before and after injection of radioactive contrast. showed a moderate area of increased density at the midline midbrain compressing the third ventncle with a visible enhanced midline density extending to the level of the rostro-dorsal pontine area obliterating the Aqueduct of Sylvius (Fig. 2). An impression of wall-eyed bilateral internuclear ophthalmoplegia secondary to a midbrain spaceoccupying lesion, probably glioma, was given in our clinic. Visualization of the mass at surgery revealed a well-defined, firm, indurated mass measuring 15 X 20 mm compressing the third ventricle in the midline midbrain and partially obliterating the Aqueduct of Sylvius. Histopathologic section of the mass revealed chronic granulomatous inflammation with evidence of caseation necrosis, compatible with tuberculous granuloma. The patient died after 3 days from postoperative complications. Discussion Bilateral internuclear ophthalmoplegia associated with exotropia and loss of convergence most likely indicates a midbrain lesion involving both medial rectus subnuclei and the medial longitu- March 1985 Inocencio, Ballecer Figure 2. 1'1I,tinfu,illn 'C,1I1 dl'nw'blr,lll" rn~" Jt rn id Ii Ill' .lrl"l. ,itting lin thl' third vl'ntrick, ro,trJI tll thl' ml''C'ncl'ph, llt..ln. dinal fasciculus. Dr. Martin Lubow has coined the term "Webino" (wall-eyed bilateral internuclear ophthal~oplegia) to describe patients with these findings. Internuclear ophthalmoplegia, as we know ':low, dates back to the first description by ~lison- In 1906 and was labeled ophthalmoplegIa Internuclearis in 1922 by LHermitte 3 It is characterized by a failure of the medial rectus muscles on the side of the lesion to act in the horizontal gaze to the opposite side and nystagmus of the abducting eye in lateral gaze to the side opposite the lesion. The lesion involves the medial longitudinal fasciculus (MLF) connecting the pontine paramedIan reticular formation (PPRF) and the ipsilateral abducens nucleus with the contralateral medial rectus subnucleus in the midbrain. 4 " The most common causes of internuclear ophthalmoplegia are vascular diseases and demyelinative diseases," 7 in general, although It can also ~e caused by intraparenchymal neoplastic leSIOns. When signs are bilateral, the etiology is most commonly multiple sclerosis." Cogan reported various cases with bilateral internuclear ophthalmoplegia occurring in association with posteril1r fossa tumors, brain stem infarction compressiVt' lesions, and Arnold-Chiari malfor~ mations with hydrocephalus." Since then, bilateral internuclear ophthalmoplegia has been described in a variety of conditions as head III • trauma, basilar artery aneurysm, II branched-chain ketoaminociduria, Ie and s'ome forms of spinocerebellar degenerations. 11 Ocular findings SImIlar to bIlater,ll. JI1!l'rnuclear ophthalnll)plegia have .bel'n descTlbl'~i4 111 myasthenia gravis," abetahpllprotell1l'mla, and In patIents in coma due ~~,) I~~verdose of sed,ltiVt' ~ypnotics or phenytom. Latdy, cases of bIlateral internuclear ophthalnll~rlegia associated with carcinof!1atous menmglhs and cryptococcal meningitis\' were reported in the literature. 33 Tubl'rllll'lsis VIIl'bin,' / / I Fasciculus N.m F,~~·~SUP'COlllculua ". ~"'''''. . Aqueduct of Sylvlua , . ~ ~d . '. \- -,If--_IV..l,,-Nu.m / ~"P, ...~E.'''''. ,. ~MLF //'- ~~ ~~.\,.;, -- ---ML r-' ~ ,.1V',~ .. W • f" 41 •~ -.-- Red nu. , '" 1,\. "";" . . , '. - 1'1 ...... .__._- --Substantoe ··I'.Vlft Nigra . ~ /1 cerebrel II peduncle Figure 3. M,dbr,lIl1 ,,'dl\111 ,11 Ihl' 1,'v,·1 "I Ih,' ,up,'''''' (uIIJ(ulu' ~1LF II1dical"~ I1wdi,11 lun).;lludil1,ll I,"(\lulu,; ML, I1wdl,11 il'mnl'(u' Aml\\" ,h",," iL-SlUI1 rl"pOI1SlbiL- Ill' ,,",lll-"\,,'d b1l,'I\'",1 1I11"rJlUlil'ar "phth,1Im"pl,·).;o.1 There have been conflicting studies done to e'\plain the pathogenesis of internuclear ophthalmoplegia. One such clinicopathologic study done bv Yoshikura et aI., reveals a limited lesion of the t;Vl) longitudinal fasciculi associated with right internal nucleus degeneration in the midbrain,' 4 An attempt to explain the neuro-ophthalmologic findings described in our case in relation to the pathologic site is not very difficult. The extensive strategic position of the mass at the level of the mesencephalic area up to the level of the rostro-dorsal area of the pons may have given rise to these findings, either by local structural compression or ischemic effects attributable to the tuberculous mass. We surmise that the lesion most probably touched the medial rectus subnuclei or affected fibers passing from or to the oculomotor subnuclei and the PPRF (Fig. 3), To explain further, one may refer to the Warwick's schema of the oculomotor nuclear complex of the third cranial nerve and its anatomic position The nuclear complex lies beneath the aqueductal grav matter of the rostral midbrain at the level of the superior colliculus,'" The MLF passes just lateral and inferior to the oculomotor comple'\. This relative area of proximity of the medial rectus subnuclei being located in the inferior aspect l)f the oculomotor nuclear comple:\., III the MLF fibers, may have been the site aiiected bv the lesion. Therefore, it seems appwpriate to C'lllclude that the tuberculous mass dfl'ct at the Mea of the midbrain was responsible f,lr the findings described in our C,1S1', further supported bv the demonstration of the lesion and its n,1ture bv computerized tomographic scan and the oper,ltive histopathologic results. References I, D,1<off. R.B. ,lnd Ifoyt. VII,F,: Supranucll'M disorders Ilf o(uldr control s\"tl'nb in lll,ln. In Ti,e Cli/frol of Elll' MII1'l'I1Il'nt" Y. Bach. P. Rita. C. C"llin.,. and J Hyd~. Eds Academic Press Inc. Nt'\" Yurko 1971. p 223. ~ Wilsun. SAK: Case nu. 3. presented at the clinical ml't'ling lIf the 't'urological Society of the United Kingdum, un March 29, 1906 Brail/ 27: 298,1906. 3 Duk~,Eldt'L \\'.5.: Tcxtb",l/; (If 0l'hthlll/ll,'/ogy, Vol. 4. Henry Kimptun, Lundun, 19-t9. p -t165. -t. Huber. A. and Bludi. F (Eds) Evc 5i,\II' and 5ymp(, 1m, 111 Bra/II T/I1I/,Jr- (3rd ~d.). C. \' \Iosby Co. Saint Llluis. 1476, p. 52 5. C!as~r. J.: ,\'curIHll'hthl1/Ill"lt"\l/, Harper & Row. Hagerstll\\'ll, \ lan'land. 1478. p. 215. 6. Smith. I L, and Cugan, DC: Internudear ophthalmuplegia: A re\'iew of 58 cases. Arch. Ophtha/Illll /. 61: 687. 1959. I. Fi.izsch. R: Die Internukleare Ophthalmoplegie: CunsideratillllS sur 75 cas d'Ophtalm'1plegie internucleaire, ()phth,1/III"/I,,\/,'11162: 331-3-t2, 1971 8 C"g,m, DC, and \\'ray. 5 Internuclear ophtha!muplegia as ,111 e.ul\' sign "f brainstem tumors. '\','Url,/,l\l/ 20: t>29, 1470 q Cllg,m, 'DC. Intl'rnuc!e.u llphthalmllplegia, typical ,md atyplc,ll .4rd1. ()I'hth'lllll"/. 84: 583-589. 197L1 10. Beck R.\\' ,md \Ieckler, R.I: Internuclear 'lphth,)lll1llp!ep,l ,1fter he,ld trauma. AIIll. Ol'htllalIIIII/. 13: t>7'I-t>7'5, 1481 11 De\l're,HI', \1.\\'., Brust.IC\1.. and Keane, J.R.: Internucle.u llphth,llnwplegia caused by subdural 11<'111,1111111,1 .\·Cllr,,/,I\l( 27: 251-256,1979. 12. \1,)(' Dlln,lld. LL. ,m'j SheL P.K.: Ophthalmoplegia ,IS ,1 Sign uf metabolic disease in the newborn. .\"·/I,.,'I",\II 27: 471-473, 1977. \.I. \\'l'inl'r. Lr, K'lI1igsmark BW.. Stoll. }.. et al.: I krl'dit.HY llli\'ll-pllIlt'lCerebellar atrophy with retin, ll degl'l1<'ratilln: Rep'lrt llf a family through six genl'r,)ti"lb. A,.d1. ,\!cllr,l/. 16: 36-t-376, 1967. I~. '11'1'. R.D. Cl'g,m. D.C. and Zee, DS: Ophthalnwpkgi, l ,md diss,lci,lted nystagmus in abetalipopr" lt'int'll1i,). ,4rd1. t)l'hthll/II/(I/. 94: 571-575, 1976. 15. 5l1lwn. R.I'.: Fllrced downward ocular deviation ,l(currl'nce during oculo-vestibular testing in sed,1ti\' t' drug-induced coma. Arch. Nfl/ro/. 35: 456- t58, 1978 It>, Spector. R.H., Davidoff, R.A., and Schwartzman, 34 Journal of Clinical Neuro-ophthalmology R.J.: Pht'm'lllin-indu'l'd llphth,llnwpll'gi,l. N,'/I,.o/" S-1/26: \l131-10,H, Ill7o. 17. ClI IWt',l, E.F., ,1I1d I kiIIII ,111 , K.M.: Nl'UH'"phthalmi, ,1Spl'ds lli ll'ntr,ll l1l'rVl'US svsll'lll l"rvpllllllllllSis: Intl'rnlIlk'.Jr llphth,lllllllpkgi,l ,lnd supr, lnUlll'.H llphth,llnwpkgl,l ..\,..-1,. ()"I,III,lIll1ol. 87: lo~-Iol\. Ill7::' 11\. Fllrd. S.C .. l'l ,11: Hi 1.1 ll'r,l I inll'rnulIl',H llphth'llIllllplt'gi, l in l,HliI1l11ll,ltllus nll'ningitlS. /. elill. .\','11/"(1-"1'"11",11111'/. 3: 1::'7-130. 1'1 1\3. III 't lIShil-.ur,l. N. Iililll'l 1\1. "l,b,l\',lShi, 1\1, T,ll-.l'U-March 1985 Inocl'ncio, Ballecl'r lI1i, 'I, ,lnd 11,11,1110, M.: Ophlhalmllpl0gil' internulIe', lir" bil,lll'r,lk 1~I'l'. Ne/ll'l'/. 135: 21. 1'17'1. ZO. W,lrlvid" Ie R"pH'sl'nlatilln IIf thl' l'xtraucular musdl's in till' oluillmlllllr 'ompll'x. f. COIIIV NC/I/ 11/.98: Hl), Il /S3 W,.ill' for r"l'rillis 10: hllilan I' Inllll'ncio, M.D., Sl'rvIn' d'Ophl'llmlllllgil' IV, Cl'ntrl' National d'OphtalmoIllgil' dl's Quin/l'- Vingls, 2H Rul' de Charenton, 75571, 1'.His, C"dl'~ 12, fr,lnn'. 35 |