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This article was downloaded by:[CDL Journals Account] On: 12 March 2008 Access Details: [subscription number 785022369] Publisher: Psychology Press Informa Ltd Registered in England and Wales Registered Number: 1072954 Registered office: Mortimer House, 37-41 Mortimer Street, London W1T 3JH, UK

Neurocase Publication details, including instructions for authors and subscription information: http://www.informaworld.com/smpp/title~content=t713658146 Rapid Relief of Thalamic Syndrome Induced by Vestibular Caloric Stimulation Vilayanur S. Ramachandran a; Paul D. McGeoch a; Lisa Williams a; Gerard Arcilla a a Center for and Cognition, University of California San Diego, La Jolla, CA, USA

First Published on: 21 June 2007 To cite this Article: Ramachandran, Vilayanur S., McGeoch, Paul D., Williams, Lisa and Arcilla, Gerard (2007) 'Rapid Relief of Thalamic Pain Syndrome Induced by Vestibular Caloric Stimulation', Neurocase, 13:3, 185 - 188 To link to this article: DOI: 10.1080/13554790701450446 URL: http://dx.doi.org/10.1080/13554790701450446

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Downloaded By: [CDL Journals Account] At: 18:54 12 March 2008 -al [email protected] E-mail: execution of thisstudy. input. discrepant sensory Gi of the thalamic/posterior insula Thalamic painrepresent might response toadiscrepancy– painmaybe,fu that gested po fartheron these Speculating sphere alerts one to discrepancies – allowing reorientation. on behavi stability confer to sphere tends to “smooth over” discrepancies in sensory input in responsetodeafferentation. reorganisationfrom central ofth regional (complex RSD pain of pain,su ofchronic other types McCabe, Haigh, Halligan, &Blake, 2003), have shownthat Rogers-Ramachandran, 2000), and others(Flor et al., 1995; nent and incurable. slight reliefthe disorder but generally is considered perma- ing pain. and anti-epileptic medication can provide pressure, sometimesev astroke.The pa after face soon contralateraldevelops intheandthat limbs,body dysaesthesia and allodynia intense by characterized is 2001) (Dej thalamicpain Chronic Introduction RAMAC S. VILAYANUR Pain Thalamic Relieves Stimulation Vestibular Caloric Stimulation Rapid ReliefofThalamicPain NNCS DOI: 10.1080/13554790701450446 online /1465-3656 print 1355-4794 ISSN: http://www.psypress.com/neurocase Neurocase , to current treatments. We found that in two patients with this patients with two that in found We treatments. to current pain unrelenting isofsevere, presentation thalamus. Theclassic involv causativelesion Themostusual astroke. following ofpatients minority ina develops painsyndrome post-stroke Central for University Center of CaliforniaSanDiego, Brain andCognition, La Jolla, CA, USA stimulation, whereas placeboproce stimulation, Keywords: whichinturninhibits of painin cingulate. insula, the generation the anterior Address correspondence to Dr Paul McGeoch, Center for Brain a Center to Dr PaulMcGeoch, Address correspondence Founda theC.Robbins Our workis bytheR.Geckler and funded It wassuggested (Ramachandran, We (RamachandranHirste & (2007) (2007) Dejerine–Roussy syndrome, thalamic centralpain,post-st thalamic pain, syndrome, Dejerine–Roussy 13 , 185–188 , 185–188 in isrelentlessand theslightest touchor en apuffofair,ca HANDRAN, PAUL D. M HANDRAN, PAUL erine–Roussy) syndrome (Donaghy, syndrome erine–Roussy) © 2007 Psychology Press, animprint ofTaylor the &FrancisGroup, anInformabusiness ven thesubstantial vestibular our, whereas the right hemi- right the whereas our, departure from the statusquo. the from departure ndamentally, the organism’s r response to pain signaled by painsignaled to response r ssibilities Harris (1999) sug- ch as phantompain and the alamic andcortical pathways a pathological amplification pain type 1), result mainly result 1), type pain dures had no effect. We proposed that dures had this effect. Weproposed no is in, 1998; Ramachandran & 1995) that the left hemi- 1995) that theleft n trigger excruciat- trigger n C GEOCH, GEOCH, LISA andGERARDARCILLA WILLIAMS that involves the entire contralateral half of the body. It i It the body. halfof contralateral the entire involves that condition their pain was substantially improved by vestibular ca byvestibular improved was substantially their pain condition Syndrome InducedbyVestibular nd Cognition, UCSD, La La Jolla, nd Cognition,UCSD, CA 92093, USA. Tel: +1 858 822 0606 tions. However neither funding source had any role in the hadanyroleinthe design neitherfundingsource tions. However no pain,for the to 10, worst before and aftereach procedure. This scale goesfrom0,for rate theintensity of their painusing a visual analogue scale water andover,atleast, a30 procedures irrigation the patient supine withtheirh water. that weonly anticipated any po water ofdifferent te using be would we that told simply were they although them, nature ofthe caloric irrigati of procedures that may or may not reduce their pain. The patients. Theyweretold that logical examination a neuro- history, each patient’s based on wasconfirmed nosis approved bytheUCSDinstitut post stroke,in order tothis test conjecture. The study was We recruited twopatients w Methods tested this. vestibularrelieved caloric by stimulation.In thispaper we input to the insula we postulat In both patients allpatients theproc In both obtain consentwas Informed roke pain, vestibular stimulation, insula, anterior cingulate, anterior insula, vestibularstimulation, roke pain, because vestibular stimulation activates the po becausevestibularstimulationactivatesthe mperatures. They were blind tothe fact wereconducted using about 30 ml of nd imagingfindings. on procedure was explained to we were investigating a number ith Dejerine–Roussy syndrome ith Dejerine–Roussy pain imaginable. Theseratings -s period.Theywere asked to ed that thalamic pain might be eads at 30 degrees. Allofthe degrees. eads at30 tentialeffectice-cold fromthe ional review board. The diag- edures werecarried outwith ed, in writing, fromboth s largelyrefractory es the lateral es the sterior loric or . Downloaded By: [CDL Journals Account] At: 18:54 12 March 2008 next 30 min. In order to control for the unpleasant and unpleasant for the to control In order min. 30 next laterherated his pain ; halfanhour as 5. irrigation began. Hereported, with surprise, thathis pain was safterthe 30 observed was of onset water. The ice-cold with canal left the irrigated and min 10 waited hispainwasunch that reported left ear canal with water at body temperature. Thepatient caloric procedure,wefirst perfo sided pain as8.5 out of10. As a placebo control forthe were flexor. the on pin-prick and light touch sensitivityto increased testing but showed sensory normal power unremarkable. Histone, allodynia on the left side of neither immediate norlong-termrelief. anaesthetic. Thishadnot altered his painall; at producing local of injection epidural an hehadundergone in1996 that proate and amitriptyline, all to no avail. He also commented phenyt tried he hadpreviously that He reported . and oxazepam oxycodone, done, metha- regime of medication his current despite This was lenting, severe pain affecting the entire left side of his body. lacunar inthe right th year.remained Ithas constantsince. Imaging (MRI) revealed worsened and reached This allodynia. dysaesthesia and by ness wasslowly replaced about after few days,however, thesubsequent over resolved weakness side. The theleft of weakness and numbness onset sudden developed he 1992 In The first patient (FY)wasa right-handed 87-year-old male. Results stimulation, andtheireyes were checkedfornystagmus. , how the painful side of their body felt throughout the experienced they whether asked were patients procedures ac counterbalanced was cedures distraction. The order of administration of the different pro- ofdiscomfort, indi complained to their heads.These ice packs of theice-coldboth water they to controlfor the unpleasant and potentially distractingeffect but without theassociated neural tical administration procedure to canal with body temperature wate an ear of ashamirrigation also underwent they dure itself (4 ice-cold with irrigated FY and supine forCC. they found their pain wasmost were all carriedoutwhilst the 186 His pain then gradually rose in intensity againover the roseinintensity His painthen gradually visual-analogue char Using the than other and, alert mentally was he examination On When weinitially saw himin In theexperimental condition,thepatients’ earwere canals V. S. RAMACHANDRAN ETAL. V. S.RAMACHANDRAN its zenith over the courseofthenext ° C) water. Asa control to the proce- his face, hiscranial nerves were alamus andinternal capsule. patientin was theposition that had crushed ice packs applied cating theeffectiveness ofthis anged and still at8.5.Wethen and anged were applied until thepatients 2 months the left-sided numb- left-sided the months 2 oin, gabapentin, sodium val- sodium oin, gabapentin, uncomfortable: standing for standing uncomfortable: rmed a sham irrigationa rmed ofthe entire left side. Bothplantars and reflexeswere equal and 2006 he complained of unre- of hecomplained 2006 ross thesessions.Duringall the experimental condition effects. In order toattempt effects.Inorder r. Thisconsisted ofaniden- t heratedconstant his left also in the left ventromedial and lateral thalamus. – lobe and frontal posteroinferior insula and the including infarct intheterritory ofthe lapse. Imaging, including a recent head CT, showed an old pro- disc cervical left aknown to corresponding dermatomes theon leftside,savefor reduced pinprick sensation intheC6/7 extensor rightplantar. Neurological examination was normal an arm and leg and ofherright profound, ligent and oriented. She hada mild right , a response toquestions her “yes”or“no” answers recovery over the 14years. from perhaps normal, prisingly refractory tomedication. constant and severe intensity. Herpain had proved largely This worsened over severalmonths and then plateaued at a sided allodynia and dysaesthesia In the weeks following the stroke she developed severe right- an and with arighthemiplegia carotid left a after awoke she 1992 In female. ther stimulations. keen is Notably, he pain asa7. The least reduction hasbeen in hisleft leg, whereherates the he pain (0) in hisfaceand rate his painwasthat still substa “over adecade.” Four hadfallenovernight rating to a went home. he that irrigation this after was It 5. 6 to from fall to rating hour. next the over again rosebacktobaseline then and 4.5, ear right the of irrigation After extent. same the to not but leg, left his in pain of reduction some There was pain. no but “numbness” some was virtuallyleftof his arm free most face sideofhis left and The was uniform. not reduction that the 6. However,hecommented aoverall wasaround reported that he Surprisingly, another 30 min and at that time he rated his pain level as5. 8to5.5.He usfor with hadfallenfrom pain level stayed Immediatelythisafter stimulation thepatientreported thathis ear. right the of stimulation rating of8 out of 10. Wethen performed an ice-cold caloric after. hera and procedure control this dizzy. However,hi of feeling complained he until wasapplied It hadlo thatthisprocedure told pack wasapplied to hisleft ice an water, ice-cold ofthe effect distracting potentially On neurologicalexamination her mental status wassur- 69-year-old wasaright-handed (CC) patient second Our He contactedthenext us morning report to that pain his Irrigationwith oftheleftear ice-cold water caused his pain He againunderwentice-cold wa Eleven days later thepatientfacility.returned toour Indeed, 15 min after the ice pack his pain increased to a suggestedthat thatshe was lucid,intel- thefirs weeks after pinna forover a minute. was He ntially reduced. Henowhasno Despite herexpressiveaphasia, s pain level was unchanged by s painlevelwasunchanged Againnystagmuswasseen. his painwasstillreducedand d the pain inhis left armas 1. expressive (Broca’s) . expressive(Broca’s) 3 he 3 and felt the besthehadin his pain rating painrating his fell 6.5 to from left middlecerebral– wered paininotherpatients. and nonverbal signaling in to continue to attendfar- tocontinue to for of pain. Inthearmthere was of ted it as a 7 both before and 7 both a ted itas suggestive of thalamic pain. functional restitution and ofpaininhisleftearand terears. irrigation ofboth t visit he reported t visit Downloaded By: [CDL Journals Account] At: 18:54 12 March 2008 demonstrate that this no longer hurt. “touasking visitorsstarted to tionseveral by weeks in bothpatients. Claimingthat such a First, thepainreduction hasoutlasted thecold caloric irriga- have beena placebo effect? This apredator. dodging movements sudden thatwould stimulation bular “gate” otherwisebest to disablin fish intheDevonian seasoran & Dietrich, 1999; Ostrowskyal.,et 2002) Whetheryou area sented at least partially in close anatomic proximity. (Brandt altogether surprising that they netically primitive orienting resp options. Like painitself, the thalamic pain and may pave the way for novel therapeutic that vestibular caloric stimul Our findingson these twopatients stronglysupport the idea Discussion been “foryears.” She also vo had it lowest the was pain mother’s her that reported daughter was notas greatly relieved and rated thisarea as4.5. Her these areasas3.Again sherepo herfaceor in had allodynia patient reported that herpainwasstill reduced. She no longer rating. pain on her effect no It ence. had pain. Thepatient indicated that onforehead her for40 saftertelling her it would reduceher ing effect oftheice-cold water, weplaced a crushedice pack this stage as an additional place overall as4.5. Her general demeanor wasmuch improved. At was still reduced compared to pretreatmentand sherated it 3 (arm) and 5(leg). 0(face), to above ratings the from dropped Herpain formed. After 25minaright-sided ice-cold water caloric was per- her leg.Herpainratingsrema 8on and day’s irrigation) previous from residual (presumably left ear.Pain before irrigation was4on her face, 6on her arm bodytemper performed asham, again. was stilllessthannormal,thou Evenafter7hthepain pain. the from screamed have would touc when even low remained pain Her leg. right onher 5 and arm herright face, 3on her 2on to dropped Thepainrating appeared. nystagmus left ice-cold water caloric irrigation over a 30-s period until a Wethenperformed touch. light by amplified considerably This was true onrepeated analoguescale.Her rating was7before caloric irrigation. Might thesedramaticto responses thecaloric procedure 7 contacted when Remarkably, When wereturnedto The nextday,asa placebo We again askedpatient this rate to herpain onvisual- the see the patient 5dayslater thepain ation can profoundly modulate in pain the rated and right arm vestibular system is a phyloge- a is system vestibular ined unchanged post procedure. post ined unchanged ch or stroke” her right arm toor stroke”her rightarm ch inevitably occur as you make you as occur inevitably lunteered that should interact and be repre- control for the procedure, we this wasanunpleasant experi- arboreal primate, it might be might it primate, arboreal bo, to control for the distract- questioning. Herpainwas questioning. g chronic pain withthe vesti- gh it had started to increase hed, whereas normally she hed, whereasnormally ature water irrigation in her isunlikelyfor four reasons. rted that the pain in her legrted that thepainin onse and,perhaps, it isnot weeks after treatment, this her mother had VESTIBULAR STIMULATION RELIEVES THALAMIC PAIN RELIEVESTHALAMIC STIMULATION VESTIBULAR work. It has not escaped our notice that central pain due to due pain central that notice escaped our It hasnot work. and insula arealso damaged th particular, if the vestibular In . the of duration and location asexact things such likely thateffectiveness the ofthe procedure will depend on more patients; preferablyin adouble blind manner. Itis This finding,though Addendum Craig, & Tracey,Craig, & 2005),usin Godinez, Zambreanu, (Brooks, shown ithasbeen Indeed reflects underlyi patients, the in theface andarm compared to rior cingulate.Presumably, the acting to inhibit the be mediated by the vestibular painmay ofthalamic inhibition vestibular the If so, (2000). terior insula (cold) and anterior cingulate (pain), see Craig pain receptor signals passing through the thalamus to the pos- coldand of integration normal the of todisruption arises due cortex. Ithasbeen proposed thatcentral post-stroke pain gation activates, amongst other ar (Naito et al., 2003; Suzuki et demonstrated previously alsobeen Ithas signals. 2002) lar (Brandt &Dietrich, 1999) and pain (Ostrowsky et al., arm andleg. report adifferential reduction highly unlikely that both patients would independently placebo effect,itwouldbe in theirpainwereduetoa their in caused somereduction procedure wouldhave ceptible toplacebothenthisinvasive saw her.Onewouldimaginethat gone acervical epidural injection onlya week before we alleviate their thalamic pain. Indeed patient CC hadunder- These hadbeenanaesthetic. carriedoutinfutileattemptsto immediate or sustained –to epidural injections oflocal initialcationeffect. haveany nor procedure, tepidwater cebo procedure is untenable. Second, in neither patient did the pla- unpleasant toan due distraction from is reduction sustained permanent and more more and permanent effects andwhether re procedure. It would be interesting to seewhetherthere are ment, has been successfully treated by asimple, non-invasive co generally condition chronic is this first instance, inth thischronic form of . Indeed,we believe this the caloric procedure offers a newtherapeutic approach to to the foot area. the faceto arearostralturn, rostral hand area,which the is,in posterior insula is somatotopi It is known that the posterior theposterior that It isknown Third, neither patient had previously shown any response– Given thalamic pain is notoriously resistant to treatment, to resistant notoriously is pain thalamic Given sensation of painarising fromthe ante- complete remission. exciting,will needtobereplicated on connectionsbetween thethalamus e historyof , that a al., 2001)thatcold caloricirri- cally organised for pain, with for organised cally g functional imag ng topography the of insula. topography ng en theprocedure isunlikely to cortex in the posterior insula, posterior the in cortex pain. Fourth, ifthereduction peated irrigation can produce the distracting ice pack appli- differential reduction in pain nsidered refractoryto treat- the leg,experienced byboth insula receivesinsula bothvestibu- in painbetweentheir face, eas, the contralateral insular if these patients were sus- ing that the 187 Downloaded By: [CDL Journals Account] At: 18:54 12 March 2008 Brooks, J.,Zambreanu, L.,Godinez, Craig, A. D. (2000). The functional anatomy of lamina I and its role in central itsrole Iand of lamina anatomy functional The A. D.(2000). Craig, Brandt, T., & Dietrich, M.(1999).Brandt, T.,&Dietrich, The References stimulation. may we spinal chordinjury) fibromyalg causes (e.g., other 188 Donaghy, M. (2001). with highresolution Somatotopic organisation ofthe human 293–312. tions anddisorders. post-stroke pain syndrome. syndrome. pain post-stroke Oxford: Oxford Un V. S. RAMACHANDRAN ETAL. V. 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