©฀JAPI฀ •฀ VOL.฀56฀ •฀ FEBRUARY฀2008฀ www.japi.org฀ 121 Case Report Locked-in Syndrome in Snakebite S Prakash*, C Mathew**, S Bhagat** Abstract Two฀ young฀ patients฀ are฀ described฀ who฀ made฀ complete฀ recovery฀ from฀ locked-in฀ syndrome฀ (LIS)฀ after฀ snakebites.฀LIS฀was฀a฀presenting฀feature฀in฀a฀patient฀of฀presumed฀snake฀bite฀who฀showed฀complete฀response฀ to฀polyvalent฀Anti-snake฀venom฀(ASV).฀This฀case฀suggests฀that฀elapid฀snake฀bite฀should฀be฀suspected฀in฀ unresponsive฀patient฀found฀in฀early฀morning฀in฀endemic฀areas฀of฀snake฀bite฀in฀monsoon฀season.฀The฀second฀ case฀was฀admitted฀in฀complete฀LIS฀state฀with฀history฀of฀rapidly฀progressive฀ptosis,฀diplopia,฀ophthalmoplegia,฀ bulbar฀symptoms฀and฀quadriparesis,฀6฀hours฀after฀snake฀bite.฀Complete฀improvement฀by฀ASV฀in฀second฀ patient฀highlights฀the฀need฀of฀differentiation฀of฀complete฀LIS฀from฀coma฀and฀brain฀death฀in฀patient฀of฀snake฀ bite,฀as฀former฀have฀good฀prognosis.฀© INTRODUCTION L ocked-in฀ syndrome฀ (LIS)฀ is฀ characterized฀ by฀ qadriparesis฀ and฀ anarthria฀ in฀ conscious฀ patient.฀ Patient฀ can฀ be฀ communicated฀ by฀ eye฀ movement฀ and฀ blinking.฀Communication฀is฀not฀possible฀in฀total฀LIS. 1 LIS฀ may฀ be฀ either฀ of฀ central,฀ usually฀ ventral฀ pons฀ 1 ,฀ or฀ of฀ peripheral฀ origin. 2 ฀ Diagnosis฀ of฀ central฀ cause฀ is฀ important฀ for฀ rehabilitation฀ purpose,฀ as฀ even฀ limited฀ physical฀activity฀can฀improve฀quality฀of฀life. 1 ฀Knowing฀ the฀ peripheral฀ causes฀ is฀ very฀ important,฀ as฀ one฀ may฀ make฀ erroneous฀ diagnosis฀ of฀ brain฀ death฀ in฀ a฀ patient฀ with฀complete฀LIS฀due฀to฀fulminant฀polyneuropathy฀and฀ severe฀neuromuscular฀junction฀Blockade. ฀2,3 ฀฀In฀two฀case฀ reports,฀we฀discuss฀the฀importance฀of฀LIS฀in฀patients฀of฀ snakebite฀and฀review฀the฀literature. CASE REPORTS Case-1 A฀40฀years฀male฀was฀brought฀to฀the฀emergency฀room฀ in฀unresponsive฀state฀with฀no฀signifcant฀past฀history.฀ In฀ early฀ morning฀ patient฀ did฀ not฀ show฀ any฀ response฀ when฀his฀wife฀tried฀to฀make฀him฀awake.฀At฀presentation,฀ patient฀had฀no฀motor฀response฀to฀painful฀stimuli.฀There฀ was฀ptosis,฀no฀spontaneous฀eye฀movement฀and฀absent฀ oculocephalic฀and฀oculovestibular฀refexes.฀Deep฀tendon฀ refexes฀ and฀ planter฀ refexes฀ were฀ absent.฀ Pupil฀ was฀ of฀3฀mm฀size฀with฀sluggish฀reaction฀to฀light.฀Physical฀ examination฀revealed฀no฀abnormality.฀His฀respiratory฀ effort฀ was฀ sluggish.฀ Endotracheal฀ intubation฀ was฀ performed฀and฀patient฀was฀mechanically฀ventilated.฀A฀ provisional฀diagnosis฀of฀stroke฀was฀made,฀but฀MRI฀Brain฀ *Assistant฀Professor;฀**Juior฀Resident,฀Department฀of฀Neurology,฀ Medical฀College,฀Baroda,฀Gujarat. Received฀:฀10.10.2007;฀Accepted฀:฀29.10.2007 was฀ normal.฀ Hematological฀ examination฀ and฀ blood\ serum฀ biochemistry฀ (sugar,฀ creatine,฀ liver฀ function฀ tests,฀ electrolytes,฀ and฀ thyroid฀ profles)฀ were฀ within฀ normal฀ limits.฀ Chest฀ X-ray฀ and฀ cerebro฀ spinal฀ fuid฀ (CSF)฀ examinations฀ did฀ not฀ reveal฀ any฀ abnormality.฀ The฀ electroencephalograph฀ showed฀ alpha฀ and฀ theta฀ activity฀with฀no฀epileptiform฀discharges.฀Further฀careful฀ examinations฀of฀skin฀revealed฀reddish฀spot฀over฀back฀ of฀chest.฀It฀was฀not฀typical฀fang฀mark฀of฀snakebite,฀but฀ patient฀was฀given฀injection฀polyvalent฀ASV.฀We฀noticed฀ spontaneous฀eye฀movement฀within฀an฀hour฀of฀treatment.฀ He฀ communicated฀ with฀ eye฀ movement.฀ There฀ was฀ complete฀improvement฀of฀ptosis฀and฀ophthalmoplegia฀ in฀about฀30฀hours.฀Patient฀was฀extubated฀after฀3฀days.฀ Proximal฀ muscle฀ weakness฀ took฀ 7฀ days฀ to฀ improve฀ completely.฀The฀patient฀could฀remember฀well฀the฀period฀ when฀he฀was฀unresponsive.฀He฀did฀not฀give฀history฀of฀ any฀possible฀snakebite฀or฀toxin฀and฀drug฀exposure. Case 2 A฀ 25฀ years฀ old฀ male฀ was฀ bitten฀ on฀ toe฀ by฀ an฀ unidentifed฀ snake฀ 8฀ hour฀ prior฀ to฀ his฀ admission.฀ He฀ was฀brought฀to฀emergency฀room฀in฀unconscious฀state฀ with฀ history฀ of฀ rapidly฀ progressive฀ ptosis,฀ diplopia,฀ dysphagia,฀dysarthria,฀dyspnoea,฀and฀weakness฀of฀all฀ four฀limbs.฀Patient฀was฀unresponsive฀to฀painful฀stimuli.฀ Light฀refex,฀oculocephalic฀refex,฀oculovestibular฀refex,฀ deep฀ tendon฀ reflexes฀ and฀ superficial฀ reflexes฀ were฀ absent.฀ Swelling฀ at฀ bite฀ site฀ was฀ noted.฀ Patient฀ was฀ mechanically฀ ventilated฀ because฀ of฀ poor฀ respiratory฀ efforts.฀Hematological฀examinations฀and฀blood/serum฀ biochemistry฀were฀within฀normal฀limits.฀A฀provisional฀ diagnosis฀of฀snakebite฀with฀hypoxic฀encephalopathy฀was฀ made.฀Injection฀polyvalent฀ASV฀was฀started.฀There฀was฀ spontaneous฀eye฀movement฀after฀6฀hour฀of฀treatment฀ and฀patient฀communicated฀with฀eye฀movement.฀Patient฀ improved฀completely฀in฀5฀days.฀Patient฀was฀able฀to฀recall฀