understanding anosognosia for hemiplegia after stroke

CLINICAL FEATURE

Understanding Anosognosia for Hemiplegia After StrokeElizabeth M. Byrd1, MSN, RN, CCNS, Rita J. Jablonski1, PhD & David E. Vance1, PhD

AbstractBackground: Anosognosia for hemiplegia (AHP) after stroke is a complex cognitive behavioral disorder that removes awareness ofone-sided paralysis (hemiplegia). As a result, stroke survivors afflicted with AHP may bemore likely to have unrealistic expectationsfor stroke rehabilitation, display unsafe behaviors and experience falls, and ultimately suffer the physical and psychological conse-quences of frequent falling.Objective: The purpose of this article is to describe AHP by discussing anosognosia within the context of contemporary theoreti-cal understandings, examining current imaging evidence of the disorder, and summarizing emerging interventions designed toreinstate self-awareness in anosognosic patients.Method: Systematic review with a focus on defining and describing AHP based on human experimental studies was conductedwithin a 10-year period.Results: Eleven studies were identified. The content and foci of the 11 studies fell into one of three categories: theory testing,imaging evidence, and interventions for individuals with AHP.

Keywords: Anosognosia; falls; rehabilitation; stroke.

Anosognosia for hemiplegia (AHP) is a phenomenon thatwas first described in 1914 in an article published byDr. Joseph Babinski as cited in Langer (2009). He de-scribed his observations while attending to two patientsafter they experienced a stroke. In each of the instances,the patients had a lingering left hemiplegia. The intellec-tual capacity of the individuals seemed to be preserved,and there was no evidence of confusion, confabulation,or hallucinations. Yet, each of the patients exhibited anunawareness and/or denial of their left hemiplegia. Whenasked tomove the affected limb, the first patient remainedsilent and reacted as if the question had been addressedto another individual. The second patient, when asked tomove the same affected limb, though she was unable todo so, remarked that she had completed the task as di-rected. Babinski (1914) called this seeming unawareness“anosognosia” and speculated that the phenomenon wasdue to the combined effects of location of the infractedbrain lesion and sensory losses experienced poststroke

Correspondence: Elizabeth M. Byrd, MSN, RN, CCNS, School of Nursing,University of Alabama at Birmingham, 1701 University Boulevard, Birmingham, AL35294. E-mail: [email protected]

1 University of Alabama Birmingham School of Nursing, Birmingham, AL, USA

Copyright © 2018 Association of Rehabilitation Nurses.

Cite this article as:Byrd, E. M., Jablonski, R. J., & Vance, D. E. (2020). Understanding

anosognosia for hemiplegia after stroke. Rehabilitation Nursing,45(1), 3–15. doi: 10.1097/rnj.0000000000000185

January/February 2020 • Volume 45 • Number 1

Copyright © 2019 by the Association of Rehabilitation Nurse

(as cited in Langer, 2009). The modern definition ofanosognosia is a general lack of awareness or the under-estimation of a specific deficit in function due to a brainlesion (Kortte & Hillis, 2009). Although this phenome-non has been associated with neglect and can occur inthe presence of neglect, anosognosia and neglect aredistinct syndromes and should not be confused. Withanosognosia, it is theorized that the affected individualis receiving false feedback from a limb that is recognizedas belonging to self (Kortte &Hillis, 2009). Neglect, how-ever, is an impairment of the individual to respond tostimuli occurring in the hemispace opposite of the lesion(Kortte & Hillis, 2009).

Today, it is estimated that AHP affects 20%–30% ofindividuals that suffer a stroke. Patients that experiencethe typical manifestations of anosognosia tend to haveunrealistic expectations of outcomes while in rehabilita-tion (Orfei, Caltagirone, & Spalletta, 2009) or refuse toparticipate in rehabilitation activities altogether (Cherney,2006). More importantly, they are disposed to disregardappropriate safety measures, such as ambulating withoutassistance, which can lead to falls and serious injury ifnot addressed early in the rehabilitation stay (Hartman-Maeir, Soroker, & Katz, 2001). But the relationshipbetween AHP after stroke and falling behaviors is notentirely understood. In fact, most of the anosognosia lit-erature stems from neuropsychology journals, whereasnursing and allied health journals have remained silenton the issue. Understanding AHP after stroke and its

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4 Anosognosia

relationship to patient safety events in the stroke rehabil-itation setting could potentially lead to a more reliableway to predict stroke patients who will have fall events.Furthermore, a more comprehensive understanding ofAHP after stroke would inform the development of inter-ventions to reverse the manifestations and consequencesof this syndrome. The purpose of this systematic reviewarticle is to describe AHP after stroke by synthesizingfindings from human research studies conducted from2007 through 2017.

Search Strategy

Two databases were used: PsycInfo and PubMed. Thesearch was conducted on June 6, 2017. The followingsearch strategy was used in the PsycInfo databases: thekeywords “anosognosia,” “hemiplegia,” and “stroke”were searched using the “abstract fields.” Publicationswere limited to 2007 through 2017 and to humans aged18 or older, resulting in 18 articles. For the PubMed data-base search, the words “hemiplegia,” “anosognosia,”and “stroke” were searched in both the title and abstractfields. The same limitations (human research only, age 18or older, research published from 2007 to 2017) werealso applied. Twenty-two articles were identified. Theresults from both searches were combined for a total of40 articles. After excluding duplicates and review articles,a total of 22 articles remained. Application of furtherexclusion criteria (research involving apraxia, acquiredbrain injury, and cognitive anosognosia) resulted in theexclusion of 11 more articles, leaving 11 total articles,which are included in this review.

Results

No qualitative studies were found in the searches. Thecontent and foci of the 11 studies fell into one of three cat-egories: theory testing, imaging evidence, and interven-tions for individuals with AHP. These articles are reviewedbelow to produce the most current description of AHP(see Figure 1 andTable 1). Using these categories, the resultssection is organized so that we first provide an overviewof theoretical understandings of anosognosia. Second, webriefly examine current imaging evidence of the disorder.Finally, we summarize the emerging interventions that seekto reinstate self-awareness in anosognosic patients.

Theory Testing

According to Bottini et al. (2009), the motivational andpsychodynamic interpretations of AHP after stroke havelost favor with the scientific community. Instead, thecontemporary theories concerning the mechanism for the


presence of AHP are grounded in neurobiological pro-cesses: malfunctions between the motor control systemand the accompanying sensory feedback loop (Bottiniet al., 2009). There is existing evidence that central ner-vous system function parallels previously created internalmodels that mimic and predict dimensions of one’s ownbody in relation to the external world (Frith, Blakemore,&Wolpert, 2000).Movement is based on action intentionandmotor planning. The success of these actions is attrib-uted to systems within the central nervous system thathave been termed predictors and controllers. In healthy(nonstroke) persons, muscles are contracted via stimula-tion from the central nervous system through a neuronalprocess known as the predictor, which estimates the endconsequence of the movement (see Figure 2). Anotherneuronal process, the controller, captures the relationshipbetween the desired action and the action that wasachieved. Feedback is then used to predict more efficientand effective future movements (Frith et al., 2000). Inother words, intentional movements involve the coordi-nation of motor and sensory nerves. The appropriatecontraction of specific muscle groups occurs with concur-rent processing of sensory information to successfullycomplete an intended movement. These interacting motorand sensory functions do not only ensure the successfulcompletion of an internal movement but also enable indi-viduals to learn from previous intentional movementexperiences to improve efficiency and effectiveness offuture movements (Frith et al., 2000). This feedback loopcontinuously updates so that future movements are opti-mally efficient. If a movement is not conducted as intended,the comparatorwill detect themismatch between the actionplan and movement, and that information will be used toinform future motor awareness (Fotopoulou et al., 2008).

The theories discovered in this systematic review allcontribute the symptoms of anosognosia to a failure ofthe feedback loop to update the motor cortex of the suc-cess of the intended movement. Where the theories differis in the mechanism that causes the feedback loop toupdate (Fotopoulou et al., 2008; Jenkinson, Edelstyn, &Ellis, 2009; Preston & Newport, 2014; Saj, Vocat, &Vuilleumier, 2014; Vocat, Saj, & Vuilleumier, 2013).

According to Frith et al. (2000), in AHP, predictorand controller roles malfunction because relevant areasof the brain have been damaged via infarction. Thoughthere is evidence to suggest that individuals with right-sided stroke are able to activate the motor cortex (Langer,2009) in order to move the left side of the body, the indi-vidual is not able to correctly identify the position ofmovement of the paralyzed limb because the predictorhas estimated, based on previous experience from thiscommand and movement memory, that movement has


Table

1Matrix

tableforarticlesused

insynthesis

Autho

r/Date/ArticleTitle

Sample

ProcedureandMeasures

DataAnalysis

Streng

thsandLimitatio

ns

Theory

testing

Vocatet

al.(2013)

Therid

dleof

anosog

nosia:D

oes

unaw

arenessof

hemiplegia

involveafailureto

updatebeliefs?

11controlparticipants;

9consecutivepatientswith

afirsthemisp

heric

stroke

and

fulllefthemiplegia

Globalexecutiveandmem

oryfunctio

nassessed;globalcog

nitivefunctio

nwas

measuredviaMMSE.

Ridd

leTest—Participantsweregivenclues

andwereaskedto

comeup

with

the

one-wordanswerandratetheirconfidence

thatthey

hadchosen

thecorrectword.

Ano

sogn

osicpatientsweresig

nificantly

moreoverconfidentintheirrespo

nses

comparedwith

nosogn

osic(p

=.016)

andcontrols(p

=.006).

Ano

sogn

osicpatientshadsig

nificantly

fewer

correctrespon

sescomparedto

nosogn

osic(p

=.78).

Streng

ths

•Novelstud

yLimitatio

ns•Smallsam

plesize

with

low

statisticalpo

wer

Sajetal.(2014)

Action-mon

itoringimpairm

entin

anosog

nosia

forh

emiplegia

5controlparticipants;10patients

with

arig

ht-hem

ispheric

stroke

andfulllefthemiplegia,

5of

which

wereanosog

nosic

Assessm

entof

AHPusingstructured

interviewto

assessverbalaw

arenessof

abilityto

perfo

rmactio

nsandacompo

site

anosog

nosia

questio

nnaire.

Globalcog

nitivefunctio

nutilizing

MMSE.

Phase1—

Participantsaskedto

perfo

rmor

imagineactio

nswith

theirright

orleftarm.

Phase2—

Participantsareaskedwhetherthe

prioractions

wererealized,imagined,

ornew.

Sign

ificant

differenceam

ongtheAHP,HP,

andcontrolgroup

forleftrealized

movem

ents(p

<.001),leftattempted

movem

ents(p

=.001),leftimagined

movem

ents(p

=.002),andalso

right

realized

(p=.004)and

right

imagined

movem

ents(p

=.007).

Streng

ths

•Novelstud

yLimitatio

ns•Smallsam

plesize

with

low

statisticalpo

wer

Fotopo

ulou

etal.(2008)

Theroleofmotorintentioninmotor

awareness:Anexperim

entalstudy

onanosog

nosia

forh

emiplegia

4hemiplegicpatientswith

anosog

nosia

and4hemiplegic

patientswith

outanosog

nosia

Globalcog

nitivetestinginclud

ingtheWTA

R,BehavioralInattentionTest,visu

alfield

testing,AHPqu

estio

nnaire,and

Cogn

itive

Estim

ates

Test.

Alife-sized

rubb

ermod

elof

alefthand

was

used

togive

false

visualfeedback

ofmovem

ent.

Part1—

Participantswereaskedto

raise

and

lower

theirarm

.Part2—

Participantsweretoldto

stay

still

andthattheinvestigator

wou

ldmove

theirarm

.Part3—

TheParticipantsweretoldthatno

movem

entw

ouldbe

made.


answ

erineach

trialm

ovem

entdetectionandconfidence

inmovem

ent.

TheAHPgrou

phadadecrease

incorrect

respon

seson

lyinthecond

ition

where

they

hadintend

edto

move.

Even

theno

n-AHPparticipantsbelieved

they

generatedmovem

entswhen

presentedwith

false

visualfeedback

ofthemovingprosthetichand

.

Streng

ths

•Theorytesting

•Novelstud

y•N

odifferences

betweengrou

psinglob

alcogn

itive

functio

nor

reason

ingabilities

Limitatio

ns•Smallsam

plesize,sugg

estib

ility

oftheprostheticarm

shou

ldbe

takeninto

accoun

twhen

consideringtheresults

(continues)

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Table

1Matrix


insynthesis,Con

tinued

Autho

r/Date/ArticleTitle

Sample


DataAnalysis

Streng

thsandLimitatio

ns

Jenkinsonet

al.(2009)

Imaginingtheimpo

ssible:M

otor

representatio

nsinanosog

nosia

forh

emiplegia

18patientswith

dense

hemiplegia;22

age-matched

healthyvolunteers;10control

patients

MMSE

andNationalA

dultReadingTest.

Assessm

entof

AHPusingstructured

interviewto

measure

verbalaw

arenessof

abilityto

perfo

rmactio

ns.

Grip

selectiontask

form

otor

imageryand

motor

control.

Cong

ruency

betweenmotor

imageryand

motor

controlw

assig

nificantly

lower

intheanosog

nosia

patientsversus

the

healthyvolunteers(leftmotor

imagery,

p=.006;right

motor

imagery,p=.001).

Streng

ths

•Group

swerematched

forage,

degree

ofhemiparesis,and

leng

thof

timebetweenstroke

andparticipationinthestud

yLimitatio

ns•Premorbidintellectualfun

ction

was

lower

inthepatientswho

experienced

astroke

•Smallsam

plesize

Preston&New

port(2014)

Noisy

visualfeedback

training

impairs

detectionofself-generated

movem

enterro

r:Implications

for

anosog

nosia

forh

emiplegia

22healthyparticipantswith

nohistoryofneurolog

icalinjuryor

illness;allrig

ht-handed


reachou

tand

touchatargetwith

onearm;feedb

ackwas

givenusingavBOTrobo

ticmanipulandu

m.

Noise

Cond

ition

—Feedback

provided

was

inaccurate

basedon

theparticipant’s

movem

ent.

No-Noise

Cond

ition

—Feedback

provided

was

basedon

theparticipant’s

movem

ent.


give

asubjective

judg

mento

ftheirperfo

rmance.

Thecond

ition

(noise

vs.nono

ise)h

ada

significante

ffecton

theparticipants

self-judg

ment(p

=.0008).

Streng

ths

•Novelstud

yLimitatio

ns•Implem

entatio

nof

motor

correctio

ndu

ringreaching

couldhave

been

activated

atan

unconsciou

slevel

Imag

ingevidence

Fotopo

ulou

etal.(2010)

Implicitaw

arenessinanosog

nosia

forh

emiplegia:Uncon

scious

interfe

rencewith

outcon

scious

re-representation

14adultpatientsrecruitedfro

man

acutestroke

rehabilitation

center;7

patientswith

anosog

nosia

and7patients

with

outanosog

nosia

AHPconfirm

edutilizing

theBertietal.(1996)

scaleandtheFeinberg

etal.(2000)scale.

Globalcog

nitivefunctio

ning

assessed.

Patient

lesio

nsmappedon

slicesof

the

T1-weigh

tedMRIscan

template.

Lesio

nanatom

icallocatio

nandvolumes

estim

ated.

Alllesio

nslocatedintherig

ht-hem

isphere

MCA

territo

ry.

Sign

ificant

anatom

icallocatio

nsof

individu

alswith

AHPinclud

eareas

extend

ingto

theRo

land

icop

erculum

andanterio

rinsulato

thecaud

ateand

putamen

nuclei,and

inferio

rly,lesions

extend

ingfro

mtheam

ygdalaand

superiortem

poralpole.

Thesupp

lementarymotor

area

spared

inall

patientsdiagno

sedwith

AHP.

Streng

ths

•Balancedsamplewith

nodifferences

betweengrou

psin

age,education,tim

eof

stroke,

orglob

alcogn

itive

functio

nLimitatio

ns•Smallsam

plesize

•Lesionalanalysis

isun

reliable

dueto

anatom

icaldifferences

betweenindividu

als

(continues)

6 Anosognosia


Table

1Matrix


insynthesis,Con

tinued

Autho

r/Date/ArticleTitle

Sample


DataAnalysis

Streng

thsandLimitatio

ns

Besharatietal.(2014)

Theaffectivemod

ulationof

motor

awarenessinanosog

nosia

for

hemiplegia:Behavioraland

lesio

nevidence

16adultn

eurologicalpatients

with

right-hem

isphere

lesio

nsrecruitedfro

mastroke

rehabilitationun

it8classifiedas

having

AHP,with

theother8

servingas

the

non-AHPcontrolgroup

AHPassessmentbasedon

theBertietal.

(1996)scale.

AHPconfirm

edwith

theFeinbe

rget

al.

(2000)scale.

Globalcog

nitivefunctio

nassessed

utilizing

theWTA

R,theMMSE,the

WechslerA

dult

IntelligenceScaleIII,the

Mon

treal

Cogn

itive

Assessm

ent,theBehavioral

InattentionTest,the

Fron

talA

ssessm

ent

Battery,andtheCo

gnitive

Estim

ates

Test.

CTandMRIscansob

tained

from

each

participantand

manually

reoriented

using

statisticalparametric

mapping

then

reconstructedon

totheMon

treal

NeurologicalInstitutetemplate.

Voxel-b

ased

lesio

nsymptom

mapping

techniqu

eutilizedas

well.

Alllesio

nswerefro

mafirst-everstroke,

mainlyintheMCA

territo

ry.

TheAHPgrou

phadoverlayof

lesio

nsand

involvem

entof

corticalandsubcortical

areascomparedto

theno

n-AHPgrou

p,which

hadmorefocaldam

agein

subcorticalareason

ly.

Thepo

steriorinsula,angu

larand

superior

tempo

ralgyri,andinferio

rfrontalgyrus

weresig

nificantly

associated

with

differences

inaw

areness.

Streng

ths

•Sam

pledidno

tdifferb

ased

onage,education,or

general

cogn

itive

functio

ning

Limitatio

ns•Smallsam

plesize

•Lesionalanalysis

isoften

unreliable

Baiere

tal.(2014)

Ano

sogn

osiaforh

emiparesis

afterleft-sid

edstroke

66participantsscreened

with

1subjecteligiblefor

participation

62-year-oldrig

ht-handedwom

anwith

left-hemisp

here

stroke

participated

inthestud

y

AHPconfirm

edwith

theBisiach

etal.

(1986)scale.

Degreeof

paresis

oflim

bsmeasuredwith

Likertscale,where

0=no

movem

ent

and5=norm

almovem

ent.

Cogn

itive

impairm

enttestedutilizing

the

Mini-M

entalTest.

Quo

tient

forh

andedn

essassessed

according

toEdinbu

rghtest.

fMRIutilizedto

assessbloo

dflowdu

ring

sentence

generatio

n.

fMRIconfirm

edthatthelang

uage

center

forthisindividu

alwas

locatedon

the

right

side.

Lefthemisp

hereplayed

ado

minantrolein

self-aw

arenessconcerning

limbfunction,

which

iscontradictoryto

literaturethat

attributesAH

Pto

right-sided

lesions

only.

Streng

ths

•Procedu

reandmeasuresused

arevalidated

inprevious

stud

ies

Limitatio

ns•C

asestud

ywith

onlyon

eparticipant

•Aph

asiafollowingleft-sid

edstroke

isstillan

obstaclefor

recruitm

entinAHPstud

ies

(continues)




Table

1Matrix


insynthesis,Con

tinued

Autho

r/Date/ArticleTitle

Sample


DataAnalysis

Streng

thsandLimitatio

ns

Interventio

nsFotopo

ulou

etal.(2009)

Self-ob

servationreinstates

motor

awarenessinanosog

nosia

for

hemiplegia

Case

stud

y67-year-oldrig

ht-handed

individu

alwith

aleft-sid

edstroke

recruitedto

participate

AHPconfirm

edutilizing

theBertietal.

(1996)scale.

Globalcog

nitivefunctio

nassessed

utilizing

theCo

mbandRazorTest,Hayling

Test,ProverbsTest,and

Cogn

itive

Estim

ates

Test.

Theparticipantwas

askedifshecould

perfo

rmcertaintaskswith

herp

legiclim

b,was

askedto

completethetask,and

was

then

askedho

wshefeltsheperfo

rmed.

Theassessmentwas

filmed,and

shewas

askedifshewou

ldliketoseethevideoclip

ofherd

uringtheprevious

assessment.

Theparticipanthadasudd

enand

completetransformationinaw

areness

whenseeing

herselffrom

athird

-person

view

viavideoplayback.

Theparticipantalso

hadan

emotional

reactio

nto

realizingherd

isability.

Streng

ths

•Novelstud

yLimitatio

ns•C

asestud

y•Largerstudies

areneeded

togeneralizethisfinding

tothe

AHPpo

pulatio

natlarge

Besharatietal.(2015)

Ano

ther

perspectiveon

anosog

nosia:Self-o

bservatio

nin

videoreplay

improves

motor

awareness

2independ

entcase

stud

ies

perfo

rmed

by2independ

ent

research

grou

psintheUnited

King

dom

andItaly

2participantswith

right

MCA

infarctsrecruitedfor

participation

AHPconfirm

edutilizing

theBertietal.(1996)

scale,andconfirm

edwith

theFeinberg

etal.(2000)scale.

Participantw

asaskedifthey

couldcomplete

atask,thenaskedto

demon

stratethetask,

andthen

askedho

wthey

believe

they

perfo

rmed.

Theparticipantwas

then

askedto

view

avide

oof

theaboveproced

ure.

Self-ob

servationutilizing

videoplayback

contrib

uted

tothereinstatem

ento

fmotor

awarenessof

both

individu

als.

Bothparticipantsexperienced

anem

otional

reactio

nto

realizingtheird

isability.

Streng

ths

•Based

oninterventio

ndescrib

edby

Fotopo

ulou

etal.

(2009)andhadsim

ilarfinding

sLimitatio

ns•Twoindepend

entcasestud

ies

•Largertrialsareneeded

before

thesefinding

scanbe

generalized

totheAHP

popu

latio

nMoroet

al.(2015)

Error-basedtraining

andem

ergent

awarenessinanosog

nosia

for

hemiplegia

4patientswith

left-sid

edhemiplegiaand

right-hem

isphere

stroke

recruitedfro

maneurolog

ical

rehabilitationun

it

AHPwas

confirm

edutilizing

theBisiach

etal.

(1986)scale.

Globalcog

nitiveaw

arenessassessed

viathe

MMSE,the

BehavioralInattentionTest,the

CombandRazorTest,visualextin

ction

andverbalmem

oryassessments,and

the

Fron

talA

ssessm

entB

attery.

Allpatientsscreened

ford

epressionutilizing

theDepressionInventory.

Participantswereaskedifthey

couldperfo

rmacertaintask

utilizing

theirp

legicarm,

then

askedto

perfo

rmtheactio

n,and

then

askedto

reflectup

onpo

tential

reason

sforfailure.

Attem

ptsto

perfo

rmactio

nsfollowed

bya

structured

analysisof

perfo

rmance

can

contrib

uteto

therecoveryof

awareness

inpatientswith

AHP.

Allparticipantsimproved

awarenessand

maintaintheirrecovery.

Allparticipantshadan

emotionalreaction

whenaskedto

reflecton

theirinability

toperfo

rmtasks.

Streng

ths

•Novelstud

yLimitatio

ns•Smallsam

plesize

Note.MMSE

=Mini-M

entalStatusExam

ination;AHP=anosog

nosia

forh

emiplegia;WTA

R=WechslerTesto

fAdu

ltReading;MCA

=middlecerebralartery;fMRI=functio

nalm

agnetic

resonanceimaging;HP=hemiplegia;

CT=compu

terized

axialtom

ograph

y.

8 Anosognosia


From: Moher D, Liberati A, Tetzlaff J, Altman DG, The PRISMA Group (2009). Preferred Reporting Items for Systematic Reviews andMeta-Analyses: The PRISMA Statement. PLoS Med 6(7): e1000097. doi:10.1371/journal.pmed1000097

Figure 1. PRISMA Diagram.


occurred. Over time, the motor cortex should be updatedthat movement did not occur in the plegic limb; but in thestroke patient, this updating does not occur via the nor-mal feedback loop, and there continues to be a discrep-ancy between the action initiated by the controller andthe feedback produced by the predictor (see Figure 1).The inability to update via a typical feedback loop resultsin a state of unawareness of deficit or disability (Frithet al., 2000) that manifests itself clinically as an almostdelusional denial of deficit. According to this theory, thepatient’s awareness is dominated entirely by their intentionto move versus the failure to process sensory information


from the plegic limb that indicates that the movement didnot occur (Fotopoulou et al., 2008).

Vocat et al. (2013) have termed the failure of the feed-back loop the inability of one to update his or her beliefs.These researchers hypothesized that anosognosic individ-uals would fail to change their beliefs when confrontedwith conflicting information when compared to the per-formance of control participants undergoing the sameprocedure (Vocat et al., 2013). In Vocat et al.’s experi-ment, 11 control participants with no evidence of existingneurological injury or disease and nine right-hemisphericstroke patients were given clues that described words that



Figure 2. Theoretical Framework for Anosognosia for Hemiplegia after a Stroke.

10 Anosognosia

were progressive in nature and then asked after the intro-duction of each new clue to guess what the word was.For example, the first clue was ambiguous in detail, witheach additional clue providing more precise informationregarding the target word. Participants were also instructedto rate their confidence in their answer. Compared to thecontrol group, the anosognosic individuals showed signif-icantly fewer correct responses and were more likely torepeat incorrect responses from a previous clue. This pat-tern shows that the anosognosic patients were unable toupdate their beliefs and modify their responses even whenconfronted with a new clue that was incongruent with theirprevious guess. It should also be noted that there was nodifference in the control group and the experimental groupin terms of global cognition, general reasoning ability, andsemantic knowledge (Vocat et al., 2013). This study sug-gests that a deficit in the generation of and adjustment ofbeliefs when confrontedwith reality is associatedwithAHP.

In a later study, Saj et al. (2014) utilized an encodingand recognition intervention to investigate the relationshipbetween action intention and the self-monitoring of move-ments. Five control and 10 first-time right-sided strokepatients were asked to move their arm or imagine movingtheir arm (if plegic), and then, in the second phase the indi-vidual was asked whether the action was realized, or per-formed as directed, or imagined. Again, as in the priorVocat et al. (2013) study, there was no difference in neuro-cognitive testing between groups, which further supportsthat AHP cannot be explained or accounted for based on


global cognitive function. The AHP group produced ahigher number of incorrect responses; they also reportedmovement when no movement occurred at a higher ratethan the other groups. Incorrect recall of nonexecutedleft-sided motor movement was the most frequent errormade by anosognosic patients. The results of this studyfurther support the theory that a disturbance affecting anindividual’s ability to detect a mismatch between intentionand movement realization plays a significant role in AHP(Saj et al., 2014). Fotopoulou and colleagues (2008) usedthe same method for testing the relationship betweenmotor intention and motor awareness. In this experi-ment, a prosthetic arm was used to generate false visualfeedback to control participants and four anosognosicparticipants. The control group had a high level of correctresponses when confronted with false visual feedback.The AHP group, however, was impaired in detectingmovement of the rubber hand only in the conditionwherethey had intended to move their hand. In other words,when the anosognosic patient was asked to move theplegic limb and the prosthetic arm was used to generatefalse visual feedback of that movement, the anosognosicpatients believed they had generated the movement. Thistheory-testing study also supports the notion that motorintention to move drives motor awareness, and becausethere is a failure to update the actual position of the limbbecause of damage to the comparator system, anosognosicpatients believe they have moved an impaired limb whenthey have not (Fotopoulou et al., 2008).



Jenkinson et al. (2009) utilized a grip selection task totest motor imagery and motor control.Motor imagery is amental process by which an individual is asked to visualizecompleting a certain motor task and then to verbally de-scribe the outcome of the task. Motor control is the indi-vidual’s ability to complete the task as required. Eighteenpatients with a dense left hemiplegia were matched with22 healthy volunteers. In the motor imagery task, the par-ticipantswere asked to visualize reaching out and grabbinga wooden dowel. One half of the dowel was pink, and theother half was yellow. Participants were asked to verbalizewith each grip which color their thumb would face. Witheach grip task, the dowel was rotated 45° so that the indi-vidual would have to imagine how their hand would gripthe dowel. For the motor control procedure, the participantswere asked to reach out and grip the dowel as it rotated 45°each time.The researcher recorded the orientation of the in-dividual’s thumb in relation to the color of the dowel, andthe responses between intention and control were com-pared. The hypothesis of this study was that patients withAHP can generate motor representations concerning boththeir plegic and functioning limbs. In the anosognosic sam-ple, the motor imagery and the motor control accuracywere significantly lower versus that of the healthy volun-teers. Jenkinson et al. (2009) concluded that the predictedsensory consequences of movement form the basis of motorawareness, which is consistent with the previous findingthat motor intention informs motor awareness. Both ofwhich overrides sensory feedback in anosognosic patientsand indicates a failure of the sensory feedback loop to themotor cortex (Jenkinson et al., 2009).

Lastly, Preston and Newport (2014) hypothesized thatthe comparators, which are theorized to be active memoriesof movements and sensations, are not damaged but may beexperiencing a pathological slackening of awareness thresh-olds. Because of the damage left to cortical tissue after a stroke,typical processes, such as the fine-tuning ofmotormovements,fail to reach conscious awareness. Preston and Newport(2014) tested this theory on 22 neurologically healthy controlsand simulated noisy visual feedback of reaching movementsusing a manipulatable robotic arm. Participants were askedtomove a cursor on a computer to a target andwere requiredto complete the task in 1,250 milliseconds. The individualswerepresentedwith screen images thatwere either anaccuraterepresentation of their movement or a noise-induced repre-sentation of their movement, meaning that the cursor de-viated from the individual’s actual course of movement.The results of this study suggest that if noise or deviations ofmovement were added to visual feedback, participants wereless likely to be able to perceive the deviations of movementand attribute the deviations to themselves. These researcherssuggested that inducingor addingnoise increases the threshold


at which one becomes conscious and aware of discrepan-cies between motor intention and actual motor movement(Preston&Newport, 2014). These researchers argued thatthe threshold component explains why such large discrepan-cies can occur between patients with AHP. But, to subscribeto this theory means that a very important assumption mustbe accepted: That the visual noise that was introduced to thehealthy participants mimics that which patients with AHPexperience. It is more likely that AHP is an incredibly com-plex disorder that clinicians and researchers are just begin-ning to understand, and that its pathogenesis is related to acombination of deficits not yet entirely understood (Vocat,Pourtois, & Vuilleumier, 2011).

Imaging Evidence for AHP

Recent lesion mapping work of anosognosic patients fol-lowing a stroke has revealed that the majority of patientswith a history of AHP had an infarct of themiddle cerebralartery and its territory (Baier et al., 2014; Besharati et al.,2014; Fotopoulou, Pernigo, Maeda, Rudd, & Kopelman,2010). Fotopoulou and colleagues (2010) investigated asample of 14 patients poststroke. Seven of the patients wereconfirmed as having AHP per the classical Berti, Ladavas,and Della Corte (1996) scale and verified by the Feinberg,Roane, and Ali (2000) method of identifying anosognosia.Utilizing weighted magnetic resonance imaging (MRI), vol-umes of affected cerebral tissue and the anatomical locationof the lesions were compared to the seven-patient controlgroupwhohadalso experienceda strokebutwerenosognosic.All 14 of the patients had either a single ischemic or hemor-rhagic stroke thatwas confined to the right hemisphere,mostlyin the territory of the middle cerebral artery. Three of theanosognosic patients had largemiddle cerebral artery infarctsthat affected both cortical and subcortical tissue. Three otheranosognosic patients had subcortical damage affecting thebasal ganglia, insula, and surrounding white matter. The lastpatient in the anosognosic group had a large hemispheric le-sion that was mostly subcortical but extended to the medialparts of the frontal and occipital cortex.

Compared with the volume of affected tissues expe-rienced by the nonanosognosic group, there were no signifi-cant differences. Differences in anatomical location betweengroups revealed that the anosognosic group had a cluster oflesions that extended from the rolandic operculum and ante-rior insula to the caudate andputamennuclei. Therewas alsoevidence of some inferior lesion clusters involving the amyg-dala and the superior temporal pole. The findings of thisstudy conclude that the lesional differences between thegroups is the involvement of the insula, inferiormotor areas,basal ganglia and surrounding structures, and limbic struc-ture in the anosognosic sample. Also noteworthy is that in



12 Anosognosia

this study, as in Berti et al. (2005), the supplementarymo-tor area in the anosognosic patients was spared, lendingsupport to the theory that motor planning is intact inanosognosic patients (Fotopoulou et al., 2010).

Besharti et al. (2014) used the classical voxel-basedlesion symptommapping approachon a sample of 16patientsrecruited from a stroke rehabilitation unit. Eight of thepatients tested positive for AHP per the Berti et al. (1996)scale, and the other eight patients served as controls. Asin the study before, lesion volume and lesion mappingwere compared between the groups. All the patients, withand without anosognosia, had right middle cerebral ar-tery infarcts. There was no difference in volume betweenthe groups. Via lesion mapping, the nosognosic sampledid not have any lesional involvement that extended intothe subcortical areas. Individuals who had damage ex-tending to the putamen, the anterior insula, and the ante-rior periventricular white matter were more likely to beless aware of their deficit (Besharati et al., 2014).

There is a long-standing debate on whether AHP isa phenomenon that is restricted to a right-hemisphericinfarct. Previous data lend evidence that this disorder typ-ically presents after a right-sided stroke, and only a minor-ity of patients with left-sided strokes has been describedin the literature (Orfei et al., 2007). There is a concernthat potential patients who have suffered a left-sidedstroke and who may be anosognosic are excluded fromstudies because of their inability to participate in the clas-sical interviews used to confirm the presence of the disor-der. Left-sided infarcts often infringe on the language center,creating concern that aphasia may obscure the presenceof anosognosia (Hartman-Maeir et al., 2001).

Baier et al. (2014) tested 66 acute stroke patients withleft-sided infarctions for AHP and then utilized MRI todetermine whether the language center of the individualwas in the right or left hemisphere (Baier et al., 2014). Fromthe original sample, only 44 could be formally tested for thepresence of AHP. Of those 44, only one patient, a 62-year-old right-handed woman, tested positive for AHP afterstroke. Like previously mentioned theory-testing literature,though the patient was anosognosic, she did not havecognitive impairment when tested with the Mini-MentalStatus Examination. Yet, when asked about her plegic arm,she was grossly anosognosic. To test the hypothesis that herlanguage center was asymmetrically located in the righthemisphere versus the left, the researchers utilized a func-tional MRI procedure that detected changes in blood flowto different areas of the brain based on activation. The par-ticipantwas shownapicture andwas asked to state a simplesentence describing how the picture appeared. The functionalMRI procedure confirmed that this individual’s languagecenterwas located in the right hemisphere versus the left. This


study has implications moving forward in that it is specu-lated that up to 3%of right-handed individuals have rightversus left lateralization of language functions. The assump-tion that AHP only occurs after a right-sided infarct shouldbe investigated further in larger studies, and research toexplore the integration of left-sided stroke survivors intoAHP research should be pursued (Baier et al., 2014).

Interventions for AHP

Three researchers reported interventions for AHP basedon Vuilleumier’s (2004) ABC Model of Updating Beliefs.In each of the three (Besharti, Kopelman, Avesani, Moro,& Fotopoulou, 2015; Fotopoulou, Rudd, Holmes, &Kopelman, 2009; Moro, Scandola, Bulgarelli, Avesani,& Fotopoulou, 2015) studies, the participants were givenan opportunity to Appreciate their beliefs by being askedto perform a task with the affected limb. Then, the inves-tigator questioned the participant’s initial Belief on howwell they performed the dictated task. Lastly, the partici-pant was allowed the opportunity to Check their perfor-mance on the task by being asked questions on how theybelieve they did or by being confrontedwith video evidencethat they did not complete the task (Vuilleumier, 2004).The researchers each thought that the ability to observeoneself from a third person perspective could potentiallylead the participants to update their beliefs of their perfor-mance by revisiting the question aimed at understandinghow they perceived their performance on the task.

The foundational case study by Fotopoulou, Rudd,Holmes, and Kopelman (2009) describes a 67-year-oldright-handedwomanwhopresented during the acute strokephase with a large right middle cerebral artery infarct anda severe left-sided paralysis. The patient maintained thatshe couldmove her left side, though it seemed to beweakerthan her right side. Though she had a flaccid paralysisof the left side, she adamantly stated that she performedmany of the tasks that the investigator asked her to dowiththe paralyzed limb. She also frequently attempted to getout of the bed unassisted or stand from her wheelchair.Fotopoulou and colleagues video-recorded one of theirtask assessments of the patient and then asked if she wouldlike to see her performance on the recorded video. Theypositioned the camera in front of her showing her upperbody only and allowed her to watch the 90-second clipof the assessment. Immediately after watching the video,the patient spontaneously stated that her expectations hadnot been very realistic. The patient experienced a suddenand dramatic new awareness of her paralysis that persistedthe following day.When askedwhy she changed hermind,she stated that she did not realize that she looked like thatuntil she saw the video (Fotopoulou et al., 2009).



Besharati, Kopelman, Avesani,Moro, and Fotopoulou(2015) replicated themethods and intervention of the pre-vious study but applied the interventions to two patientsin the chronic phase of stroke recovery to determine thefeasibility and effectiveness of the intervention. Insteadof focusing on the upper body only, these researchers ini-tiated an intervention on one of the patients that includedboth the left upper and left lower extremities. Both pa-tients’ awareness was tested several times before the videointervention to ensure that there was a presence of AHP.One patient was an 88-year-old right-handed womanwith a dense right middle cerebral artery stroke. The sec-ond patient to receive the intervention was a 70-year-oldright-handedmanwith a right-sidedmiddle cerebral arterystroke as well. The methods of the experiment mirror theFotopoulou et al. (2009) experiment in that the partici-pant’s awareness and cognitive function was tested; theywere asked to perform a task, asked how they think theydid on the task, and then confrontedwith video evidence offailure to perform the task. The 88-year-old woman wasthe individual who received the intervention for the leftlower extremity, which mirrored the intervention for theupper body. Video replay of task performance in both ofthese participants contributed to significant reinstatementof motor awareness (Besharati et al., 2015).

The final intervention study utilized error-based train-ing to induce awareness of deficits in four patients withdamage to the right hemisphere.Much like the interventionsabove, three steps were followed: the researchers assessedparticipants’ awareness, the participants were asked to judgetheir ability to perform certain actions, and the partici-pants were asked to perform the action. After these threesteps, the investigator invited the patients to discuss theirperformance and perhaps identify some reasons that thetask could not be completed. It is noted in this publicationthat there were many discussions between the medicalstaff and the patient and family support system concern-ing the patient’s stroke and subsequent impairment. Evenwith these conversations, at baseline, all patients wereanosognosic and were unaware of their deficits. Thoughthe sample size was small, the intervention outlined abovecontributed to the recovery of awareness of motor defi-cits. All the patients in this study improved awarenessand maintained recovery of awareness over time (Moro,Scandola, Bulgarelli, Avesani, & Fotopoulou, 2015).

Another consideration for any interventions that seekto reinstate one’s self awareness is the potentially upsettingnature that the discoverymay have on the patient. In eachof the studies above, the researchers were cognizant tobuild a relationship with their patients and did not seek tointervene until they felt the patients were in a safe place.There were also provisions in the research protocols that


allowed the investigator to stop the intervention or the inter-view if the patient became too upset. At this point, therapeu-tic communication, understanding, and encouragement wereoffered to the participant, and the intervention was resumedat a later time. In each of the patient encounters, when self-awareness was realized, there was some form of emotionalresponse. One patient cried and wondered how she wouldever live or work again. Another patient withdrew andwouldnot interact with the investigator for a long period of time(Besharati et al., 2015; Fotopoulou et al., 2009;Moro et al.,2015). These emotional reactions should be a point of con-cern for anyone interested in intervening in the presence ofanosognosia. There should always be protocols and addi-tional support built in to any research program to help theindividual to cope with their disability in a healthy way.

Discussion

Although the concept of AHP has been in the scientific lit-erature for over 100 years (as cited in Langer, 2009), ourunderstanding of the phenomenon is evolving. Only 11studies were identified in the systematic review, and thecontent and foci of the 11 studies fell into one of threecategories: theory testing, imaging evidence, and interven-tions for individuals with AHP. By synthesizing the aboveresults, we can conclude that motor intention in affectedindividuals is preserved, and they are able to generate anintention to move via the motor cortex (Frith et al., 2000).Though the movement does not occur, sensory feedbackis based on previous movement memory rendering theindividual unable to update his or her beliefs and knowthat they are plegic on one side (Vocat et al., 2013). Thereis also biological evidence from lesion analysis studiesthat suggests that individuals who suffer from AHP havean infarct at or in the territory of the right middle cerebralartery (Besharati et al., 2014), withmore precise locationsof damage from the rolandic operculum and anteriorinsula to the caudate and putamen nuclei, and the amyg-dala and the superior temporal pole (Fotopoulou et al.,2010). Though AHP is known to be a disorder associatedwith a right-sided infarct, there is debate on whether thepresence of aphasia often associated with a left-sided in-farct renders one unable to participate in an AHP test orquestionnaire. Aphasia is thought to obscure the presenceof AHP in individuals with left-sided infarcts up to 40%(Baier et al., 2014). Future research and studies shouldseek ways to incorporate and include those with left-sided infarcts into studies concerning AHP.

There were few interventions that reinstated motorawareness in an individual with AHP after stroke. The suc-cessful interventionsutilizedvideoplayback that allowed theparticipant to view themselves attempting to complete a



14 Anosognosia

directed task (Besharati et al., 2015; Fotopoulou et al., 2009).This intervention utilizedVuilleumier’s (2004) “ABC”modelfor updating beliefs. The “Appreciate, Belief, Check”modelconfronts an anosognosic individual with video evidencethat they were unable to perform a task. Viewing this from athird-person point of viewwas successful in reinstating motorawareness in the studies described above (Besharati et al.,2015; Fotopoulou et al., 2009). Another method successfulin reinstating motor awareness utilized the same “ABC”model and gave the individual an opportunity to reflect onwhy certain actions were not performed as directed. Thisstudy used reflective reasoning and initiated the reinstatementof motor awareness to the participants involved (Moro et al.,2015). The intervention studies, however, had very smallsample sizes of two or three individuals per intervention.More research, including larger trials with more diversepopulations, should be conducted before these interven-tions are generalized to the anosognosic population.

In each of the intervention studieswhere an anosognosicpatient is confronted with his or her new disability for thefirst time, there was an emotional response of some sort.Some shed tears, and one participant withdrew from theresearchers and refused to participate until he had proc-essed the revelation (Besharati et al., 2015; Fotopoulouet al., 2009; Moro et al., 2015). Any research that seeksto intervene in AHP should consider the emotional conse-quences of confronting one with their disability for thefirst time. Measures that offer emotional support andcounseling should be part of any study design that seeksto reinstate motor awareness to one who is not aware.

Nursing and Clinical Implications

Anosognosia for hemiplegia after stroke continues to drawattention from scientists because of the profound effect thecondition can have on recovery and quality of life of strokesurvivors (Orfei et al., 2007). Individuals who have AHPhave motor impairments that result in gait and self-caredisturbances. The central issue of the phenomenon is thatthe individual believes that he or she can function in a normalmanner. Because of this, common safety precautions areoften disregarded by the patient, placing them at risk for aninjury both in the hospital and once discharged (Hartman-Maeir et al., 2001). Another consequence of anosognosiais an inability to understand the importance of attendingrehabilitation and participating in therapeutic interventionsthat aid in recovery. Often anosognosic patients will refuseto participate in therapy because they believe they are ableto function normally (Kortte &Hillis, 2009). Anosognosiahas also been associated with longer rehabilitation staysand with poorer functional outcomes among stroke sur-vivors (Hartman-Maeir et al., 2001). Specifically, this study


found that individuals with anosognosia after stroke weredeemed unsafe at discharge and never achieved indepen-dence in basic activities of daily living as evidenced bysignificantly lower Functional IndependenceMeasure scores1 year after discharge from the rehabilitation center.

Conclusion

Themajority of the literature concerning AHP is from neu-ropsychology journals, and the basic understanding andramifications of the phenomenon are just beginning to beunderstood. Hartman-Maeir et al. (2001) state that indi-viduals with anosognosia are more likely to disregard safetymeasures and are therefore more likely to fall, but the rela-tionship between anosognosia and falling has not been prop-erly established through research. Current gaps that must beaddressed include studies that investigate whether the pres-ence of anosognosia is a positive and reliable predictor forfalls during stroke rehabilitation. The interventions discussedin this article need to be conducted with larger sample sizes.Likewise, utilizing a robust experimental design will aid inbuilding evidence that the confrontationmethoddoes, in fact,reinstate awareness and can be used so that anosognosticpatients have safer and more effective rehabilitation stays.Likewise, the ethical concerns of utilizing video cameras orcell phones will need to be developed and tested to ensurethere are no Health Insurance Portability and Account-ability Act (HIPPA) violations and that the confrontationtechniques used have resources available to mitigate theemotional distress the patient could experience.

Conflicts of InterestThe authors report no real or perceived vested interestthat relate to this article that could be construed as a con-flict of interest.

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ucation articles related to stroke, go toenter.com.

This activity is also provider approved by the California Board of Registered Nursing, ProviderNumber CEP 11749 for 1.0 contact hour. Lippincott Professional Development is also anapproved provider of continuing nursing education by the District of Columbia, Georgia,Florida, West Virginia, New Mexico, and South Carolina, CE Broker #50-1223.

Payment:• The registration fee for this test is FREE for members through March 31, 2020, and $10.00after March 31, and $12.50 for nonmembers.1. ARN members can access the discount by logging into the secure “Members Only”area of http://www.rehabnurse.org.

2. Select the Education tab on the navigation menu.3. Select Continuing Education.4. Select the Rehabilitation Nursing Journal article of your choice.5. You will appear at nursing.CEConnection.com.6. Log in using your Association of Rehabilitation Nursing username and password.The first time you log in, you will have to complete your user profile.

7. Confirm the title of the CE activity you would like to purchase.8. Click start to view the article or select take test (if you have previously read the article.)9. After passing the posttest, select +Cart to add the CE activity to your cart.10. Select check out and pay for your CE activity. A copy of the receipt will be

emailed.



understanding anosognosia for hemiplegia after stroke

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