Sergio Della Sala y Anna Cantagallo

8/12/2019 Sergio Della Sala y Anna Cantagallo

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PRESERVED INSIGHT IN AN ARTIST WITHEXTRAPERSONAL SPATIAL NEGLECT

Anna Cantagallo1 and Sergio Della Sala2

(1

Unit Operativa di Medicina Riabilitativa, Azienda Ospedaliera, Ferrara, Italy;2Neuropsychology Research Division, Department of Psychology,University of Aberdeen, U.K.)

ABSTRACT

Several reports of cases of experienced artists showing neglect after a brain lesion canbe gleaned from the literature. The analysis of their drawings might provide better insightinto the symptoms of neglect than that of non-artistss production. However, most of thesereports are anedoctal. We describe in some detail the case of neglect of a distinguishedartist, the internationally known Federico Fellini (FF), whom we followed-up for twomonths after his right parietal stroke. The neuropsychological profile of his neglectsyndrome was characterized by left visuo-motor neglect which persisted for two months. Atonset, FF also showed indications of neglect dyslexia as well as some evidence of implicitprocessing of the neglected parts of visual stimuli. However, there was no sign of personaland representational neglect, and FF was well aware of his motor and attentional deficits.FFs neglect was characterised by several dissociations, of which the lack of functionalcarryover despite intact conceptual and semantic insight is the most relevant.

Key words: neglect, anosognosia, working memory, artist

INTRODUCTION

Several anedoctal reports of cases of experienced artists showing neglectafter a right brain lesion can be gleaned from the literature (Gardner, 1976;Wurtz, Goldberg and Robinson, 1982; Vigouroux, Bonnefoi and Khalil, 1990;Schnider, Regard, Benson et al., 1993; Halligan and Marshall, 1990, 1997;Mazzucchi, Pesci and Trento, 1994). Given their extraordinary graphic talent and

their familiarity with visuo-spatial processing, the analysis of their drawingsafter brain lesion might provide better insight into the symptoms of neglect thanthat of non-artistss production (Marsh and Philwin, 1987). However, none ofthese cases was investigated in depth.

We describe in detail the case of a distinguished artist, the internationallyreknown film director Federico Fellini (FF)1, who was also a distinguishedpainter and cartoonist (De Santi, 1982; Miro Gori, 1994; Mollica, 1991; for acomprehensive bibliography see: www.comune.rimini.it/fellini/). We followed-uphim for two months after a right parietal stroke which caused visual neglect.

Cortex, (1998) 34, 163-189

1 The Fellini Foundation (Rimini) and Fellinis sister allowed us to use his full name for this report.


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FFs neglect was characterised by an unusual pattern of associations anddissociations, the most relevant feature of which was his complete awareness ofboth his motor and space exploration deficits. This awareness, however, did nothelp him to overcome his attentional problems.

CASE REPORT

FF was 73 year old, when in August 1993 he had a stroke in the territory of the middlecerebral artery of the right hemisphere (see CT scan in Figure 1). He was right-handed andhad 13 years of formal education.

NEUROLOGICAL AND GENERAL NEUROPSYCHOLOGICAL EXAMINATION

FF was first seen by one of us (AC) at the Rehabilitation Dept. in Ferrara,

fifteen days after his stroke. He presented with a severe motor and sensorydeficit in the left part of his body, and a left visual field defect limited to theinferior quadrant, pointing to a lesion of the optic radiations, compatible with thelocation of his brain damage (Jacobson, 1997). Eye movements were preservedin all gaze directions. His head and eyes were not extrarotated rightwards. Motorsymptoms improved only slightly over the two-month period of our follow-up,and the sensory deficits remained unchanged. He also showed florid unilateralvisuo-spatial neglect. He was transferred to another hospital two months later,where he died at the end of October, because of a second, massive, stroke.

The patient was alert and eager to collaborate with the personnel of the

hospital. From the clinical notes, it is clear that he was aware of his motorproblems and deeply concerned by them. He also showed misoplegia, i.e. hatred

164 Anna Cantagallo and Sergio Della Sala

Fig. 1 CT scan of FF one week after the stroke showing the large lesion encroaching upon thetemporo-parietal regions of the right hemisphere. The CT scan was performed at the Hospital ofRimini.


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of his paralysed left arm (Critchley, 1974), to which he referred as a bloated,damp bunch of asparagi and which he touched with his right hand only todisplace it, showing some loathing.

He was oriented in time and space, although during the night he manifestedsome episodes of confusion, usually misinterpreting his room in the hospital forhis real home (Pick, 1903). He did not show any language difficulty orgeneralised cognitive derangement. His remarks were always appropriate andoften very witty, and he soon became a favourite among the therapists andphysicians and was fondly regarded by his fellow patients. He scored 28 on theMini Mental State Examination (Folstein, Folstein and McHugh, 1975), a scorewell above the cut-off for Italian population (Measso, Cavarzeran, Zappal et al.,1993); his only two errors were: missing the leftmost segment in the copy of thegeometrical figure, and a mistake in the mental calculation task (counting backby seven). His performance on the Object Recognition Test of the VOSP Battery

(Warrington and James, 1991) was normal. He was not prosopagnosic andpromptly recognised visitors, consultants and nurses. His performance on asimple perceptual discrimination task, i.e. the length discrimination of pairs ofsegments vertically arranged (Spinnler and Tognoni, 1987) was well above thethreshold of normality (26/32, cut-off = 17). He had no verbal short term (DigitForward Serial Span = 7) or long term memory deficits (Weschler MemoryScale = 110, Memory Quotient = 122, Wechsler, 1945; Associate Learningsubtest = 15, Logical Memory subtest = 12). He was tested on three differentoccasions with the Ravens Coloured Progressive Matrices, assembled verticallyto minimise the effect of visuo-spatial neglect (Gainotti, DErme, Villa et al.,

1986) respectively 25, 32 and 50 days after the stroke. On all occasions heperformed well above the cut-off (14.75/36) for the Italian population (Basso,Capitani and Laiacona, 1987), scoring respectively 20/36, 24/36 and 32/36.Some of his mistakes (respectively 5, 3 and 1 errors in the first, second and thirdtesting occasion) could be accounted for by his neglect.

In summary, FF showed no sign of general intelligence, language, simpleperceptual or verbal memory deficits.

Assessment of Unilateral Spatial Neglect

Extinction

FF scored flawlessly (10/10) on single stimulation in both auditory (snappingof examiners fingers) and visual (wiggling of the examiners fingers in thesuperior visual field) modalities and in both right and left field. However, whentested with bilateral stimuli, FF invariably missed the visual stimulus on the left(0/10), performed at ceiling with the right targets (10/10 in both modalities), andscored 8/10 on left-sided auditory presentation. To summarise, FF presented witha clear left-sided visual extinction. Auditory extinction cannot be ruled out,although a 20% drop on double stimulation is usually not considered sufficient

evidence of clinical extinction (e.g., Arboix, Junqu, Vendrell et al., 1990;Grote, Pierre-Louis, Smith et al., 1995).

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Perceptual and Visuo-Motor Neglect

FF was given tests to assess the presence of visual perceptual and visuo-motorneglect on two occasions separated by an interval of 30 days (see Table I).

He showed signs of unilateral neglect on virtually all tasks and somerecovery of function after a month (without neuropsychological rehabilitation).However, his performance remained poor during the last testing session (seeTable I).

On cancellation tasks (Albert, 1973; Diller and Weinberg, 1977; Gauthier,Dehaut and Joanette, 1989), FF crossed most of the right-sided targets and only

some located on the left-side. On copying geometric shapes (Arrigoni and DeRenzi, 1964) he omitted several details of the left part of the single items, even


TABLE I

FFs Performance on Tasks Assessing Visuo-perceptual and Visuo-motor Neglect

Test 20 days 50 days Cut-off post-stroke post-stroke

Visuo-perceptual tasksWundt-Jastrow area illusion L 6/20* R 20/20 L 14/20* R 20/20 L-R 2Word reading 81/91* 86/91* 3

(neglect errors: 4/10)* (neglect errors: 1/5)Non-word reading 17/30* 19/30* 3

(neglect errors: 4/13)* (neglect errors: 3/11)*Sentences reading 2/6* 6/6 Prose reading (20 lines) 6/20* 20/20 Horizontal lines-bisection(visual version)** nt from L + 3.30 < 4 / > + 4(30 single lines of various length: from R + 13.02* < 4 / > + 480-240 mm)

Visuo-motor tasksLines cancellation L 13/20* R 19/20 L 17/20* R 20/20 L-R 1

Bells test L 7/14* R 14/14 L 1/14* R 10/14 L-R

3Letter cancellation L 10/52* R 37/52 L 20/52* R 38/52 L-R 4Horizontal lines-bisection*** 1* 2* 7(3 sets of 3 single lines 204 mmlong)Horizontal lines-bisection(manual version)** nt from L + 12.84* < 4 / > + 4(30 single lines of various length: from R + 12.30* < 4 / > + 480-240 mm)Copy of geometrical figures 9/20* 11/20* 15(from Arrigoni and De Renzi, 1964)Copy of geometrical figures 4/12* nt < 7.18(from Carlesimo et al., 1995)Completion of geometrical figures 53/70* nt < 61.85

(form Carlesimo et al., 1995)Reys figure copy Omissions L 4/5 Omissions L 3/5 0,89(from Rapport et al., 1996) Omissions R 1/9 Omissions R 1/9 0.49Reys figure memory Omissions L 5/5 Omissions L 4/5 (from Rapport et al., 1996) Omissions R 7/9 Omissions R 6/9

* Pathological performance.** The distance from midpoint is expressed in millimetres. A minus sign indicates an error towards the left; a plusindicates an error towards the right.*** Scoring modality as indicated by Wilson et al. (1987) ranging from a best of nine to a worst of zero. A score of 1indicates a mean error between 11 and 15 mm towards the right, a score of two indicates a mean error of 6-10 mm.nt = not tested; = not available; L = left; R = right.


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Fig. 2 FFs performance on the Reys figure: first assessment, (a) copy and (b) memory, andsecond assessment, copy (c) and memory (d). Note in (d) the production of a clock with the roman

numbers from I to VI on the right and arab digits from 10 to 12 on the left and anticlockwise.(Original in colours)

a

b

c

d


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a

Fig. 3 Some examples of lines-bisection performances of FF. He often tended to personalise hisperformance (a). However, in so doing, some times he clearly showed that his perception of thestimulus reached the leftmost extremity (b).


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b


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when the task required him to fill in gaps on uncompleted outlines (Carlesimo,Caltagirone, Fadda et al., 1991; Carlesimo, Caltagirone, Gainotti et al., 1995).However, he did not show evidence of object-centred neglect in multiple arrays,a finding confirmed by his performance on the test devised by Gainotti et al.(1986).

FF was also asked on two occasions to copy the Rey figure (1944) and toreproduce it from memory after 10 mins (see Figure 2). His performance wasscored using the procedure proposed by Rapport, Farchione, Dutra et al. (1996),which takes account of the laterality of the omissions (see Table I), and thencompared with that derived from the means plus two standard deviations of thenorms provided by the same authors. FFs left neglect is apparent, and persistentafter a month. His memory performance also is rather poor. The patientsimaginative spontaneous production can be appreciated on the right side of hiscopies (Figure 2).

When asked to bisect three identical sets of three stepwise horizontal lines of

similar length on the same page (Wilson, Cockburn and Halligan, l987), FFdisplaced his mark consistently toward the right extremity (see Table I). Hisperformance on all these visuo-motor tasks improved only slightly in the secondtesting session after a month.

To gain some insight into FFs poor performance on lines bisection, wetested the patient following a procedure similar to that proposed by Reuter-Lorentz and Posner (1990). FF was asked to bisect two sets of 30 lines ofdifferent lengths randomly positioned at the right, centre or left of the page, withtwo testing procedures. One form was identical to the traditional clinical line-bisection assessment: bisect lines using a ball-point pen held in his right hand.

The second form, which Reuter-Lorentz and Posner (1990) refer to as directed-visual, involves the experimenter slowly performing the task and then askingFF to indicate verbally when he thought she had reached the midpoint.Moreover, half of the stimuli in both modalities had to be scanned from right toleft, and the other half from left to right. FFs performance is set forth in TableI. FFs performance was compared with that of ten normal controls, the worstperformance of whom was used as the cut-off score (Cantagallo, Ferraresi andBasaglia, 1993). FF scored very poorly on the manual form, irrespective ofwhether his hand was positioned at the left or the right end of the line. Hisperformance in the visual form was just as bad when the experimenter started

from the right end of the line. However, he improved considerably when theexperimenter moved from the left end of the line. These results are in agreementwith those of Reuter-Lorentz and Posner (1990). FFs poor performance on thevisual form starting from the right end of the line minimises the possibility ofinterpreting these data solely in terms of directional hypometria. These findingsare consistent with the hypothesis that visual orienting might play an importantrole in neglect, and perhaps in its rehabilitation (e.g., Riddoch and Humphreys,1983; Lin, Cermak, Kinsbourne et al., 1996; for a critical view see Riddoch,Humphreys, Burroughs et al., 1995).

FF had a tendency to add some graceful diversions on top of the lines he was

asked to bisect (Figure 3a). In so doing, he demonstrated that, at least in someinstances, he had implicit awareness of both the right and the left end-point of



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the line in the manual form, though still misplaced the bisect point rightwards(Figure 3b).

FF also showed overt neglect when first assessed on other tests measuringonly the processing of visual stimuli without any motor requirements. On theWundt-Jastrow Area Illusion Test (Massironi, Antonucci, Pizzamiglio et al.,1988), he produced 14 neglect responses out of 20 stimuli on the left, whileperforming at ceiling on the right. He improved considerably in the secondtesting session (6/20 errors).

FF was also examined with tests assessing neglect dyslexia. He was asked toread aloud 91 words and 30 non-words of increasing length from 4 to 9 letters(Miceli, Laudanna, Burani et al., 1991). Each word was typed in bold large fontin the centre of a white card and presented individually. There was no timeconstraint to inspect and read the words. Errors (see Table I) were classifiedaccording to the criteria decribed by Ellis, Flude and Young (1987) as neglecterrors and non-neglect errors (these included non-word neologisms or reading

a non-word as a word). FFs neglect errors included omissions (e.g. grazie/thanks instead of disgrazie/misfortunes) and substitutions (e.g. piace/he likesinstead of giace/he lies). The pattern of his errors indicates that FFs neglectaffected the encoding of the letter position, not only of the letter identity (seecase VB, reported by Ellis et al., 1987). FFs performance was marginally betterafter a month. On the six-sentence reading test (Zoccolotti, Antonucci, Judica etal., 1989) FF showed a severe neglect dyslexia on first assessment, omittingwords in four sentences; deficits which vanished after a month. A similar resultemerged from the reading of a passage of text (20 lines); he omitted one ormore words at the beginning of 14 lines in the first testing session, but scored at

ceiling in the second assessment.FF showed no evidence of neglect dysgraphia either in spontaneous writingor writing from dictation.

To summarise, FF showed left neglect on a wide variety of visuo-motor taskswhich persisted two months after his stroke. He also showed neglect albeit lesssevere, on purely visual tasks, without a motor output, and some neglectdyslexia. However, his reading performance improved considerably by thesecond session.

Implicit Processing

FF was tested with the Burning House test (Marshall and Halligan, 1988)using the material devised by Bisiach and Rusconi (1990). In this test the patientis presented with four pairs of stimuli (two houses, two banknotes, two vasesand two glasses) one above the other, which differ only in one aspect: one of thetwo houses has flames coming from the roof, one banknote is torn in a corner,one vase has no flower protruding from one side, and the rim of one glass isbroken. The bad half of the stimuli is present either on its left or to its right.FF was presented with 32 such stimuli pairs, 16 right (four for each kind ofstimuli) and 16 left intermingled with 32 pairs of stimuli which were identical.

His task was to detect whether the two stimuli were same or different. Heperformed flawlessly with the pairs of different stimuli showing the oddity on



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the right, and failed to detect the difference in only three of the different pairs(banknotes) with the oddity on the left. Nevertheless, forced to chose which ofthe two banknotes he would prefer, he invariably chose the untorn one for thefollowing reasons: it is bigger, there is something written, there is noCraxi2.

The patient was also tested with 30 pictures, 15 of which were chimerae. Hewas asked to sort the real objects or animals from the chimeric compositions. Hemisinterpreted three chimerae for real objects (a horse with a big squirrels tailpointing to the left; a basket with a tennis racket handgrip on its left and atrombone with a rifle butt coming out from its left). FF was asked to name theobjects and answered respectively that they were a horse with a rather heavyback; a basket; a trombone to fire notes.

To summarise, there is some evidence that FF showed implicit processing forstimuli of which he neglected the left half.

Personal Neglect

Personal neglect was assessed with three tests that required movementstowards both sides of the body: the One Item Test for personal neglect and thenaming and pointing to one own body parts reported by Bisiach, Vallar, Peraniet al. (1986a) and the Comb and Razor test (Beschin and Robertson, 1997). InBisiach et al.s (1986a) One Item Test, the patient is asked to touch his left handwith his right. FF was tested on two occasions, 20 and 50 days after the stroke,he performed the task well, without hesitation on both instances. He alsoperformed flawlessly on the 12 items (four left, four right and four along themidline) of the naming and 12 items of the pointing to his own body parts, witheyes open. On the Comb and Razor test (Beschin and Robertson, 1997), FF wasasked to simulate combing his hair and shaving himself. The task was performedtwice, 20 days after the stroke and a month later. The score is calculated as thedifference in the number of strokes on the right and the left side of the body,and expressed as the percentage of strokes on the left relative to the totalnumber of strokes (left, right and ambiguous). FF scored 40% and 39% in thetwo subsequent sessions, which is a normal performance according to the cut-offscore (> 0.35%) provided by Beschin and Robertson (1997).

ln other words, FF did not show elements of personal neglect.

Representational Neglect

FF was asked to mentally visualise a familiar piazza from his home town andto describe it from a vantage point, either imagining himself at the extremeSouth or the extreme North of the square (Bisiach and Luzzatti, 1978). He wastested twice, 23 days after the stroke and a month later. On the first testingoccasion he reported five elements from the left side and two elements from theright side of the square. When asked to describe the same mental image from


2 Craxi is the former Italian prime minister involved in a notorius bribe scandal.


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Fig. 4 FFs drawing frommemory, twenty-five days after thestroke, of (a) a daisy, (b) a bike, (c) atable. Note the missing petals, themissing spokes on the left wheel and theuncompleted face of the cyclist. The

laid-out table was drawn on the rightside of the page, but there is no furtherevidence of neglect.

a

b

c


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the opposite perspective, FF reported three elements from the (now) left and twofrom the right. The re-test 30 days later showed a similar result (three elementsfrom the right, five from the left, and in the reversed perspective three from theleft and five from the right). Comparable results were gathered from other lessformal assessments. Asked to draw a map of his home town, imagining himselffrom the vantage point of the central railway station, FF reported three elementsfrom the left and three elements from the right of his imagined point of view.Asked to describe the map of Italy, he reported three towns from the left andtwo from the right half of the country, correctly placing Rome in the centre.

In summary, FF showed no obvious evidence of representational neglect in

tasks drawing upon long-term memory representations (visual imagery tasks withverbal reproduction).

Drawing from Memory

Further information about his visual imagery comes from drawing frommemory. It has to be emphasised that FF was a skillful and ingenious artist,known for his creativity, and well established as professional artist. Twenty-fivedays post-onset he was asked to draw with a blue ball-point pen each of thefollowing: a pair of scissors, a duck, an eagle, a bunch of grapes, a tree, a sofa,

the map of Italy, a daisy, a bike, a table (these last three drawings arereproduced in Figure 4), a human figure (Figure 5) and a face (Figure 6). In


Fig. 5 - FFs drawing from memory (twenty-five days after the stroke) of a female human figurefrom three different perspectives on three separate sheets of paper, (a) front, (b) back and (c) inprofile. In all three instances some details are missing from the left part of the human figure. In (a)the patient realised his omission, and made up for it adding in a second time with a red pencil theright arm of the woman he had drawn with a blue ball-point pen. In (c) the patient added thecommand draw me! as if coming from one of the female neuropsychologists assessing him .

a b c


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several instances FF omitted particulars on the left side, although his neglect, asdetected by this task, was rather mild (see Figures 4, 5 and 6). He also tendedto draw on the right side of the page, which he never did before his stroke.

During the same period of time FF produced several spontaneous drawings,from which there is very little evidence of unilateral neglect (see Figure 7). He


Fig. 6 FFs drawing from memory twenty-five days after the stroke of a human face. Someparticulars are omitted, such as the forehead of the female examiner and the back of his own head.Note that he expressed his feelings by depicting himself as a pygmy completely in the hands of hisexaminer.


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never showed any deficit in writing, either spontaneously or under dictation andalways demonstrated a proper use of the space in the page.

About a month later (sixty days after the stroke) his ability to draw recoveredcompletely and his characteristic style had re-asserted itself, as is clear from

Figure 8, in which three drawings are compared with one pre-stroke sketch.There is no hint of neglect in this more recent production.


Fig. 8 Three spontaneous drawings (a, b, c) of FF sixty days after his stroke compared withone sketch (d) he produced before his brain lesion [illustration taken from De Santi, 1981, Copyright(c) 1976 by Diogenes Verlag AG Zrich]. The style is equivalent and there are no signs of neglect.(Original in colours)

a b

c d


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To summarise, FF showed mild evidence of unilateral neglect, in drawingfrom memory, one month post-stroke. His drawing skills were completelyrecovered two months after the brain lesion.

Tactile Searching Task

FFs ability to create and manipulate mental representations of novelinformation was assessed with a tactile exploration task, the Tactile SearchingTest (De Renzi, Faglioni and Scotti, 1970; Beschin, Cazzani, Cubelli et al.,1996). This test consists of a maze cut into a wooden board. Four alleys (2cmwide and 2cm deep) spread from the centre ending in four arms: upper andlower right and left corners. The task of the patient, who was blindfolded, wasto move his forefinger from the centre of the maze and find a marble (target)that had been placed at the end of one of the four arms. The whole test

consisted of 32 trials, eight for each of the four possible positions. Following theprocedure of De Renzi et al. (1970), the time limit for each trial was 90 secs.Any failure to locate the target within the time limit was scored as an omissionand a searching time of 90 secs was computed. FF, tested 30 days after thestroke, found all the targets within the allotted time in the upper and lower rightpositions as well as in the upper left. However, he missed seven out of eighttargets in the lower left alley of the maze. His mean search times for the rightupper and right lower positions were 17.0 and 7.6 secs; however, his searchtimes for the left upper and left lower were 70.1 and 89.0 secs. The overalldifference in FFs searching time between the right and left half of the maze was

highly significant (t = 16.5, d.f. =15, p < 0.0001).To summarise, in the tactile maze test, FF showed neglect for the left space,

particularly evident in the lower quadrant. Remarkably, FF had a visual defectaffecting solely his left lower field. Unfortunately, FF was not examined withtests assessing directional hypometria (Mattingley, Bradshaw and Phillips, 1992;Bisiach, Geminiani, Berti et al., 1990; Tegnr and Levander, 1991), whichtherefore cannot be ruled out.

Awareness of Neglect

We have mentioned above that FF was well aware of his hemiparesis and thefunctional impotence it entailed. It is clear from the clinical notes, the interviewswith his relatives and friends, the patients diary, and his self-portaits asparetic, that he was not anosognosic for his motor deficit from the onset of hissymptoms. Faced with the possibility of a permanent deficit, FF reacted that hewould rather die than live as an incomplete man (Chandler, 1994, p. 368).During a conversation he revealed to a friend that Its terrible when the nind isgoing as fast as ever, faster, and the body will no longer take orders from it. Itslike being trapped in someone elses body (Ibid, p. 368). A few days after his

stroke, he was trying to amuse his relatives, scoffing at his fake left hand,which he could not move. On a further occasion, he explained in some length,



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to a friend, the neurological nature of his deficits (G. Angelucci, personalcommunication, 1996). He was also aware of his attentional deficits. Reportinghis experience of being tested with the Bell Test, FF told his friend that he couldnot see them all, but just to tease the clinician, he drew some extra bells, sothat his score would be good enough (Ibid, 1996).

When we first saw him, he was also aware that he had some readingproblems and that his drawings were not as good as they should be. Soon afterthe first neuropsychological testing session, two weeks after the stroke, hemanifested a clear awareness of his unilateral neglect: he asked us to change hisvisiting-card and to print on the new one: FF, Scoagulato, Emiparetico,Eminattento, Emianestesico (FF, unclotted, hemiparetic, heminattentive,hemianaesthesic). He spontaneously and thoroughly described his neglectproblems to the physiotherapists who were treating him. On one occasion, tryingto explain his symptoms to a visitor, he scornfully said that, had the sevendwarfs entered his room from the left, he would have not noticed them. FFs

awareness of his left neglect is testified by different sources of evidence. Somecan be gathered from test performances that we have discussed above.Immediately after he completed the line cancellation task, he added some extralines, stating that he was pretty sure he had forgotten some items on the left. Hemockingly wrote BASTAH (instead af basta/enough) and HO FINITOH(instead of ho finito/I have finished) at the end of his performance on theletter-H cancellation task, crossing-out his added Hs. One of the characters hedrew on the lines he was supposed to bisect (Figure 3b), rather explicitly ordersGo to the half-way point and the character on the left replies Forget it!.

In the following days, FF also produced spontaneous drawings in which his

awareness of his neglect was straightforward (see Figure 9a and b) as well as hsawareness of his failure in some easy visuo-motor task (see Figure 9c).

Despite knowing at a semantic level that he was neglecting this was notsufficient to overcome or reduce his phenomenologic expression of the spatialconsequence of his neglect.

DISCUSSION

FF was a remarkable artist who, after a right parietal stroke, presented with

multimodal unilateral left spatial neglect. However, he did not show personalneglect or representational deficits and was always well aware of his left motorand attentional problems. He also showed some hints of implicit processing ofthe parts of the stimuli he was not attending to.

FF is not the first artist with neglect to be described. Previous cases includeseveral German artists, whose works have been collected by R. Jung (1974);these include Anton Raederscheidt (Wurtz, Goldberg and Robinson, 1982) andLovis Corinth (Gardner, 1976; see also the cover of Parkins book, 1996); OttoDix, who had a right-sided stroke when he was 76 years old (Maffei andFiorentini, 1996); the British painter and sculptor Tom Greenshields (Halligan

and Marshall, 1990, 1997), who also showed other visuo-spatial distortions, suchas metamorphopsia (see the cover of the book edited by Halligan and Marshall,



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Fig. 9 Three spontaneous drawings of FF, twenty to thirty days after his stroke: (a) the patientasks himself Where is the left?, (b) the patient on the left of the page draws an elephant, which

asks Doctor may I stay here?, (c) the patient manifests his frustration at not being able to solvesome easy visual tasks (What is this?).

a

b

c


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1996); the Italian painter Guglielmo Lusignoli (Mazzucchi, Parma, Pizzamiglioet al., 1990; Mazzucchi, Pesci and Trento, 1994); and the cases reported byVigouroux et al. (1990) and by Schnider et al. (1993). Moreover, Marsh andPhilwin (1987) reported the case of the painter IK, who showed neglect of theright space in his paintings, due to a glioblastoma in the left posterior lobe.However, these artists were apparently unaware of their deficit (Gardner, 1976;Vigouroux et al., 1990; Schnider et al., 1993; Mazzucchi et al., 1994) or theirawareness paralleled the recovery of their visuo-spatial functions (Uhr, 1990;Halligan and Marshall, 1990; Mazzucchi, personal communication, 1997).

Awareness of Neglect and of Motor Deficits and Implicit Processing

FFs full awareness of his deficits is interesting. Neglect is often thought ofas a disorder of access to consciousness (Marshall, Halligan and Robertson,1993). Anosognosia (unawareness or denial of impairment) of visuo-spatial

disorders and (less frequently) of the sensory and motor deficits, is oftenmentioned as a cardinal feature of the neglect syndrome (Cutting, 1978;McGlynn and Schacter, 1989; Weintraub and Mesulam, 1989; Diller and Riley,1993; Heilman, Watson and Valenstein, 1993; Kolb and Wishaw, 1996; Bisiach,1998) and indeed it is maintained to be an essential part of its definition(Critchley, 1953; Weinstein and Kahn, 1955; Beaumont, Kenealy and Rogers,1996). The association between the presence and the severity of neglect andanosognosia is further supported by the high correlation between scores onanosognosia self-evaluation questionnaires and the performance on conventionaltests assessing neglect (Azouvi, Marchal, Samuel et al., 1996). Moreover,

Pizzamiglio, Antonucci, Judica et al. (1992) noted that an increase of thepatients awareness of their deficits was coupled with an improved performancein exploratory tasks. The link between awareness and recovery has beenreiterated several times (Gialanella and Mattioli, 1992; Diller and Riley, 1993;Hjaltason, Tegnr, Tham et al., 1996). Interestingly, Marshall et al. (1993) notedthat professional artists who have neglect but are unaware of it, fail to completetheir drawings, despite the fact that they have spent a considerable amount oftime (and attention) on the work (p. 316). Given the relevance of awarenessin rehabilitation techniques (Prigatano and Weinstein, 1996; Cocchini, 1996) andperformance improvement in tests of neglect (Riddoch and Humphreys, 1983;

Diller and Riley, 1993), it should be underlined that the complete awareness ofhis neglect symptoms did not help FF to overcome them (see e.g. Figure 3b).The pattern of performance for FF (e.g. in line-bisection tasks) seems to

demonstrate that overt extrapersonal neglect is possible even when the patient isconceptually totally aware of his left-sided deficits in exploration. Similarobservations can be gleaned from the literature. For instance, Azouvi et al.(1996) mentioned, in a fleeting comment, that one of their patients, a 63-yearman, examined eight weeks after his stroke, showed a severe left hemiplegia andneglect, of which he was well aware, as demonstrated by his performance on aquestionnaire ad hoc devised to assess the patients awareness of their everyday

difficulties, including neglect. Recently, Berti, Ldavas and Della Corte (1996)reported the cases of two patients, TF and TG, well aware of their neglect



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dyslexia and drawing neglect, who nevertheless made reading and drawingneglect errors. These findings speak against the interpretation of anosognosia asa defensive adaptation against the stress induced by the impairement (e.g.,Weinstein and Kahn, 1955).

FF also showed some evidence of implicit processing of parts of the stimulihe was not attending to. This finding replicates previous observations withvarious paradigms (Marshall and Halligan, 1988; Vallar, Rusconi and Bisiach,1994; McGlinchey-Berroth, Milberg, Verfaellie et al., 1993). We have arguedthat to account for this phenomenon it is possible to advocate a deficit of visuo-spatial working memory for the contralesional hemifield (Ellis, Della Sala andLogie, 1996; Beschin, Cocchini, Della Sala et al., 1997). The necessaryassumption on which this argument is based is that working memory is not aform of gateway between sensory input and long-term memory, but has to beconceived as a system which processes and manipulates information activatedfrom long-term memory (Cowan, 1988; Beschin et al.,1997; for detailed

discussion see Logie, 1995, and Richardson, Engle, Hasher et al., 1996). On thecontrary, it is postulated that, in normal circumstances, perceptual inputs are notavailable in working memory prior to long-term memory access. This hypothesisimplies that information might be implicitly processed accessing storedknowledge in long-term memory, without being available within workingmemory. A direct consequence of such an hypothesis is that patients withneglect should be able to covertly process meaningful percepts in their neglectedvisuo-spatial field but not meaningless stimuli, which cannot be activated fromlong-term memory traces. Indeed, this is what we have found in a recent seriesof experiments, comparing the implicit processing of information which could

and could not be stored in the subjects long term memory (Ellis, Della Sala andLogie, l996 and in preparation; Ellis, 1996). A second caveat of the workingmemory hypothesis as an account of implicit processing in neglect is the need toassume that a lateralised brain damage might give rise to a hemifield-specificvisuo-spatial working memory problem. Fortunately, recent investigationsof visual neglect in monkeys (Gaffan and Hornak, 1997a) and experimentswith patients who underwent a partial corticectomy (Gaffan and Hornak,1997b; Hornak, Oxbury, Oxbury et al., 1997) indicated the possibility that visualworking memory could be selectively impaired for the half-scenes presentedcontralateral to the side of the lesion, a deficit that the authors named

hemiamnesia (Gaffan and Hornak, 1997b). This hemifield specificity effect isthought to be even stronger than the right hemisphere specificity for visualmaterial (Hornak et al., 1997). These findings give new impetus to theinterpretation of the phenomenon of implicit processing of explicitly neglectedinformation in the hemifield contralateral to the lesion, as a partial failure ofvisuo-spatial working memory.

In conclusion, it might be postulated that the dissociation between impairedspatial performance coupled with awareness of such deficit shown by FF, mightbe mapped onto the relative impairment of visuo-spatial working memoryfunctions (responsible of the impairments in visuo-spatial tasks) and the intact

conceptual awareness, which capitalises on the good long-term memoryfunctioning.



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Dissociations in the Profile of FFs Neglect and Their Relation with Awareness

Neglect is a very complex syndrome, and indeed the term is used as anumbrella label to encompass various phenomena that can be observedindependently from one another (Halligan and Marshall, 1992, 1994; Kinsella,Olver, Ng et al., 1993; Kinsbourne, 1994). Several patterns of dissociationsamong the different symptoms (and different tests taxing the same symptom) ofneglect have been reported. The cognitive profile of FF supports some of thesedissociations.

Marshall and Halligan (1993) wondered whether there are patients who showneglect in situations where a visual stimulus is presented but have no neglecton imaginal tasks (p. 193). This is precisely the pattern shown by FF, whopresented with a clear extrapersonal neglect, but with preserved mentalrepresentations of space (see also Brain, 1941; Anderson, 1993; Bartolomeo,DErme and Gainotti, 1994). This evidence, taken together with the pure

representational cases recently documented (Guariglia, Padovani, Pantano et al.,1993; Beschin et al., 1997; Coslett, 1997) upholds the notion of a doubledissociation between representation and perception in neglect, undermining thehypothesis of a common underlying mechanism.

Bartolomeo et al. (1994) pointed out that some of the dissociations shown inneglect patients may reflect a difference in recovery rate or in compensatorymechanisms, rather than representing evidence of two independent deficits. Withregards to FF, this might explain the dissociation between reading and othertasks: the dissociation between dyslexia and other components of visuo-spatialneglect was apparent in the second testing session, but it was much more subtle

when we first examined the patient (see Table I). Most patients with neglectdyslexia also show visuo-spatial neglect (Ellis, Young and Flude, 1993). Thisled Ellis et al. (1987) to maintain that neglect dyslexia arises only in patientsshowing a more generalised visual neglect. However, possible dissociationsbetween the two signs can be gleaned from the literature (see also Ellis andYoung, 1995, ch. 8). For example, patient JOH reported by De Lacy Costelloand Warrington (1987) presented with left-sided dyslexia and right-sided neglectin copying, drawing and line-bisecting (but see Ellis et al., 1993, for a criticaldiscussion on the interpretation of this case). The modifications in time of thepattern of associations and dissociations between reading and other tasks of

visuo-spatial neglect shown by FF, call for some caution in conjecturing doubledissociations between neglect dyslexia and other signs of spatial hemineglect.A clearer dissociation shown by FF was that between extrapersonal space

and personal exploration. This kind of dissociation has been seldom reported,and indeed rarely looked for. FF who had a clear neglect for extrapersonalspace, performed normally on all tasks assessing personal neglect. Guariglia andAntonucci (1992) reported the opposite dissociation: their patient, EDS,following the surgical removal of a hematoma in the right parietal lobe,presented with a chronic form of severe personal neglect without signs ofextrapersonal neglect. Ramachandran, Altschuler and Hillyer (1997) included in

their elegant paper about mirror agnosia four patients who performedflawlessly on a personal neglect task while being severely impaired on tasks



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assessing extrapersonal space. Bisiach et al. (1986a) in a series of 97 right-braindamaged patients found only one case of personal without extrapersonal neglect,but nine cases of the converse dissociation. However, their assessment ofpersonal neglect consisted only of the order to touch ones own left hand.Similar cases in the acute stage of their disease, are sometimes referred to inpapers focused on other issues (e.g., Willanger, Danielsen and Ankerhus, 1981a,1981b; Vallar, Sterzi, Bottini et al., 1990). More systematic assessment ofpersonal neglect has been recently reported by Beschin and Roberston (1997),who assessed 44 brain-lesioned patients and 17 controls with a standardised testfor personal neglect (the Comb and Razor Test) and a battery of conventionaltests assessing extrapersonal space exploration. They reported several doubledissociations between personal and extrapersonal neglect. Although these doubledissociations were not strong enough to fulfil Shallices (1988) criteria, thefindings indicate that exploration of personal and extrapersonal space might wellbe separated. Further evidence supporting such dissociation, comes from the

study by Zoccolotti and Judica (1991), who reported very low correlationsbetween personal and extrapersonal items of classic neglect tests. The dearth ofobservations available does not allow definite conclusions. However, the profileof FF adds to the evidence gathered from the literature in pointing to a possibledissociation between the ability to explore personal and extrapersonal space.More cases, and systematic investigations are needed to settle the question.

The pattern of dissociations shown by FF reported above, is relevant to ourmain discussion about the association between neglect and awareness.

The presence of anosognosia cannot be simply traced back to the severity ofneglect as Levine, Calvanio and Rinn (199l) seem to imply discussing six patients

with marked extrapersonal neglect all anosognosic (see also Table 28.1 in thebook chapter by Feinberg, 1997, which summarises the conjoint occurrence ofneglect and anosognosia). In fact, two patients with very severe neglect and awareof their deficits have been reported in the Bisiach et al.s series (1986b).

Bisiach, Vallar, Perani et al. (1986b) demonstrated that extra-personal neglectcould be double-dissociated from anosognosia for motor impairment (see alsoBisiach and Geminiani, l991). Instead, the dissociation between unawareness andpersonal neglect was much less salient. On the other hand, reported patients withpure representational neglect (case NL, Beschin et al., 1997) and with pureextrapersonal neglect (such as FF) are well aware of their motor and attentional

deficits. Therefore, the alternative hypothesis might be postulated that neglectshould encompass more than one domain to be associated with anosognosia, orthat personal neglect is the key feature for unawareness to emerge (see alsoFredericks, 1963). Some support for this latter hypothesis comes from the studyreported by Cutting (1978), who found no cases of patients with personal neglectwithout anosognosia in a series of one hundred acute hemiplegics. Moreover,patient EDS with pure personal neglect mentioned above (Guariglia andAntonucci, 1992) although recognising the presence of his motor symptomswhen questioned, never demonstrated any awareness of the future consequencesof his deficit...or awareness of personal neglect (p. 1003).

A caveat to the link between lack of awareness (of ones motor deficits) andpersonal neglect, comes from the study by Adair, Na, Schwartz et al. (1995),



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who assessed the occurrence of anosognosia and personal neglect in twentypatients undergoing intracarotid barbiturate injection during evaluation forsurgical therapy of intractable epilepsy. All patients showed anosognosia, butonly four demonstrated clear personal neglect. This led the authors to concludethat personal neglect alone is neither necessary nor sufficient to precludeawarenes of a motor deficit (p. 2197). However, the lack of a doubledissociation between anosognosia and neglect weakens this conclusion, as theauthors themselves acknowledged. Another difficulty in linking a specific kindof neglect profile to anosognosia may come from the possible dissociationbetween awareness of ones own motor and spatial exploration deficits. Forexample, patient NF, reported by Beschin, Cubelli, Della Sala et al. (1997),showed clear awareness of his motor defcit, although he was anosognosic forhis neglect. Whether or not these two kinds of anosognosia should bedisentangled is matter for future investigations.

Further single cases showing different patterns of associations anddissociations of symptoms as well as sound descriptions of patients showingpure deficits of personal, extrapersonal and representational space exploration,together with group studies of unselected brain-damaged patients, are necessaryto verify or falsify this working hypothesis. The case of FF demonstrated thatneglect is a manifold phenomenon and allowed considerations into itsheterogeneity; his drawings and insight added some charm to it.

Acknowledgements. We thank Nic Beschin, Roberto Cubelli, Peter Halligan and OliverTurnbull, for their comments on a previous version of the manuscript; Hans Spinnler for hisencouragement; Giovanni Guerzoni and Peter Bates who helped us with the illustrations forthis paper; Claire Spivey who amended the English; and the Azienda Ospedaliera of Ferrarawho funded the visit of AC to Aberdeen. We also thank the Fellini Foundation[www.comune.rimini.it/fellini/] and his sister for their support and for their consent to thispublication. Most of all we are grateful to FF, who taught us how insightfulls lightness can be.

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Sergio Della Sala, Department of Psychology, University of Aberdeen, Kings College, AB24 2UB Aberdeen, U.K.E-mail: [email protected] Cantagallo, Unit Operativa di Medicina Riabilitativa, via Boschetto, 20, 44100 Ferrara, Italy.E-mail: [email protected]

(Received 28 July 1997; accepted 14 September 1997)


Sergio Della Sala y Anna Cantagallo

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