Veuruprreholopia Vol . 30, No . I, pp . 01-09, 1992

(X128-3932 .92 SO .ue+0
.00
1992 Pecgamnn Press plc

Primed in Great Britain.

NOTE
REVERSED LATERALITY OF CEREBRAL FUNCTIONS IN A NON-RIGHTHANDER : NEUROPSYCHOLOGICAL AND SPECT FINDINGS IN A CASE OF
`ATYPICAL' DOMINANCE"
A . PAnovANt,t P. PANTANO, M, h ito conk, M . IAt.OB(INi, V . DI PIENO and C . L . I.ENZI
Department of Neurological Sciences, University 'La Sapienza', Rome, Italy

(Recetued 29 March 1991 : accepted 29 July 1991)
Abstract -A 54-year-old non-right-handed man with positive familial sinistrality showed a pure right
hemisphere syndrome following a left hemisphere stroke . Severe right side hemineglect, lranscortical
motor dysprosodia, spatial dysgraphia and visuo-constructive impairments were observed . At no
time were the expected left hemisphere abnormalities such as aphasia, alexia, right-left disorientation
or finger agnosia noted . A left fronto-temporal subeortical lesion was documented on CT scan . A Tc99m HM-PAO SPECT study revealed no cerebral blond flow changes in the right hemisphere while
in the left hemisphere a fronto-temporo-parietal cerebral blood flow reduction was evident . This case
of a complete reversed laterality of cognitive functions argues for a distinction to be made between
`anomalous' cerebral dominance and 'atypical' cerebral dominance .

INTRODUCTION
THE r52N'Anomalous Dominance' (AD) has been widely used in reference to

those cerebral organization patterns of
cognitive functions that are not consistent with typical brain-behaviour relationships [120 .
Most left-handers (LH ) and right-handers (RH) have left hemisphere dominance for language (14) and a right
hemisphere dominance for visuospatial skills, spatial attention and several aspects of emotion [I 2J .
A number of studies, however, documenting exceptions to this standard have been reported and different profiles
of AD have been described with regard to clinical presentation . There is evidence, in fact, that a given cognitive
function may be represented in individuals varying in degree of lateralization [34] that is, either unilaterally (left
hemisphere or right hemisphere) or bilaterally (left hemisphere and right hemisphere)- In addition, the pattern of
lateralization of any one cognitive function may be independent of the pattern of lateralization of another cognitive
function [7, 29] .
With regard to intrahemispheric organization of cognitive functions, although anomalous dominance has been
correlated with anomalous clinico-anatomical relationships [6], there is evidence that this is not a rule [2] . It may be
questioned, therefore, whether the term `anomalous' should be extended to profiles of atypical lateralization
associated with appropriate intrahemispheric organization of cognitive functions .
Reversed laterality of cerebral dominance represents an unusual profile of AD in which all the specialized
cogni live functions associated with the left hemisphere are processed by the right hemisphere and vice versa [22, 38] .
A pure 'right hemisphere' syndrome without aphasia following a lesion in the left hemisphere, both in LH and RH,
rarely has been reported [II . 16, 22, 26, 33, 36, 38] .
We describe a case of a non-right-handed patient with strong familial sinistrality who, after a left fronto-temporal
infarct, had a complete reversed 'right hemisphere syndrome without aphasia and apraxia . Details of the
neuropsvehnlogieal findings . CT scan images and cerebral blood flow (CBF) as measured by Single Photon
Emission Computerized Tomography (SPECT) are presented below .

*Dedicated to Professor Carlo Loeh on occasion of his retirement .
tAddress all correspondence to : Alessandro Padovani, Department of Neurological Sciences, Universitz'La
Sapienza', Servizio di Neuroriabilitazione . Viale dell'Universita, 30. 00185
.. Rnma, Italy .
81

82

NOTE

CASE REPORT
A 54-year-old non-right-handed white male (F . A .) with 3 years of education, Suffered a cerebral vascular accident in
the territory of the left middle cerebral artery and was admitted to the Stroke Unit of the Department of
Neurological Sciences, Rome University Hospital, on 30 January 1990 . On the Edinburgh Handedness Inventory
test [32] the patient scored +84 which is characteristic of right-handedness . A left-hand preference during
childhood, however, and a strong familial sinistrafity was documented by history : the father, one of two siblings and
the daughter were, in fact, reportedly left-handers .
Al admission, fluctuating consciousness with agitation, confusion, mild disinhihition, press of speech and
anosodiaphoria were observed . At one week post-onset, as consciousness cleared, the neurological examination
evidenced mild left-gaze preference with turning behaviour to the left, right central seventh, mild dysarthria, severe
right hemiparesis, right hemianesthesia .. right Babinski sign, right side hemineglecl and both right visual and
auditory extinction on douhle simultaneous stimulation . .At discharge (6 weeks post-onset) these findings were still
present .
The Mental Status Examination (Table 1) was carried out 2 weeks post-onset and the patient was followed until
discharge . At this time, the patient was co-operative and attentive provided that external distractors were kept to a
minimum . Anosodiaphoria had disappeared and F .A . was clearly depressed and concerned about his motor
problems .

TABLE 1 . Neuropsychological lest scores of F.A .
WAIS Verbal It)
Information
Comprehension
Arithmetic
Similarities
Digit span
Vocabulary

98
12
8
6
9
9
12

PIQ
Digit symbol
Picture completion
Block design
Picture arrangement
Object assembly

Language
Boston naming test
Verbal fluency PFL
Memory
Logical memory (WEMS)
Visual memory (WMS)
Paired associated (WMS)
Ray auditory verbal learning list
Recall trial l
3
4
5
Recognition
Delayed recall
Stick design (BDAE)
Corsi block-tapping
Visual perception/spatial
Judgement of line orientation
Rey-Osterrielh complex figure copy
Hooper visual organization test
Mesulam visual cancellation (omissions)
left
right
Mesulam verbal cancellation (omissions)
left
right
Albert line cancellation (omissions)
left
right

46/60
23

10 .5
2
13
3

It
14
15
8
2
3

13,/30
16/36
8,80
1'30
8/30
1130
14,/30
2/20
17,/20

79
2
6
6
5
3

Non

83

During the examination, explorative behaviour toward the left side was consistently noted . Orientation to time
and place was normal . On the WAIS [40], F .A . achieved a verbal score of 98 and a performance score of 72 .
language
F .A .'s speech was fluent, monotonic, mildly tangential, without paraphasias, agrammatism or word-finding
problems .Comprehension was excellent even for complex material (Boston Diagnostic Aphasia Examination) [l5] .
Repetition was flawless . Visual confrontation naming (Boston Naming Test) [24] was adequate but perceptual
errors were noted . Verbal fluency, i .e . ward-list generation for words beginning with the letters P, F and L
(Controlled Word Association Test) [5] and word-list generation for animals, fruits and vegetables, was within the
normal range .
Right-left orientation was normal either to double-other person or double-own body commands as was finger
identification and naming (subtests of the Boston Spatial Quantitative Battery) [15] . A writing sample revealed a
severe spatial dysgraphia with perseveration, overwriting, hesitation, misformed letters and letter reversals . Reading
comprehension of paragraph length material as well as written calculation for three digit problems were impaired
secondary to his right hemineglecl .
/'rnxis
Bucco-facial praxis was intact as well as limb praxis . Whole body commands were performed correctly .
(duo-spatial

On Judgement of Line Orientation [4], F.A . achieved an age corrected score of 13, which placed him in the
severely defective group .
A severe right-side hemineglecl was evident on the following tasks : Line Cancellation Test II] . Verbal and Visual
Cancellation Test ('Fig . 1) [30], Line Bisection and drawings to command and copy . Drawings (Fig . 2) . also,
showed poor motor control, perseveration, hesitation with reiteration of strokes on the left side, spatial distortion
primarily involving the right side, overdrawing and closing-in . On visuoconstructive tasks, such as Block Design of
the WAN, the patient's performance was poor but improved when time constraints were removed . He tended to
overfocus on the left side of the design as well as to place more blocks than were called for on the left side . Closing-in
and broken configuration (i .e . loss of the 2 by 2, or 3 by 3 matrix) was frequently seen .
On a mental object assembly task (Hooper Visual Organization Test) [21] performance indicate severe
perceptual fragmentation .
.~(jcct

Prosody was assessed through an unslandardized bedside examination as described by Ross [35] . Production of
affective prosody and affective facial gesturing either spontaneously or to verbal command were severely restricted .
The patient's affective prosodic repetition, however, was normal as well as affective prosodic comprehension . In
addition, the patient was able to reproduce pantomimes of emotional gestures involving the face and to identify the
conveyed emotion . This pattern is consistent with Ross's description of Transcorttcal Motor Dysprosodia [35] .
.Memory

Performance on the Weehsler Memory Scale [39] was adequate for immediate and delayed recall of verbal
material . Immediate and delayed visual reproduction of designs, however, was severely impaired . On Stick Design
Reproduction (Boston Spatial Quantitative Battery) [15], performance was defective and massed by frequent figure
reversals .

NEUROIMAGING STUDY
Three weeks post-onset . F .A. performed CT and SPECT scans- CT scan showed a large left Pronto-temporal
subcortical infarct (Fig . 3) involving the pummen and the anterior limb of the internal capsule .
In spite of the non-standard dominance lateralization, the left occipital lobe was wider than the right whereas the
right frontal lobe was wider than the left, that is a typical profile of brain asymmetries in right-handers [8] . Regional
cerebral blood flow (rCBF) was assessed at rest by using a Tomomatic 564 (Medimatie . Denmark) SPECT device
and Te-99m HM-PAO [31] .
The study was performed by injecting iv . 20 mCi of Te-99m HM-PAO . Three slices were obtained at .-0.5 cm,
+4.5 em cord +8 .5 cm above the orbitomeatal line . Spatial resolution was 9 mm and slice thickness 10 mm, fullwidth at a half-maximum (FHWM) . Irregular regions of interest (ROIs) were manually drawn over the cortical
ribbon and the basal ganglia according to an anatomical reference atlas [37] .
Data analysis was performed by a semi-quantitative method by calculating the ratio between the activity (counts)
in a single region of interest (ROI) and the activity in the left cerebellar hemisphere, that was not interested by the
`diasehitic' phenomenon . The same procedure was performed for an age-matched healthy control group (n=3) . A

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Patients performance on the Mesulam s random letter cancellation test .

F it ; . 2 . Patient's spontaneous drawing of clock, daisy and cross .

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Fte . 3 . CT scan at 3 weeks post-onset reveals a left fronto-temporal subeortical lesion with
involvement of both the putamen and the anterior limb of the internal capsule .

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Frc . 4. SPECT scan at 3 weeks post-onset evidences a cerebral blood flow (CBF) reduction in the
left frontal, temporal and parietal lobe and left thalamus .

Non

87

95% confidence interval was calculated for normalized values of each cerebral RO1, in the control group . The
SPELT study showed a significant CBF reduction in the left froonto-temporal-parietal regions and in the left
thalamus (Fig . 4 ;, whereas no significant -CBF reduction was evidenced in the ROIs over the right hemisphere
(Table 2) and in the left occipital areas .
TAttLe 2 . Regions of interest (ROT) cerebral blood flow (ratio

ROI/cerebellum) in F.A . and healthy controls (n=3)
RO(
Right

Left

ant, frontal
post . frontal
ant, temporal
post, temporal
parietal
occipital
thalamus
ant, frontal
post. frontal
ant, temporal
post . temporal
parietal
occipital
thalamus

F.A .

Mean

Controls
S .D .
95% CI .*

0 .824
0.858
0.930
0 .914
0 .922
0 .961
0.939
0 .785t
0.681t
0 .671]
0 .7731'
0 .712+
0 .890
0 .716t

0 .886
0 .966
0 .988
0 .927
0 .957
0 .998
1 .020
0 .943
0 .964
0 .960
0 .974
0954
1 .000
1 .010

(0 .034)
(0 .111)
(0 .098)
(0 .040)
(0 .(05)
(0 .122)
(0 .108)
(0 .04!
(0 .110)
(0 .091)
(0 .050)
(0 .090)
(0 .104)
(0 .098)

0 .802-0.97
0 .690-1 .24
0 .744-1 .23
0 .828-1 .03
0 .813-1 .07
0 .694-1 .30
0 .747-1 .29
0 .840 1 .04
0 .691-124
0 .734-Lt9
0 .850-1 .10
0 .796-1 .22
0 .748-126
0 .748 1 .25

t Confidence interval .
* Below the lower 95% confidence interval CBF value .

DISCUSSION
Although F .A . sustained a left fronto-temporal infarct, he showed evidence of neither aphasia, right left
disorientation, nor verbal memory impairments . F .A. was not apraxic even though the location and extent of his
lesion on CT have been correlated to apraxic in right-handers [28J . In spite of a strong fight-handedness score, F .A .
has been considered as a non-right-hander given his personal and familial history .. highly suggestive of a right-hand
conversion during early childhood . The lack of apraxic, therefore, was not entirely surprising as praxis has been
found to be related with handedness [I 3J .
His behavioural deficits, however, were consistent with the pattern expected of a RH with a right hemisphere
lesion . The patient showed a transcortical motor dysprosodia, a persistent right-side unilateral neglect syndrome,
spatial dysgraphia, spatial dyslexia and non-verbal memory deficits . Additionally, on visuospatial and constructive
tasks, those features usually seen in patients with a right hemisphere lesion [23] such as piece-coral approach,
perceptual fragmentation and broken configuration were also demonstrated .
These clinical findings are, thus, consistent with a mirror'right hemisphere syndrome following a left hemisphere
damage [22] and support the occurrence of a `true' reversed laterality of cerebral dominance with attentional and
visuospatial functions inversely represented in the left hemisphere.
A possible involvement of the right hemisphere in the occurrence of the symptomatology as a consequence of
either direct vascular damage or transhemiapheric diaschisis can be ruled out by the CT and SPELT scans that
showed no morphological and functional alterations in any of the right hemisphere regions .
With regard to cerebral organization of cognitive functions, anomalous dominance has been assumed to be
associated with incomplete or bilateral lateralization as well as anomalous intrahemispheric representation of
cognitive functions [6J . This assumption has not been demonstrated in F .A . Rather, the full clinical picture of I- A, is
within the expectations of 'intrahemispheric' localization by standard clinical correlations for hemineglect [t7, 18,
20] :cod for visuospatial impairments [27] . The presence of unilateral neglect, motor perseveration, closing-in,
evidenced in F.A ., are consistent with frontal dysfunction [25] . Additionally, although the anatomical correlates of
transcortical motor dysprosodia have not yet been fully delineated in right-handers with typical cerebral
dominance . F .A . mirrors the reported case with transcortical motor dysprosodia [35] secondary to a subeortical
right hemisphere lesion .
These results, thus far, confirm the statement [3, 19] of a general conformity between the clinico-anatomical
relationships of classical syndromes and those observed in patients with inverted or atypical laterality [to . 11, 16,19,

88

NOTE

22, 38] . It may be assumed, therefore, that F .A . does not have an anomalous eerebmt dominance of cognitive
functions, but rather an atypical cerebral dominance, that may be defined as a non-standard cerebral dominance of
appropriately intrahemispherically organized cognitive functions .
It is conceivable that the two non-standard profiles of cerebral dominance may represent different underlying
neurobiotogicat mechanisms .
Major causes of unusual patterns of cerebral dominance are either a markedly delayed development of the left
hemisphere (given the earlier maturation of the right hemisphere and its lesser vulnerability to modulating
influences during fetal development) or early extensive damage of the left hemisphere (which is initially programmed
to be dominant for speech and language) [12] . These mechanisms, however, do not explain such cases as F .A . in
whom all functions of the right hemisphere are appropriately lateratized in the left hemisphere and vice versa .
It should be assumed, then, that factors such as those interfering with the ontogenic development of the brain, may
play an important role in the phenomena of 'anomalous' lateralization of cerebral dominance due to incomplete or
bilateral organization of cognitive functions, in particular language . On the other hand, 'atypical' lateralization of
cerebral dominance as seen in 'mirror' crossed aphasia as well as cases like F .A ., may rather entail a specific genetic
endowment [9] .
Additional cases, focusing on the nature and the extent of cognitive and affective dysfunctions and their cerebral
morphofunetional correlates could provide further clarification of non-standardized cases and provide further
support to the proposed distinction between 'anomatous' dominance and atypical' dominance of cerebral functions .
Acknowledgements The authors wish to thank Drs Edith Kaplan, Michael Alexander and Luigi Bozzao for their
critical reading of the manuscript and their helpful suggestions .

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