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Original Paper

Eur Neurol 2008;60:43–46 Received: August 29, 2007


Accepted: November 14, 2007
DOI: 10.1159/000127979
Published online: April 25, 2008

Transient Directional Disorientation as a


Manifestation of Cerebral Ischemia
Tadashi Ino Hideo Usami Kazuki Tokumoto Toru Kimura Kyoko Ozawa
Shigenobu Nakamura
Department of Neurology, Rakuwakai-Otowa Hospital, Kyoto, Japan

Key Words splenial lesion [2–6]. Functional neuroimaging studies


Directional disorientation ⴢ Functional magnetic resonance involving navigation in large-scale spaces have also
imaging ⴢ Cerebral ischemia ⴢ Parieto-occipital sulcus shown that the retrosplenial area is activated during the
task [7–9]. We found that the peak of activation in the
retrosplenial area during a mental navigation task is con-
Abstract stantly located in the parieto-occipital sulcus (POS) in
The authors describe 2 patients who presented with tran- the individual subject analysis [8]. We also reported an
sient directional disorientation (TDD) as a manifestation of fMRI study of a patient who presented with directional
cerebral ischemia. The patients suddenly lost sense of direc- disorientation following retrosplenial hemorrhage and
tion in a familiar environment despite preserved ability to proposed that the ‘sense of direction’ in a large-scale lo-
recognize landmarks, and recovered within a short time. comotor environment is subserved by the visual area
Brain MRI revealed an ischemic lesion in the right medial oc- along the POS, and that bilateral deterioration of this
cipital lobe and the corpus callosum in case 1 and in the right function causes directional disorientation [2]. Here we
parieto-occipital sulcus (POS) in case 2. After ictus, fMRI report 2 cases who suffered from transient directional
study of a navigation task was performed, which demon- disorientation (TDD). Anatomical and functional neuro-
strated the activation of the POS unilaterally in case 1 and imaging findings of these cases further support our hy-
bilaterally in case 2. We propose that TDD of our patients is pothesis.
related to temporary dysfunction of bilateral POS.
Copyright © 2008 S. Karger AG, Basel

Case Report
Introduction Case 1
A 72-year-old right-handed man with diabetes mellitus sud-
Directional or heading disorientation is defined as im- denly became unable to understand the direction to a familiar
paired sense of direction despite preserved recognition of store while walking from his home, on the night of August 15th,
2001. He was not aware of visual field defect and could recognize
buildings and landscape, resulting in an inability to nav- familiar buildings and landscape normally, but he could not de-
igate in a familiar environment [1, 2]. Most of the cases termine which direction to return home. He was finally able to
with this symptom have been reported following retro- return home after receiving assistance from a passerby. The next

© 2008 S. Karger AG, Basel Tadashi Ino


0014–3022/08/0601–0043$24.50/0 Department of Neurology, Rakuwakai-Otowa Hospital
Fax +41 61 306 12 34 Otowachinjicho 2, Yamashina-ku
E-Mail karger@karger.ch Accessible online at: Kyoto 607-8062 (Japan)
www.karger.com www.karger.com/ene Tel. +81 75 593 4111, Fax +81 75 581 6935, E-Mail rakuwadr042@rakuwadr.com
Fig. 1. Diffusion-weighted images (upper
part, TR/TE = 4,000/100 ms, b value =
1,000) and FLAIR images (lower part, TR/
TE = 8,000/110 ms) showing ischemic le-
sions in the right medial occipital lobe and
splenial portion of the corpus callosum in
case 1 and in the right POS in case 2.
FLAIR MRI in case 2 was taken 7 days af-
ter onset.

of April 20th, 2006. He decided to visit our hospital but he could


not find the front door of his own home. He called a taxi, managed
to find the entrance, and came to our hospital. When he arrived
at our hospital, he first went to the lavatory, but when he stepped
out from the lavatory he could not find the way to the emergency
room, even though he had visited many times. He was then
brought by the staff to the emergency room. Neurologically, he
was normal except for a left lower quadrantanopsia. Diffusion-
weighted MRI showed an ischemic lesion involving POS (fig. 1).
About 1 h after onset, he completely recovered his sense of direc-
tion and his left lower quadrantanopsia disappeared.

Functional MRI
fMRI study during mental navigation was carried out 29 days
after onset in case 1 and 7 days after onset in case 2. The experi-
Fig. 2. Activation of the APO during mental navigation task mental procedure was approved by the Medical Ethics Commit-
(k 1 30, p ! 0.05, corrected for multiple comparisons using fam- tee, and all patients gave written informed consent. Echo-planar
ilywise error correction), superimposed on the subject’s anatomi- images (TR = 5 s, TE = 50 ms, flip angle = 90°, FOV = 200 mm,
cal image normalized to the MNI standard space. matrix = 64 ! 64, slice thickness = 6 mm, gap = 1.2 mm) and a
three-dimensional T1-weighted image for anatomical normaliza-
tion (TR = 11.6 ms, TE = 4.9 ms, flip angle = 8°, FOV = 220 ! 220
mm, matrix = 256 ! 256, slice thickness = 2 mm) were obtained
morning, his directional disorientation had totally disappeared. using a 1.5-tesla Siemens Vision Plus. The design of the task was
Seven days after ictus, he came to our hospital at the outpatient identical to our previous reports [2, 8]; consisting of seven 30-sec-
clinic for periodic treatment of diabetes mellitus and was intro- ond mental navigation epochs, in which the patients were given
duced to our department by his physician. No abnormality was two names of familiar places in Kyoto City and were required to
found by routine neurological and neuropsychological examina- mentally navigate between them, and eight 30-second control ep-
tion. His visual field was normal by confrontation test. Diffusion- ochs, in which the patients were given two successive numbers
weighted and FLAIR MRI revealed ischemic lesions in the right and were instructed to count mentally from the last number. Im-
medial occipital lobe and the splenial portion of the corpus cal- mediately after the fMRI scan, the patients were required to de-
losum (fig. 1). scribe the route on the paper between the same places provided in
the scanner. Both subjects had lived in Kyoto City for more than
Case 2 30 years, and their correctness of the responses during fMRI was
A 74-year-old right-handed man with atrial fibrillation and confirmed by this assessment.
hypertension noticed left visual field defect in his home, at noon

44 Eur Neurol 2008;60:43–46 Ino/Usami/Tokumoto/Kimura/Ozawa/


Nakamura
The data were analyzed with Statistical Parametric Mapping ity. It is generally accepted that activation of the extrastri-
(SPM2). After realignment, normalization, and smoothing with a ate visual cortex is observed during visual imagery, al-
9-mm full-width, half-maximum Gaussian filter, statistical para-
metric maps corresponding to a comparison between the task though it is unsettled whether the activation of the pri-
condition and control baseline were generated. Activated areas mary visual cortex can be observed or not [16, 17]. There
were regarded as significant if more than 30 voxels exceeded p ! are scant data of the visual-processing-related functions
0.05, corrected for multiple comparisons using familywise error of the POS in humans, but recent study indicated that the
correction. The anterior bank of the POS (APO) was activated inferior portion of the POS is involved in the generation
bilaterally in case 2, and only the left side of it was activated in
case 1 (fig. 2). In case 1, activation of the right POS was not found of the first harmonic response of the steady-state visual-
even when the level of significance was set more liberal to p ! 0.05, evoked response caused by visual flicker stimuli [18]. It is
uncorrected for multiple comparisons. also reported that this region is related to the feeling of
vection, particularly rollvection [19].
In case 1, the activation of the right APO was not found
Discussion even when the level of significance was set at p ! 0.05,
uncorrected for multiple comparisons. On the other
Our patients presented with TDD as a symptom of ce- hand, our previous study showed that the APO was acti-
rebral ischemia. Recently, cases of TDD of unknown eti- vated bilaterally in 14 out of 16 subjects when the level of
ology have been reported [10, 11], and some of them were significance was set at p ! 0.05, corrected for multiple
considered to be an epileptic manifestation [10], but vas- comparisons [8] and in all subject at p ! 0.001, uncor-
cular origin was not suggested for these cases. Our pa- rected for multiple comparisons (unpublished data).
tients had vascular risk factors such as diabetes (case 1), Therefore it is reasonable to assume that the lack of acti-
hypertension (case 2), and atrial fibrillation (case 2). Sud- vation of the right APO in case 1 was caused by the stroke,
den onset and disappearance within a short time of their albeit this region was structurally normal as shown by
symptoms suggest artery-to-artery or cardiac embolism MRI. Although PET or SPECT was not obtained, fMRI
and prompt recanalization. MRI revealed a fresh infarc- will be more sensitive at detecting disturbance of a spe-
tion in both patients. In particular, the lesion was located cific function. This impairment may be caused by the dis-
mainly in the right POS in case 2. In fMRI during mental connection from surrounding areas due to the lesion of
navigation, activation of APO was found in both cases. the corpus callosum. Because he had already recovered at
In monkeys, newly defined visual areas (V6 and V6A) the time of fMRI scanning and could perform the navi-
have been identified in the APO by recent anatomical and gation task perfectly, it is possible that in the retrosple-
physiological studies [12, 13]. V6A includes neurons nial area normal function of only the left APO is adequate
whose activities were modulated during reaching toward for navigation. Normal function of only the right APO
a nonvisual memorized target [14]. Neurons which have will also be adequate for navigation, since it has been sug-
a receptive field anchored to head-centered coordinates gested that for navigation the right retrosplenial area is
irrespective of eye movement were also identified in V6A dominant over [4, 6] or equivalent [2] to the left retrosple-
[15]. These characteristics of neuronal activities will be nial area. Therefore, it is reasonable to assume that bilat-
effective for the perception of the directional relation be- eral APO were functionally disturbed when directional
tween the individual’s current position and the perceived disorientation developed, as suggested in our recent re-
object. Therefore, such neurons appear to be advanta- port [2]. The function of the APO on the side opposite to
geous to egocentric spatial processing during navigation the ischemic lesion would have been disturbed tempo-
although they have been considered to be implicated in rally by mechanisms such as diaschisis.
somatomotor processing in monkeys. We have observed Although TDD of vascular origin has not been de-
that the APO is activated strongly during mental naviga- scribed in the literature, it may not be a rare symptom and
tion task consistently across subjects [8]. This anatomical has been ignored because its pathophysiological signifi-
localization common across individuals appears to sug- cance is unclear.
gest that the function of APO during navigation is related
to relatively lower order cognitive function such as some
kind of visual perception, rather than higher order cogni-
tive function such as particular memory for spatial layout
of familiar environments since cortical representation for
the latter process will show great interindividual variabil-

Directional Disorientation Eur Neurol 2008;60:43–46 45


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