Crossed cerebellar diaschisis (CCD) is
defined as a reduction in blood flow in the
cerebellar hemisphere contralateral to the
supratentorial focal lesion. The phenomenon
termed parakinesia brachialis oscitans (PBO) in
which stroke patients experience involuntary
stretching of the hemiplegic arm during yawning
is rarely reported. The concurrence of CCD and
PBO has never been described. A 52-year-old man
had putaminal hemorrhage and demonstrated no
significant recovery in his left hemiplegia
after intensive rehabilitation, but his gait
improved gradually. Two months after the stroke,
the single photon emission computed tomography
(SPECT) showed CCD. Four months after the
stroke, the patient noticed PBO. The follow-up
SPECT showed persistent CCD and the patient's
arm was still plegic. The frequency and
intensity of PBO have increased with time since
the stroke. We speculate that the two phenomena
CCD and PBO might share similar neuroanatomical
pathways and be valuable for predicting clinical
recovery after stroke.
1. Introduction
Crossed cerebellar diaschisis (CCD) is
defined as a reduction in blood flow in the
cerebellar hemisphere contralateral to a
supratentorial focal lesion such as a cerebral
infarction or hematoma. CCD occurs in more than
50% of patients with supratentorial focal
lesions. Relatively few articles have described
CCD due to a subcortical hemorrhage [1].
In addition, less attention has been paid to the
phenomenon termed parakinesia brachialis
oscitans (PBO) in which stroke patients
experience involuntary stretching of the
hemiplegic arm upon yawning [2]. The
anatomical pathways involved in this involuntary
motor response are yet to be fully clarified,
and the prevalence of PBO is rarely reported.
The concurrence of PBO and CCD has never been
described. Here, we report the case of a stroke
patient with putaminal hemorrhage presenting
with concurrent CCD and PBO and discuss their
common anatomic pathway and the effects of these
conditions on clinical outcome.
2. Case Report
A 52-year-old right-handed man, who suffered
from hypertension for 5 years without regular
medicine and followup, was admitted to our
facility after acute loss of consciousness. The
patient exhibited left hemiplegia secondary to
right putaminal hemorrhage (3 * 5cm in size)
extending into the posterior portion of the
internal capsule with the mass effect. On
neurological examination, the patient had weak
muscle power (1/5 in the left upper limb and 2/5
in the left lower limb) and positive Babinski
sign on the left side. An emergent craniotomy
for removal of a hematoma was performed;
however, the patient demonstrated no significant
recovery in his left hemiplegia after the
operation. Results of a Romberg test and dynamic
testing were poor.The patient's Functional
Independence Measurement (FIM) score was 50, and
his Barthel index (BI) was 25. Amlodipine
besylate 5mg qd, valsartan 80mg qd, and
atorvastatin 10mg qd were prescribed for
secondary prevention of cerebrovascular disease.
Color Doppler sonography showed only mild degree
of intimal thickening and sparsemural calcified
plaques along the course of bilateral
extracranial common and internal carotid and
vertebral arteries as well without significant
stenosis. MR angiography of the circle of Willis
shows decreased signal intensity and number of
branches of right middle cerebral artery.
After intensive rehabilitation, the
patient's hemiplegia remained, but his gait
improved gradually. Two months after the stroke,
muscle power was reevaluated in the left upper
and lower limbs and was 2/5 and 3/5,
respectively. Single photon emission computed
tomography (SPECT) showed decreased perfusion in
the right cerebral cortex and in the left
cerebellum. CCDon the left side was confirmed
Four months after the stroke, the patient
noticed that he involuntarily stretched his
hemiplegic arm when yawning in bed. The movement
consisted of a progressive abduction,
anteroflexion, and mild internal rotation of the
shoulder, followed by armlifting with a flexion
of the elbow.Themovement lasted for a few
seconds. The involuntary activity was absent
when the patient was in a sitting position.
Repeated attempts to induce yawning by imitation
were unsuccessful. Eightmonths after the stroke,
a follow-up SPECT showed persistent CCD (Figure
1(b)). The patient's arm was still plegic, but
he could walk slowly using a cane with the
assistance of an ankle-foot orthosis.The
follow-up FIM score and BI were 102 and 70,
respectively. The patient continues to
experience persistent hemiplegia and CCD, and
his functional index has improved only
gradually.The frequency and intensity of the
phenomenon increased steadily with time after
the stroke and also occured when sitting.
3. Discussion
To the best of our knowledge, the present
case with putaminal hemorrhage togetherwith
CCDand PBOis the first such case to be reported.
Relatively few studies have been published on
CCD after intracerebral hemorrhage; however,
similar studies on cerebral infarction are
reported. The SPECT is a nuclear medicine
tomographic imaging technique and physicians
often utilize it for functional brain imaging to
assess blood perfusion of the brain such as CCD.
With respect to the relationship between
prognosis and CCD, Sobesky et al. [3]
reported that the CCD was significantly
corrected with the degree of supratentorial
hypoperfusion and persistent CCD closely linked
to poor clinical outcome and permanent
supratentorial tissue damage. Szil´agyi et
al. [4] also revealed that the severity
of CCD could be with a quantitative predictor of
functional impairment in stroke patients. In
contrast, Flint et al. [5] disagreed the
relationship between severity of CCDand volume
of ischemic stroke. The neuroanatomical pathways
contributing to CCD have been described as being
activated by disruption of the
corticopontocerebellar tract [6],
whichmainly connects with the corticospinal
tract in the posterior limb of the internal
capsule.Thecorticopontocerebellar fibers are in
close contact with the corticospinal tract, but
are more extensive than the corticospinal
fibers. In previous studies, most cases with CCD
showed pyramidal tract dysfunction. A reasonable
explanation could be that these two fibers are
close to each other and that most stroke
patients have pyramidal tract dysfunction.
Pantano et al. [7] also suggested that
destruction of the pyramidal tract is not
necessary for occurrence of CCD since some
patientswithouthemiparesis haveCCD, whilenot all
patients with hemiparesis have CCD.
The phenomenon of PBO described as
unintentional hemiplegia-associated movement
during yawing may appear during the flaccid or
spastic phase and tended to disappear when
neurological recovery was noted [2, 8].
The variants of classic PBO in two case reports
without paretic upper extremity were described
by de Lima et al. [9]. The yawning
center has not been truly identified. Some
clinical evidence suggests the major areas are
pons, medulla, basal ganglion, and hypothalamus,
particularly the paraventricular nucleus (PVN).
The PVN project to the lateral reticular
formation (couple ventilation and locomotion in
animals) and locus ceruleus of brainstem to
trigger yawning by exciting the cranial nerve
(V, VII, IX, X, XI, and XII) and phrenic nerve
[10].
Previous studies have concluded that most
PBO patients had lesions in the posterior limb
of internal capsule, involving damage to the
first neuron and interrupting the corticospinal,
corticonuclear, corticorubral, corticostriate,
corticonigral, and corticoreticular pathways
[2, 11]. Walusinski et al. [2,
8] found that the corticoneocerebellospinal
pathway (such as corticopontocerebellar tract)
is interrupted in certain PBO cases, but the
conducting system of proprioception between the
motor anterior spinal horn, the paleocerebellum,
and the lateral reticular nucleus
(spinoreticulocerebellar tract) remains intact.
They also speculated that the mechanism of
involuntary stretching of the hemiplegic arm
upon yawning is a motor signal of anterior
spinal horn in C4 to C8 which originate in the
lateral reticular nucleus and travel through the
extrapyramidal pathways of the archeocerebellum
(such as spinovestibulocerebellar tract).
Moreover, they suggest that interruption of
corticonuclear, corticospinal, and
corticoneocerebellar disinhibiting the
spinoarcheocerebellar tract might be the major
mechanism of PBO.
According to the known and hypothesized
mechanisms mentioned above, CCD and PBO seem to
share similar neuroanatomical pathways such as
corticopontocerebellar tract and the posterior
limb of the internal capsule. The possible basis
of the concurrence of CCD and PBO in our patient
was the putaminal hemorrhage that extended to
the posterior limb of the internal capsule. The
involuntary stretching in the plegic arm might
be useful in promoting muscular strengthening of
the affected limb. We believe that if this
yawning behavior could be stimulated by
medication or another mechanism, the plegic
performance of our patient might improve.
However, in our case, exploration of this
possibility was limited, and we could arrive at
no conclusion with regard to this issue. Further
studies will be carried out in the future.
Whether the persistence of CCD and PBO in this
patient corresponded to the poor recovery of
left hemiplegia is uncertain because no similar
case has been reported. The prognosis in
putaminal hemorrhage is related tomany factors
such as Glasgow Coma Scale score, pyramidal sign
to the nonhemiplegic side, midline shift, size
of hematoma, and cerebellar perfusion. We
speculate that the two phenomena CCD and PBO may
be valuable for predicting clinical recovery
after stroke when they occur together.
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Sobesky, A. Thiel, M. Ghaemi et al.,
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stroke: a PET study of serial changes and
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no. 12, pp. 1685&endash;1691, 2005.
[5] A.
C. Flint, M. C. Naley, and C. B. Wright,
"Ataxic hemiparesis from strategic frontal white
matter infarction with crossed cerebellar
diaschisis," Stroke, vol. 37, no. 1, pp.
e1&endash;e2, 2006.
[6] L.
Gold and M. Lauritzen, "Neuronal
deactivation explains decreased cerebellar blood
flow in response to focal cerebral ischemia or
suppressed neocortical function," Proceedings of
the NationalAcademy of Sciences of theUnited
States ofAmerica, vol. 99, no. 11, pp.
7699&endash;7704, 2002.
[7] P.
Pantano, G. L. Lenzi, and B. Guidetti,
"Crossed cerebellar diaschisis in patients with
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123I-HIPDM," European Neurology, vol. 27, no. 3,
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[10]
O.Walusinski, "Can stroke localisation be
used to map out the neural network for yawning
behaviour?" Journal of Neurology, Neurosurgery
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2007.
[11] R.
Töpper, M.Mull, and W.Nacimiento,
"Involuntary stretching during yawning in
patients with pyramidal tract lesions: further
evidence for the existence of an independent
emotional motor system," European Journal of
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