J Neurol (1993) 241:27-30
© Springer-Verlag 1993

Luigi Mossuto-Agatiello
G i o v a n n i Puccetti
A n n a Elisa C a s t e l l a n o

Received: 12 May 1992
Received in revised form: 1 March 1993
Accepted: 15 March 1993

L. Mossuto-Agatiello ( ~ ) - G. Puccetti
S. Giovanni Battista Hospital,
V. Luigi Dasti 7, 1-00148 Rome, Italy
A. E. Castellano
Sanatrix Institute, Pozzilli (IS), Italy

"Rubrar' tremor
after thalamic haemorrhage

A b s t r a c t Tremors in association
with t h a l a m i c h a e m o r r h a g e h a v e
been infrequently reported, and del a y e d rubral t r e m o r as a c o m p l i c a t i o n
o f such an event is quite rare. W e describe a patient with a c o m b i n e d
r e s t i n g - p o s t u r a l - k i n e t i c t r e m o r due a
t h a l a m i c h a e m o r r h a g e . M a g n e t i c resonance imaging showed evidence of
a s u b t h a l a m i c i n v o l v e m e n t but failed
to reveal any m e s e n c e p h a l i c lesion.
F i v e years after the original stroke

Introduction
" R u b r a l " t r e m o r is an unusual type o f t r e m o r w h i c h includes a resting, postural and kinetic c o m p o n e n t . C h a r a c teristically p r e s e n t at rest, it b e c o m e s m o r e p r o n o u n c e d
during m a i n t e n a n c e o f a fixed posture and further increases in a m p l i t u d e with intentional v o l u n t a r y m o v e ments.
M o s t patients with rubral t r e m o r have h a d clinical,
p a t h o l o g i c a l or r a d i o g r a p h i c e v i d e n c e o f lesions in the upper brain stem, and causes for the sporadic cases r e p o r t e d
in the literature include t u m o u r s [8], v a s c u l a r insults [3, 5,
8], head t r a u m a [1, 13], m u l t i p l e sclerosis [4], arteriovenous m a l f o r m a t i o n [6] and t o x o p l a s m a abscess [9].
T r e m o r s in a s s o c i a t i o n w i t h t h a l a m i c h a e m o r r h a g e
h a v e b e e n i n f r e q u e n t l y d e s c r i b e d [10, 14], a n d r u b r a l
t r e m o r as a late c o m p l i c a t i o n o f such an event has not
b e e n r e p o r t e d previously. Therefore, we r e p o r t a patient
with r a d i o g r a p h i c e v i d e n c e o f t h a l a m i c - s u b t h a l a m i c haem o r r h a g e w h o s u b s e q u e n t l y d e v e l o p e d a c o m b i n e d rest-

there was rapid and a l m o s t c o m p l e t e
suppression o f her a b n o r m a l m o v e ments, p r o b a b l y related to an ischa e m i c c a p s u l a r lesion. I n v o l u n t a r y
m o v e m e n t s , w h i c h r e s e m b l e rubral
tremor, can be due to lesions upstream o f the rubral and nigral outflow system.
K e y w o r d s M o v e m e n t disorders
Rubral t r e m o r • T h a l a m i c
haemorrhage

i n g - p o s t u r a l - k i n e t i c t r e m o r in the u p p e r e x t r e m i t y contralateral to the side o f the lesion. M a g n e t i c r e s o n a n c e
i m a g i n g ( M R I ) failed to r e v e a l any m e s e n c e p h a l i c d a m age. The patient h a d a m a r k e d d e c r e a s e o f her a b n o r m a l
m o v e m e n t s after a later i s c h a e m i c v a s c u l a r insult.

Case report
This 53-year-old, right-handed, previously hypertensive woman
was in good health until April 1986 when she suffered the acute
onset of right hemiparesis and was admitted to another hospital.
Her speech was dysarthric and nonfluent. She remained obtunded
and confused for about 3 days. Other details of the neurological
examination at that time are not available. Over the following
weeks speech disturbance and hemiparesis subsided, and when she
was discharged from the hospital she was able to walk and talk.
One month after the acute episode the patient developed involuntary movements of the right arm which worsened over the next few
months until she was admitted to our institute in October 1986.
On admission she was alert, oriented, and had normal speech.
Neurological examination revealed no motor weakness or sensory
deficit. Gait was normal and there were no signs of cerebellar dysfunction. Deep tendon reflexes were increased on the right and the
plantar response was flexor bilaterally. The pupils were slightly

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Fig. 1 Handwriting of patient, attempting to sign her name
anisocoric and upgaze was limited. There was a resting tremor in
the right upper extremity with a frequency of 2.5-3.0 Hz involving
flexion and extension of the fingers, hand, and forearm, as well as
pronation and supination of the forearm. The tremor was also present with posture and became pronounced during intentional voluntary movements. The hand tremor caused marked functional interference with the activities of daily living, especially writing
(Fig. 1). A small amplitude of resting tremor was also observed in
her right foot which was intermittent and not present during walking. Palatal myoclonus, myoclonic jerking and dystonic postures

were not observed. Cranial computed tomography (CT) showed an
irregular area of decreased density in the left posterior thalamus
that was assumed to represent an old haemorrhagic lesion. MRI
confirmed that the patient had had a thalamic haemorrhage with
caudal extension into the subthalamic area (Fig. 2 a, b). There was
no MRI evidence of mesencephalic damage (Fig. 3 a). Neither CT
nor MRI showed any lesion of the posterior limb of the internal
capsule (Fig. 2 c).
In May 1991, i.e. approximately 5 years after the onset of the
abnormal movements, her family noticed the sudden disappearance of the tremor. On neurological examination, 10 days later,
there was a mild right hemiparesis, more severe in the arm than in
the leg, with hyperactive reflexes and extensor plantar response.
Exteroceptive somatic sensation was preserved with right tactile
extinction on double simultaneous stimulation. Position sense and
graphaesthesia were impaired in the right hand. There was minimal tremor at rest in the right upper limb, and only right dysmetria
with the finger-to-nose manoeuvre. However, a mild intentional
tremor of the right hand was observed when the patient attempted
to write. When she walked, she carried her right arm flexed at the
elbow and showed an intermittent moderate amplitude tremor (4-5
Hz). A new MRI study disclosed, in addition to the old thalamosubthalamic lesion, multiple patchy subcortical areas of increased
signal intensity localized in the periventricular white matter (present in part at the first MRI study), left cerebellar hemisphere, and
left internal capsule (Fig. 4). Again MRI confirmed that the red nucleus and the areas around it were not involved (Fig. 3 b).

Discussion
T h e p a t i e n t e x h i b i t e d t h e late a p p e a r a n c e o f u n i l a t e r a l
t r e m u l o u s m o v e m e n t s as the m a j o r m a n i f e s t a t i o n o f c o n tralateral p o s t e r i o r t h a l a m i c h a e m o r r h a g e c o n f i r m e d b y
M R I . T h e r e w a s g o o d r e c o v e r y o f s p e e c h and m o t o r f u n c tion, and w h e n she w a s a d m i t t e d to o u r h o s p i t a l no sen-

Fig. 2 a - e MRI: a Axial section through the inferior thalamus and
b coronal section through the posterior thalamus (TR = 1600, TE =
30, 100 ms respectively) illustrating a lesion in the left thalamus and
subthalamus. The a r r o w s point to an area of pronounced signal
void consistent with haemosiderin deposition from an old haemorrhage, e A more rostral axial section T2-weighted image shows
that the posterior part of the left internal capsule is unaffected

sory i m p a i r m e n t o r c e r e b e l l a r d y s f u n c t i o n w e r e o b s e r v e d .
O u r p a t i e n t ' s t r e m o r h a d m a n y f e a t u r e s o f w h a t has
b e e n d e s c r i b e d in the l i t e r a t u r e as m i d b r a i n

or rubral

t r e m o r [7]. T h e r e is n o c o m p l e t e a g r e e m e n t r e g a r d i n g the
d e f i n i t i o n o f r u b r a l t r e m o r - w h e t h e r this d i s o r d e r is a

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Fig.3a, b Transaxial MR images through the upper mesencephalon (TR 1600, TE 30 ms; TR 2000, TE 110 ms respectively)
showing that the area around the red nucleus is intact
Fig. 4 T2-weighted axial MR image (TR/TE = 2000/t 10 ms)
demonstrates areas of high signal intensity in the left thalamus and
left posterior limb of the internal capsule. The more intense high
signal abnormality from the capsular area (arrowhead) is consistent with recent infarction
single tremor or a combination of two distinct tremors.
Some authors believe that a rubral tremor is actually a
cerebellar tremor associated with a parkinsonian tremor.
The kinetic tremor component is thought to be related to
lesions involving any part of the dentato-rubro-thalamic
pathway, whereas the rest component requires involvement of the ascending nigrostriatal pathway [5]. Midbrain
tremor is commonly referred to as rubral tremor because
of the presumed involvement of the red nucleus. However, lesions of this nucleus are not essential for the development of the tremor [ 11], whereas lesions in the environs of the red nucleus may lead to a rubral tremor
whether or not the nucleus is damaged. It appears that the
tegmental area around the nucleus is particularly important in the genesis of the tremor [13].
The radiographic data from our patient failed to reveal
any mesencephalic lesion. CT and MRI showed evidence
of an old haemorrhagic lesion restricted to the posterior
thalamus and rostral subthalamus. Therefore, the present
case shows that rubral tremor is not exclusively due to
selective damage of structures located in the upper brain
stem, and that a well-placed lesion upstream of the rubral

and nigral outflow can cause a tremor of similar type. In
spite of the lack of pathological confirmation, we believe
that the involvement of the subthalamic area was critical
in the genesis of the tremor, because probably at this level
both projection pathways from the dentate to the ventrolateral thalamic nucleus and the dopaminergic nigrostriatal pathway were interrupted.
Tremor is abolished by surgical intervention on the
motor cortex [2], by pyramidotomy at the lower brain
stem [15] or after cerebral infarct producing a hemiplegia
[12]. These observations indicate that tremor is driven by
discharge in the motor cortex transmitted via neuronal
pathways traversing the internal capsule.
Five years after the original stroke, this patient suddenly became hemiparetic again and had almost complete
suppression of her abnormal movements. A new MRI
study revealed multiple patchy white-matter foci of increased signal intensity; most of these had been seen in
the first MRI. The small bilateral lesions involving the
white matter adjacent to the lateral ventricles could be regarded as incidental subcortical lesions associated with
age and hypertension, and cannot account for the events
described, Conversely, the hyperintense area identified in
the posterior limb of the internal capsule probably represents an ischaemic lesion and may explain the recent sensorimotor deficit, as well the disappearance of the tremor,
since the involvement of the pyramidal tract presumably
caused interruption of the tremor impulses in their downward transmission.

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