Letters to the Editor

FIGURE 1. Unenhanced admission axial computed tomography through the basal ganglia.

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Putaminal Hemorrhage Accompanied by
Hemichorea-Hemiballism
To the Editor:
Hemichorea-hemiballism is a relatively rare syndrome described
most often with lesions in the contralateral subthalamic nucleus
and, less frequently, at other sites.1 Hemichorea has also been
reported as a rare manifestation of nonketotic hyperglycemia.2 We
report a case of acute hemichorea produced by a small hemorrhage or hemorrhagic infarct in the contralateral putamen. This
case is noteworthy because hemorrhagic putaminal lesions infrequently produce hemichorea3 and because dyskinesia was associated with nonketotic hyperglycemia.
A 67-year-old right-handed male with insulin-dependent diabetes, hypertension, and past alcohol abuse noted the abrupt onset of
involuntary movements involving the left arm and leg 4 days prior
to admission. General examination was unremarkable with a blood
pressure of 120/70 mm Hg. There was mild left supranuclear facial
nerve weakness. Strength of extremities was normal. Constant
choreiform movements involving the distal left arm and leg were
seen with occasional ballistic components involving the proximal
musculature. These movements disappeared during sleep. Except
for mild symmetric distal loss of vibration, the neurologic examination was normal.
On admission, blood glucose was 610 mg/dl and calculated
serum osmolality was 315 milliosmoles/1. The hyperglycemia
resolved after administration of insulin and the remainder of the
laboratory evaluation was normal.
Initial computerized tomography without contrast (Figure 1)
showed slightly increased attenuation in the right putamen representing acute hemorrhage. Two weeks later, Tl-weighted coronal
magnetic resonance imaging (Figure 2) demonstrated increased
signal in the right putamen, representing methemoglobin.
The abnormal movements resolved over the next 2 weeks
following administration of 2 mg haloperidol three times a day and

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FIGURE 2. Tl-weighted coronal magnetic resonance imaging scan through the basal ganglia 3 weeks after the acute
event.
50 mg benadryl once a day. However, 3 weeks after initial presentation, this patient experienced abrupt recurrence of left hemichorea. Blood glucose was 597 mg/dl with no ketones in the serum.
The next morning blood glucose was 235 mg/dl, and choreiform
movements had resolved.
The majority of patients with hemichorea-hemiballism have
vascular lesions, due usually to ischemia rather than hemorrhage
or hemorrhagic infarct.4 Despite the high incidence of lacunar
infarcts and hypertensive intracerebral hemorrhage in the basal
ganglia, hemichorea-hemiballism is a rare presentation, probably
because accompanying weakness masks manifestation of the
abnormal movements.
Hemichorea-hemiballism has been described with lesions in the
subthalamic nucleus, striatum, cerebral cortex, thalamus, diencephalon, and brain stem.1 Based on a series of experiments in the
monkey, in which stereotaxic lesions were placed in the subthalamic nucleus, Carpenter et al5 concluded that development of
"choreoid-hyperkinesia" required an intact globus pallidus.
Reviewing human cases of hemichorea, Martin inferred that
choreic movements resulted when the globus pallidus was released
from the inhibitory influence of the subthalamic nucleus.6 Results
of an acute putaminal lesion suggest that the putamen similarly
inhibits the globus pallidus and that removal of this inhibition
leads to hemichorea,7 as demonstrated in the present case.
Interestingly, this patient had nonketotic hyperglycemia on both
occasions that he presented with hemichorea. Both ketoacidosis
and nonketotic hyperglycemia depress the Krebs cycle so that
brain energy requirements must be met by alternate metabolic
pathways such as increased rate of utilization of GABA to succinic
acid via the GABA shunt.8 Depletion of striatal GABA in nonketotic hyperglycemia, either as a result of increased metabolic

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Stroke Vol 21, No 7, July 1990

consumption or secondary to local ischemia, may allow increased
pallidal activity with resultant dyskinesia.
Irfan Altafullah, MD
Alvaro Pascual-Leone, MD
Kathy Duvall, MD
David C. Anderson, MD
Department of Neurology
Saul Taylor, MD
Department of Radiology
Hennepin County Medical Center
Minneapolis, Minnesota

References
1. Schwarz G, Barrows L: Hemiballism without involvement of
Luys body. Arch Neurol 1960;2:420-434

2. Rector W, Herlong F, Moses H: Nonketotic hyperglycemia
appearing as choreoathetosis or ballism. Arch Intern Med 1982;
142:154-155
3. Jones R Jr, Baker R, Kott S: Hypertensive putaminal hemorrhage presenting with hemichorea. Stroke 1985;16:130-131
4. Dewey R, Jankovic J Jr: Hemiballism-hemichorea: Clinical and
pharmacologic findings in 21 patients. Arch Neurol 1989;46:
862-867
5. Carpenter M, Whittier J, Mettler F: Analysis of choreoid
hyperkinesia in the rhesus monkey; surgical and pharmacological analyses of hyperkinesia resulting from lesions in the
subthalamic nucleus of Luys. / Comp Neurol 1950;92:293-331
6. Martin J: Hemichorea (hemiballismus) without lesions of the
corpus Luysii. Brain 1957;80:l-10
7. Crossman A, Sambrook M, Jackson A: Experimental hemichorea/hemiballismus in the monkey. Brain 1984;107:579-596
8. Guisado R, Arieff A: Neurologic manifestations of diabetic
comas: Correlation with biochemical alterations in the brain.
Metabolism 1975;24:655-679

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