Published ahead of print on January 9, 2008
doi: 10.3174/ajnr.A0927
American Journal of Neuroradiology 29:526-527, March 2008
© 2008 American Society of Neuroradiology
Case Report
BRAIN
Subthalamic Lesion on MR Imaging in a Patient with Nonketotic Hyperglycemia-Induced Hemiballism
H.-J. Kima,c,
W.J. Moonb,
J. Oha,
I.K. Leea,
H.Y. Kima,c and
S.-H. Hana,c
a Department of Neurology, Konkuk University Hospital, Konkuk University School of Medicine, Seoul, Korea
b Department of Radiology, Konkuk University Hospital, Konkuk University School of Medicine, Seoul, Korea
c Center for Geriatric Neuroscience Research, Institute of Biomedical Science, Konkuk University, Seoul, Korea
Please address correspondence to Seol-Heui Han, MD, Department of Neurology, Konkuk University Hospital, Konkuk University School of Medicine, 4–12 Hwayanag-dong, Gwangngjin-gu 143–729, Seoul, Korea: e-mail: alzdoc{at}kuh.ac.kr
SUMMARY: Hemiballism with corresponding striatal T1 hyperintensity
on MR imaging has occasionally been reported in patients with
nonketotic hyperglycemia. However, the subthalamic nucleus lesion,
which is believed to be pathogenetically related to hemiballism,
is rarely documented in a living patient with nonketotic hyperglycemia.
We describe a patient with nonketotic hyperglycemia-induced
hemiballism, whose responsible lesion (ie, the subthalamus)
was demonstrated by MR imaging.
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Introduction
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Hemiballism-hemichorea (HB-HC) is an involuntary, irregular,
wide-amplitude, and poorly patterned movement disorder. HB-HC
can mostly result from a focal vascular lesion in the contralateral
basal ganglia.
1 In the absence of an identifiable focal vascular
lesion, metabolic derangements (eg, nonketotic hyperglycemia
or hyperthyroidism), brain neoplasm, and infectious diseases
of the central nervous system (eg, human immunodeficiency virus
infection) are considered to be the more common causes.
1 Recent
studies have shown that striatal hyperintensity, seen on the
T1-weighted MR imaging of the brain, can occur in the acute
stage of HB-HC, caused by nonketotic hyperglycemia.
2 These radiologic
images provided the clue for the early diagnosis of this dyskinesia.
Animal studies and subsequent human studies supported the finding
that the subthalamus is strongly related to hemiballism.
3 There
are only scanty reports documenting the subthalamic involvement
of nonketotic hyperglycemia in a living patient, which prompted
us to report a case of nonketotic hyperglycemia-induced hemiballism,
with the responsible subthalamic lesion confirmed by MR imaging.
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Case Report
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A 53-year-old woman was admitted for sudden onset of abnormal
movement on the left side. This jerky irregular involuntary
movement was first noted on the left leg 3 days before admission.
Her symptoms worsened during the last 3 days. She had no history
of hypertension, diabetes, headache, parkinsonism, or other
neurologic diseases. At admission, her initial blood glucose
level was 242 mg/dL and her hemoglobin A
1c concentration was
11.7% (normal values are 70–110 mg/dL and 4.7–6.4%,
respectively). The urine specimen was negative for ketones,
indicating nonketotic hyperglycemia. A neurologic examination
showed her sensorium to be normal. Deep tendon reflexes were
symmetrically hypoactive. The remaining results of the examinations
were unremarkable, except for left hemiballism. Brain CT, performed
on the first hospital day, revealed no abnormalities. The MR
image, obtained 4 days after her initial presentation, showed
high signal intensities in the right subthalamic nucleus (STN)
on T1-weighted and T2-weighted images (
Fig 1) and subtle high
signal intensities on diffusion-weighted images (DWIs, not shown).
Gradient-echo images and B
0 images of DWI showed no susceptibility
artifact in the right STN. Contrast-enhanced MR images showed
no additional findings. The caudate and putamen appeared normal
in all sequences of MR images. Hemiballism was improved with
clonazepam (6 mg/day) after 14 days. The serum glucose level
was normalized with insulin administration.
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Fig 1. The T1-weighted axial image (A) shows increased signal intensity (broken arrow), whereas the T2-weighted coronal image clearly demonstrates a high-signal-intensity lesion (arrow) in the right STN.
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Discussion
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Hyperglycemia-associated chorea and ballismus have been reported
predominantly in older patients with diabetes and nonketotic
hyperglycemia.
4 The T1-weighted MR images have shown unilateral
hyperintensity in the putamen and caudate, but the findings
on the T2-weighted MR images have varied.
5 Although a significant
association between hyperglycemia and hyperintensity on T1-weighted
MR images has been reported, the mechanism underlying the imaging
findings has been unknown. Protein dissociation in wallerian
degeneration secondary to acute putaminal dysfunction has been
proposed as one of the plausible mechanisms behind the imaging
findings.
6 Increased viscosity, the presence of myelin-breakdown
products, the presence of blood products, or deposition of calcium
and other minerals have been suggested as possible explanations
as well.
7–9
As to the MR signal intensities alone, our imaging findings confirmed results from previous reports. In this case, however, MR imaging revealed the sole involvement of the contralateral STN without a putaminal lesion. Although subthalamic involvement has never been reported in hyperglycemia-induced hemiballism, acute hemiballism was reported in a small cavernous angioma or infarction confined to the STN.10,11
The STN serves as the main excitatory input to the medial part of the globus pallidus, which inhibits, by using gamma amino butyric acid, the activity of the ventromedial thalamus. The destruction of the STN causes a decrease of this inhibitory function and increases thalamic excitation of the motor and premotor cortex, resulting in involuntary movement.12 This mechanism is supported by findings during deep brain stimulation of the subthalamus in Parkinson disease.13
Possible differential diagnosis in this case includes acute infarction by vascular insult. Unlike T1 hyperintensity due to nonketotic hyperglycemia, acute infarction of 3 days duration shows bright signal intensity on DWIs and low signal intensity on T1-weighted images. When T1 hyperintensity is noted in acute infarction at 3 days, it should be hemorrhagic transformation and should, therefore, show dark signal intensity on T2*-weighted images.14 Acute hemorrhage is also unlikely because there is no evidence of magnetic susceptibility artifacts on T2*-weighted images as well as on DWIs with a b-value of 0 s/mm2.
In conclusion, unilateral subthalamic involvement can be the sole imaging finding in nonketotic hyperglycemia-induced hemiballism. This case suggests a pathophysiologic explanation for hemiballism with its strategic localization.
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Footnotes
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This work was supported by the Second-Phase of BK (Brain Korea)
21 Project in 2007.
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References
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Received October 23, 2007;
accepted after revision October 30, 2007.
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Introduction