Neurol Sci DOI 10.1007/s10072-016-2486-0
BRIEF COMMUNICATION
Asterixis in the leg induced by anterior cerebral artery infarction Mun Kyung Sunwoo1 • Hyun-soon Jang1 • Sook Young Roh1 • Hyun Jung Yoo1 Eun Hye Jeong1 • Byung-Su Kim1 • Yeo Reum Choe2 • Ko-Eun Lee2
•
Received: 23 September 2015 / Accepted: 13 January 2016 Ó Springer-Verlag Italia 2016
Abstract Asterixis commonly occurs in a patient with metabolic encephalopathy, whereas focal brain lesions such as thalamus, cerebellum, or frontal area also cause focal or unilateral asterixis in the arms. We report a novel case of asterixis in the leg after unilateral anterior cerebral artery territory infarction. A 76-year-old man was admitted with sudden-onset mild right leg weakness and postural instability due to knee buckling. He was diagnosed with ischemic stroke in the left prefrontal area and cingulated gyrus by brain magnetic imaging. Needle electromyography of the right vastus lateralis muscle while standing showed intermittent periods of EMG silence, consistent with asterixis. There were no abnormal involuntary movements in the upper extremities. This case suggests that gait disturbance or postural instability after structural lesions in the prefrontal area may be directly related to asterixis in the leg, not in the arm associated with postural failure. Keywords Movement disorder Asterixis Anterior cerebral artery territory infarction Prefrontal
Electronic supplementary material The online version of this article (doi:10.1007/s10072-016-2486-0) contains supplementary material, which is available to authorized users. & Ko-Eun Lee [email protected] 1
Department of Neurology, Bundang Jesaeng General Hospital, Seongnam, South Korea
2
Department of Rehabilitation, Bundang Jesaeng General Hospital, 20, Seohyun-ro 180, Bundang gu, Seongnam, Kyunggo-do 463-774, South Korea
Introduction Hyperkinetic involuntary movements, such as tremor, chorea, dystonia, and asterixis, may follow ischemic stroke involving the basal ganglia or thalamus/subthalamus [1]. Anterior cerebral artery (ACA) territory infarction can also cause hemi-parkinsonism and asterixis in the arms, but never in the legs [2]. Here, we report a case of asterixis in the leg after unilateral ACA territory infarction involving a prefrontal lesion.
Case report A 76-year-old man who had suffered from hypertension and non-insulin-dependent diabetes mellitus for 15 years was admitted with sudden onset right leg weakness. He denied any history of involuntary movement, preceding infection or family history. On admission, his blood pressure was 148/79 mmHg and pulse was 79 and regular. The neurologic examination showed mild right leg monoparesis (IV/V, on the 0–V Medical Research Council scale), but no weakness in the upper extremities. He reported subtle hypesthesia in the right leg that recovered fully the next day. He was alert and had normal mental function except attention. The cranial nerve examination, cerebellar function tests and deep tendon reflexes were normal. Laboratory data including liver, and renal function tests and ammonia were unremarkable, except for a high fasting serum glucose level (166 mg/dL). Brain magnetic imaging revealed acute ischemic stroke in the left prefrontal area and cingulate gyrus. There were no abnormal lesions in the bilateral thalamus, basal ganglia or cerebellum (Fig. 1a). Cerebral angiography showed atherosclerotic steno-occlusion at the left anterior cerebral
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Neurol Sci
Fig. 1 a Diffusion weighted imaging revealed acute ischemic stroke in the left prefrontal area and cingulate gyrus. b Needle electromyography (EMG) of right vastus lateralis muscle while standing showed intermittent periods of EMG silence
artery related to the ischemic lesions. Three days after admission, the patient reported near falls and insecurity while walking because his right knee would buckle. Sudden, very brief flexion of the right knee occurred every few steps after physical leg exercise over a few minutes (Video 1) The patient had no tremor, myoclonus, or choreic movement on sitting or in the supine position. On stretching his arms, there was no abnormal movement or posture in either hand. He never showed parkinsonian symptoms including resting tremor, bradykinesia and rigidity. A follow-up diffusion weighted scan did not show new lesions. Electroencephalography showed no epileptiform discharges and nerve conduction study also had no evidence of neuropathy and radiculopathy in the legs. Needle electromyography (EMG) of right vastus lateralis muscle while standing showed intermittent periods of EMG silence, consistent with negative myoclonus (Video 2, Fig. 1b). Although the patient was treated with clonazepam and antiepileptic drugs, the symptom persisted for 5 months ever since he had a stroke.
Discussion Acute or delayed involuntary movement disorders develop after 1–4 % of strokes [3]. Post-stroke movement disorders manifest in parkinsonism or hyperkinetic movement
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disorders, such as tremor, dystonia, chorea and myoclonus [3, 4]. Asterixis, referred to as negative myoclonus, is characterized by intermittent interruption of sustained muscle contraction caused by the sudden cessation of electrical activity [5]. Asterixis commonly involves bilateral limb muscles in a patient with metabolic encephalopathy, whereas focal or unilateral asterixis has been reported with focal brain lesions, such as in the thalamus, cerebellum, or frontal area [2, 6–8]. Thalamic lesions, especially involving the ventrolateral thalamus, are the lesions most commonly causing unilateral asterixis [7]. One study reported 30 patients with unilateral stroke who developed asterixis [7]. Lesions inducing unilateral asterixis have been identified not only in the contralateral thalamus, but also in the frontal lobe, midbrain, or cerebellum. However, the presence of asterixis was examined using wrists dorsiflexion, and confined to the upper extremities. There was no examination of asterixis in the legs. The author suggested that frequent gait instability in patients with asterixis reflects a failure in arm posture maintenance that relates to leg posture instability. Another study demonstrated that anterior cerebral artery territory infarction caused unilateral or bilateral asterixis in three of nine cases with involuntary movement after stroke [2]. In these three cases, the author mentioned that asterixis occurred in the arms with minimal weakness, but never occurred in the legs. Different from the previous reports, we present a patient who developed asterixis in the legs, not in the upper extremities. Gait disturbance or postural instability was due to brief lapse in the right leg posture after ischemic stroke in the premotor area. The pathophysiology of asterixis caused by structural lesions remains elusive. Thalamic asterixis might be mediated by excessive inhibition of the sensorimotor cortex as a result of a structural lesion in the ventral lateral nucleus [9]. Sustained muscle contraction is related to basal ganglia-thalamocortical circuits involving the medial frontal cortex, parietal lobe, and ventrolateral thalamus [5]. Asterixis, failure of consistent muscle tone, may develop with disruption of this neuronal system in patients with lesions mainly involving the prefrontal cortex [2, 6, 7]. In conclusion, we report a novel case with postural instability caused by leg asterixis after an ACA territory infarction. This case suggest that gait disturbance or postural instability after structural lesions in the thalamus or prefrontal area may be related to abnormal movement disorders in the leg, and not to arm asterixis. Compliance with ethical standards Conflict of interest
Nothing to report.
Neurol Sci
References 1. Ghika-Schmid F, Ghika J, Regli F et al (1997) Hyperkinetic movement disorders during and after acute stroke: the Lausanne Stroke Registry. J Neurol Sci 146(2):109–116 2. Kim JS (2001) Involuntary movements after anterior cerebral artery territory infarction. Stroke 32(1):258–261 3. Mehanna R, Jankovic J (2013) Movement disorders in cerebrovascular disease. Lancet Neurol 12(6):597–608 4. Bansil S, Prakash N, Kaye J et al (2012) Movement disorders after stroke in adults: a review. Tremor Other Hyperkinet Mov (N Y) 2 5. Young RR, Shahani BT (1986) Asterixis: one type of negative myoclonus. Adv Neurol 43:137–156
6. Matsumoto H, Hashida H, Ugawa Y (2014) Unilateral asterixis caused by an internal capsule lesion. Intern Med 53(4):341–342 7. Kim JS (2001) Asterixis after unilateral stroke: lesion location of 30 patients. Neurology 56(4):533–536 8. Siniscalchi A, Gallelli L, Di Benedetto O et al (2012) Asterixis as a presentation of cerebellar ischemic stroke. West J Emerg Med 13(6):507–508 9. Inoue M, Kojima Y, Mima T et al (2012) Pathophysiology of unilateral asterixis due to thalamic lesion. Clin Neurophysiol 123(9):1858–1864
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BRIEF COMMUNICATION
Asterixis in the leg induced by anterior cerebral artery infarction Mun Kyung Sunwoo1 • Hyun-soon Jang1 • Sook Young Roh1 • Hyun Jung Yoo1 Eun Hye Jeong1 • Byung-Su Kim1 • Yeo Reum Choe2 • Ko-Eun Lee2
•
Received: 23 September 2015 / Accepted: 13 January 2016 Ó Springer-Verlag Italia 2016
Abstract Asterixis commonly occurs in a patient with metabolic encephalopathy, whereas focal brain lesions such as thalamus, cerebellum, or frontal area also cause focal or unilateral asterixis in the arms. We report a novel case of asterixis in the leg after unilateral anterior cerebral artery territory infarction. A 76-year-old man was admitted with sudden-onset mild right leg weakness and postural instability due to knee buckling. He was diagnosed with ischemic stroke in the left prefrontal area and cingulated gyrus by brain magnetic imaging. Needle electromyography of the right vastus lateralis muscle while standing showed intermittent periods of EMG silence, consistent with asterixis. There were no abnormal involuntary movements in the upper extremities. This case suggests that gait disturbance or postural instability after structural lesions in the prefrontal area may be directly related to asterixis in the leg, not in the arm associated with postural failure. Keywords Movement disorder Asterixis Anterior cerebral artery territory infarction Prefrontal
Electronic supplementary material The online version of this article (doi:10.1007/s10072-016-2486-0) contains supplementary material, which is available to authorized users. & Ko-Eun Lee [email protected] 1
Department of Neurology, Bundang Jesaeng General Hospital, Seongnam, South Korea
2
Department of Rehabilitation, Bundang Jesaeng General Hospital, 20, Seohyun-ro 180, Bundang gu, Seongnam, Kyunggo-do 463-774, South Korea
Introduction Hyperkinetic involuntary movements, such as tremor, chorea, dystonia, and asterixis, may follow ischemic stroke involving the basal ganglia or thalamus/subthalamus [1]. Anterior cerebral artery (ACA) territory infarction can also cause hemi-parkinsonism and asterixis in the arms, but never in the legs [2]. Here, we report a case of asterixis in the leg after unilateral ACA territory infarction involving a prefrontal lesion.
Case report A 76-year-old man who had suffered from hypertension and non-insulin-dependent diabetes mellitus for 15 years was admitted with sudden onset right leg weakness. He denied any history of involuntary movement, preceding infection or family history. On admission, his blood pressure was 148/79 mmHg and pulse was 79 and regular. The neurologic examination showed mild right leg monoparesis (IV/V, on the 0–V Medical Research Council scale), but no weakness in the upper extremities. He reported subtle hypesthesia in the right leg that recovered fully the next day. He was alert and had normal mental function except attention. The cranial nerve examination, cerebellar function tests and deep tendon reflexes were normal. Laboratory data including liver, and renal function tests and ammonia were unremarkable, except for a high fasting serum glucose level (166 mg/dL). Brain magnetic imaging revealed acute ischemic stroke in the left prefrontal area and cingulate gyrus. There were no abnormal lesions in the bilateral thalamus, basal ganglia or cerebellum (Fig. 1a). Cerebral angiography showed atherosclerotic steno-occlusion at the left anterior cerebral
123
Neurol Sci
Fig. 1 a Diffusion weighted imaging revealed acute ischemic stroke in the left prefrontal area and cingulate gyrus. b Needle electromyography (EMG) of right vastus lateralis muscle while standing showed intermittent periods of EMG silence
artery related to the ischemic lesions. Three days after admission, the patient reported near falls and insecurity while walking because his right knee would buckle. Sudden, very brief flexion of the right knee occurred every few steps after physical leg exercise over a few minutes (Video 1) The patient had no tremor, myoclonus, or choreic movement on sitting or in the supine position. On stretching his arms, there was no abnormal movement or posture in either hand. He never showed parkinsonian symptoms including resting tremor, bradykinesia and rigidity. A follow-up diffusion weighted scan did not show new lesions. Electroencephalography showed no epileptiform discharges and nerve conduction study also had no evidence of neuropathy and radiculopathy in the legs. Needle electromyography (EMG) of right vastus lateralis muscle while standing showed intermittent periods of EMG silence, consistent with negative myoclonus (Video 2, Fig. 1b). Although the patient was treated with clonazepam and antiepileptic drugs, the symptom persisted for 5 months ever since he had a stroke.
Discussion Acute or delayed involuntary movement disorders develop after 1–4 % of strokes [3]. Post-stroke movement disorders manifest in parkinsonism or hyperkinetic movement
123
disorders, such as tremor, dystonia, chorea and myoclonus [3, 4]. Asterixis, referred to as negative myoclonus, is characterized by intermittent interruption of sustained muscle contraction caused by the sudden cessation of electrical activity [5]. Asterixis commonly involves bilateral limb muscles in a patient with metabolic encephalopathy, whereas focal or unilateral asterixis has been reported with focal brain lesions, such as in the thalamus, cerebellum, or frontal area [2, 6–8]. Thalamic lesions, especially involving the ventrolateral thalamus, are the lesions most commonly causing unilateral asterixis [7]. One study reported 30 patients with unilateral stroke who developed asterixis [7]. Lesions inducing unilateral asterixis have been identified not only in the contralateral thalamus, but also in the frontal lobe, midbrain, or cerebellum. However, the presence of asterixis was examined using wrists dorsiflexion, and confined to the upper extremities. There was no examination of asterixis in the legs. The author suggested that frequent gait instability in patients with asterixis reflects a failure in arm posture maintenance that relates to leg posture instability. Another study demonstrated that anterior cerebral artery territory infarction caused unilateral or bilateral asterixis in three of nine cases with involuntary movement after stroke [2]. In these three cases, the author mentioned that asterixis occurred in the arms with minimal weakness, but never occurred in the legs. Different from the previous reports, we present a patient who developed asterixis in the legs, not in the upper extremities. Gait disturbance or postural instability was due to brief lapse in the right leg posture after ischemic stroke in the premotor area. The pathophysiology of asterixis caused by structural lesions remains elusive. Thalamic asterixis might be mediated by excessive inhibition of the sensorimotor cortex as a result of a structural lesion in the ventral lateral nucleus [9]. Sustained muscle contraction is related to basal ganglia-thalamocortical circuits involving the medial frontal cortex, parietal lobe, and ventrolateral thalamus [5]. Asterixis, failure of consistent muscle tone, may develop with disruption of this neuronal system in patients with lesions mainly involving the prefrontal cortex [2, 6, 7]. In conclusion, we report a novel case with postural instability caused by leg asterixis after an ACA territory infarction. This case suggest that gait disturbance or postural instability after structural lesions in the thalamus or prefrontal area may be related to abnormal movement disorders in the leg, and not to arm asterixis. Compliance with ethical standards Conflict of interest
Nothing to report.
Neurol Sci
References 1. Ghika-Schmid F, Ghika J, Regli F et al (1997) Hyperkinetic movement disorders during and after acute stroke: the Lausanne Stroke Registry. J Neurol Sci 146(2):109–116 2. Kim JS (2001) Involuntary movements after anterior cerebral artery territory infarction. Stroke 32(1):258–261 3. Mehanna R, Jankovic J (2013) Movement disorders in cerebrovascular disease. Lancet Neurol 12(6):597–608 4. Bansil S, Prakash N, Kaye J et al (2012) Movement disorders after stroke in adults: a review. Tremor Other Hyperkinet Mov (N Y) 2 5. Young RR, Shahani BT (1986) Asterixis: one type of negative myoclonus. Adv Neurol 43:137–156
6. Matsumoto H, Hashida H, Ugawa Y (2014) Unilateral asterixis caused by an internal capsule lesion. Intern Med 53(4):341–342 7. Kim JS (2001) Asterixis after unilateral stroke: lesion location of 30 patients. Neurology 56(4):533–536 8. Siniscalchi A, Gallelli L, Di Benedetto O et al (2012) Asterixis as a presentation of cerebellar ischemic stroke. West J Emerg Med 13(6):507–508 9. Inoue M, Kojima Y, Mima T et al (2012) Pathophysiology of unilateral asterixis due to thalamic lesion. Clin Neurophysiol 123(9):1858–1864
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