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Letters to Editor
Unilateral spatial neglect as a presenting manifestation of nonconvulsive status epilepticus Sir, Nonconvulsive status epilepticus (NCSE) is defined as a change in the mental status of at least 30 to 60 minutes duration, associated with continuous or near continuous ictal discharges detectable on an electroencephalography (EEG).[1] A diagnosis of NCSE requires the clinical manifestation of an abnormal mental status with reduced responsiveness, a supportive EEG, and a response to an antiepileptic drug. Unilateral spatial neglect (USN) as a manifestation of NCSE has not been reported till date. Here, we report the case of an elderly gentleman who presented with left spatial neglect with his EEG showing continuous, sharp wave discharges from the right parietooccipital region suggestive of the background presence of a NCSE. A 64‑year‑old, right‑handed man was brought in an altered mental status of a day’s duration. His wife found him to be in an acute confusional state on being woken up. He was talking irrelevantly and was disoriented. He was always looking towards his right side and was not moving his left upper and lower limb spontaneously and to any painful stimulus. On admission, he was drowsy but easily arousable on verbal commands. A command to shift his gaze towards his left side would
a
NCSE constitutes approximately 20–23% of cases presenting as status epilepticus. The clinical presentation of a NCSE
c
b
e
make him look towards the right side. There was intermittent horizontal, jerky nystagmus of both eyes towards the left side with fast component also towards the left. Several requests to lift his left upper and lower limbs always resulted in his lifting his right upper and lower limb. If the examiner called his name from his left hemispace, he would respond by looking towards his right hemispace [Video 1 (www.neurologyindia.com)]. There was no facial palsy. He had a normal tone, power and reflexes in his limbs with flexor plantars. His magnetic resonance imaging (MRI) of the brain showed diffusion restriction in the right parieto‑temporo‑occipital region with a hyperintense signal change in the right pulvinar region of the thalamus on fluid‑attenuated inversion recovery (FLAIR) images [Figure 1]. The MR angiography was normal. His EEG showed sharp wave discharges from the right temporo‑parieto‑occipital region with generalization, which disappeared on intravenous lorazepam (4mg) administration suggesting the presence of a NCSE [Figure 2]. His cerebrospinal fluid (CSF) analysis was normal and the CSF polymerase chain reaction (PCR) for herpes simplex virus was negative. A diagnosis of left USN as a manifestation of NCSE was made. He was treated with a loading dose of intravenous phenytoin (20 mg/kg) followed by continuance of a maintenance dose (5 mg/kg). A repeat EEG performed after 48 h revealed the disappearance of the epileptiform discharges. There was clinical improvement in the form of disappearance of the spatial neglect with return to normal sensorium [Video 2 (www.neurologyindia.com)].
f
d
g
Figure 1: (a-d) Magnetic resonance imaging (MRI) brain diffusion-weighted MRI (DWI) images axial view showing gyral hyperintensities in the right parieto-temporo-occipital region and right pulvinar of thalamus (white arrow); (e-g) MRI brain apparent-diffusion coefficient (ADC) showing corresponding hypointensities indicating diffusion restriction (white arrow) 262
Neurology India / March 2015 / Volume 63 / Issue 2
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Letters to Editor
Figure 2: Electroencephalogram (EEG) (bipolar longitudinal montage, sensitivity 14.0 μV/mm, low frequency filter 1 Hz, high frequency filter 70 Hz, notch 50 Hz, and speed 30 mm/s) showing sharp wave discharges from the right temporo-parieto-occipital region (red arrow) and disappearance of discharges with intravenous lorazepam
may vary from manifesting as a subtle abnormality to the presence of one or more of the following: Variation in the level of consciousness, fluctuation in behavior, semi‑purposeful movements, changes in or even a loss of speech, or uncontrolled jerky movements of the face, mouth or limbs. USN as a manifestation of NCSE has not been reported till date. USN is a common finding in patients with a right hemispheric stroke and is defined as “the failure to report, respond, or orient to novel or meaningful stimuli presented to the side opposite a brain lesion, when this failure cannot be attributed to either sensory or motor defects.”[2] It is a failure or decreased ability to move in the contra‑ lesional space despite being aware of a stimulus in that space. Normally, the right hemisphere attends to both hemispaces, while the left hemisphere attends primarily to the right hemispace. In a right hemispheric lesion, the left hemisphere attends primarily to the right hemispace resulting in a neglect of the left hemispace.[3] USN occurs as a result of lesions at the parietooccipital junction in the right hemisphere.[4] But, lesions in the occipital and frontal lobes, the thalamus, and putamen can also produce this symptom. Valler et al., (1988) suggested that the right fronto-temporo-parietal cortex could be the site of the lesion responsible for the USN.[5] Mesulam suggested that the posterior
parietal, frontal, and reticular components provide an integrated network for the modulation of attention within the extrapersonal space.[6] The posterior parietal lobe is primarily responsible for creating a sensory representation of extrapersonal events, and for targeting specific stimuli. The frontal eye fields and adjacent premotor areas regulate visual scanning, orient the head and eyes to explore the environment, and initiate motor acts for limb movement. Each component region gives rise to a different clinical type of USN when damaged. Transient regional diffusion‑weighted MRI (DWI) hyperintensity has been reported in patients with status epilepticus. MRI abnormalities in our patient may have, in all probability, been due to NCSE, since the CSF examination and angiography were normal. To conclude, this patient had a left unilateral spatial motor neglect as an ictal manifestation with the EEG showing epileptiform discharges. The motor neglect disappeared following the administration of an appropriate antiepileptic drug. NCSE should be considered as an etiology for causing USN apart from the usual vascular lesions.
Rohan Mahale, Anish Mehta, R. Srinivasa Department of Neurology, Mathikere Sampangappa Ramaiah Medical College and Hospital, Bangalore, Karnataka, India
Neurology India / March 2015 / Volume 63 / Issue 2
E‑mail: [email protected]
263
[Downloaded free from http://www.neurologyindia.com on Thursday, May 07, 2015, IP: 61.16.135.116]
Letters to Editor
References 1. 2.
3.
4. 5.
6.
Kaplan PW. Nonconvulsive status epilepticus. Semin Neurol 1996;16:33‑40. Heilman KM, Watson RT, Valenstein E. Neglect and related disorders. In: Heilman KM, Valenstein E, editors. Clinical Neuropsychology. New York: Oxford University Press; 1993. p. 279‑336. Mesulam MM. Attention, confusional states, and neglect. In: Mesulam MM, editor. Principles of Behavioral Neurology. Philadelphia: FA Davis Co; 1985. p. 125‑68. Kubo K. Localisation of unilateral spatial neglect. High Brain Funct Res 1989;9:106‑11. Vallar G, Perani D. The anatomy of unilateral neglect after right hemisphere stroke lesions. A clinical/CT‑scan correlation study in man. Neuropsychologia 1986;24:609‑22. Mesulam MM. Spatial attention and neglect: Parietal, frontal and cingulate contributions to the mental representation and attentional targeting of salient extrapersonal events. Philos Trans R Soc Lond B 1999;354:1325‑46. Access this article online
Website:
Quick Response Code
www.neurologyindia.com DOI: 10.4103/0028-3886.156301 PMID: xxxxx
Bilateral ptosis after burr hole evacuation of a chronic subdural hematoma Sir, The midline unpaired caudal nucleus of the third nerve lies in the midbrain, and supplies both the levators. Destructive lesions in this area or in the periaqueductal gray matter can lead to an isolated occurrence of bilateral ptosis. We present a unique case of a remote site hematoma in the midbrain following burr hole evacuation of a chronic subdural hematoma (CSDH) that caused bilateral ptosis. A 55‑year‑old man presented to our emergency department with a history of progressive weakness of the right upper and lower limb, aphasia, increased somnolence, and headache since the last 3 days. He was a known alcoholic for the past 30 years. The relatives recollected that the patient had a trivial fall a month ago and sustained a minor injury to the head.
and parietal burr hole drainage under general anesthesia. Around 100 ml of altered blood was evacuated slowly. Gentle saline irrigation was used to remove any remnant clots and blood products. Hemostasis was achieved and the wound closed without a drain. His neurological examination in the immediate postoperative period revealed bilateral ptosis with normal extraocular movements, and bilaterally equal and reactive pupils [Figure 3]. The patient's sensorium and right limb weakness also improved. Based on the clinical findings we inferred the lesion to be at the site of the unpaired midline nucleus of the third nerve that supplies the levators. This was further confirmed by a CT which revealed a 1 × 1.2 cm sized hematoma in the periaqueductal region of the midbrain [Figure 2]. The patient was treated conservatively and made a gradual recovery. At 6 months follow‑up, he shown a remarkable improvement except for the persistence of a mild bilateral ptosis. An intracerebral hematoma is a rare complication that they may develop following the evacuation of a CSDH. There is very little published data regarding this entity. Kotwica and Brzeziński[1] and Richter et al.,[2] have reported an incidence of 0.7–5%. There is no previous report of an intracerebral hematoma developing in the midbrain after evacuation of a CSDH in the published English literature. The oculomotor nerve arises from the oculomotor nuclear complex in the midbrain and conveys motor fibers to extraocular muscles and levators of the eyelid, and parasympathetic fibers to the pupil. The nucleus is situated in the periaqueductal gray matter just anterior to the aqueduct of Sylvius, at the level of the superior colliculi. Each oculomotor nucleus consists of multiple adjacent subnuclei that innervate specific ocular muscles. The paired lateral nuclei are the largest and are situated anterior and lateral to others; their medial portions are fused into an unpaired mass. A single midline structure, the central caudal nucleus, supplies the levator palpebrae muscles on both sides. The periaqueductal gray matter is also involved influencing the eyelid function; its destructive lesions may cause ptosis.[3] The symptoms of an intracerebral hematoma may occur either immediately or several days after evacuation of a CSDH. The hematoma can either be adjacent to the location of the CSDH or at a remote site, as occured in our case.
The underlying pathophysiology of this entity has not been clearly elucidated. Evaluation of blood flow changes in His computed tomography (CT) revealed a left‑sided, fronto-temporo-parietal CSDH, with a moderate midline patients with a CSDH using positron emission tomography has shift [Figure 1]. The routine preoperative hematological and demonstrated decreased blood flow at remote sites like the biochemical parameters were normal. We performed a frontal thalamus and the basal ganglia that is followed postoperatively Neurology India / March 2015 / Volume 63 / Issue 2 264
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Letters to Editor
Unilateral spatial neglect as a presenting manifestation of nonconvulsive status epilepticus Sir, Nonconvulsive status epilepticus (NCSE) is defined as a change in the mental status of at least 30 to 60 minutes duration, associated with continuous or near continuous ictal discharges detectable on an electroencephalography (EEG).[1] A diagnosis of NCSE requires the clinical manifestation of an abnormal mental status with reduced responsiveness, a supportive EEG, and a response to an antiepileptic drug. Unilateral spatial neglect (USN) as a manifestation of NCSE has not been reported till date. Here, we report the case of an elderly gentleman who presented with left spatial neglect with his EEG showing continuous, sharp wave discharges from the right parietooccipital region suggestive of the background presence of a NCSE. A 64‑year‑old, right‑handed man was brought in an altered mental status of a day’s duration. His wife found him to be in an acute confusional state on being woken up. He was talking irrelevantly and was disoriented. He was always looking towards his right side and was not moving his left upper and lower limb spontaneously and to any painful stimulus. On admission, he was drowsy but easily arousable on verbal commands. A command to shift his gaze towards his left side would
a
NCSE constitutes approximately 20–23% of cases presenting as status epilepticus. The clinical presentation of a NCSE
c
b
e
make him look towards the right side. There was intermittent horizontal, jerky nystagmus of both eyes towards the left side with fast component also towards the left. Several requests to lift his left upper and lower limbs always resulted in his lifting his right upper and lower limb. If the examiner called his name from his left hemispace, he would respond by looking towards his right hemispace [Video 1 (www.neurologyindia.com)]. There was no facial palsy. He had a normal tone, power and reflexes in his limbs with flexor plantars. His magnetic resonance imaging (MRI) of the brain showed diffusion restriction in the right parieto‑temporo‑occipital region with a hyperintense signal change in the right pulvinar region of the thalamus on fluid‑attenuated inversion recovery (FLAIR) images [Figure 1]. The MR angiography was normal. His EEG showed sharp wave discharges from the right temporo‑parieto‑occipital region with generalization, which disappeared on intravenous lorazepam (4mg) administration suggesting the presence of a NCSE [Figure 2]. His cerebrospinal fluid (CSF) analysis was normal and the CSF polymerase chain reaction (PCR) for herpes simplex virus was negative. A diagnosis of left USN as a manifestation of NCSE was made. He was treated with a loading dose of intravenous phenytoin (20 mg/kg) followed by continuance of a maintenance dose (5 mg/kg). A repeat EEG performed after 48 h revealed the disappearance of the epileptiform discharges. There was clinical improvement in the form of disappearance of the spatial neglect with return to normal sensorium [Video 2 (www.neurologyindia.com)].
f
d
g
Figure 1: (a-d) Magnetic resonance imaging (MRI) brain diffusion-weighted MRI (DWI) images axial view showing gyral hyperintensities in the right parieto-temporo-occipital region and right pulvinar of thalamus (white arrow); (e-g) MRI brain apparent-diffusion coefficient (ADC) showing corresponding hypointensities indicating diffusion restriction (white arrow) 262
Neurology India / March 2015 / Volume 63 / Issue 2
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Letters to Editor
Figure 2: Electroencephalogram (EEG) (bipolar longitudinal montage, sensitivity 14.0 μV/mm, low frequency filter 1 Hz, high frequency filter 70 Hz, notch 50 Hz, and speed 30 mm/s) showing sharp wave discharges from the right temporo-parieto-occipital region (red arrow) and disappearance of discharges with intravenous lorazepam
may vary from manifesting as a subtle abnormality to the presence of one or more of the following: Variation in the level of consciousness, fluctuation in behavior, semi‑purposeful movements, changes in or even a loss of speech, or uncontrolled jerky movements of the face, mouth or limbs. USN as a manifestation of NCSE has not been reported till date. USN is a common finding in patients with a right hemispheric stroke and is defined as “the failure to report, respond, or orient to novel or meaningful stimuli presented to the side opposite a brain lesion, when this failure cannot be attributed to either sensory or motor defects.”[2] It is a failure or decreased ability to move in the contra‑ lesional space despite being aware of a stimulus in that space. Normally, the right hemisphere attends to both hemispaces, while the left hemisphere attends primarily to the right hemispace. In a right hemispheric lesion, the left hemisphere attends primarily to the right hemispace resulting in a neglect of the left hemispace.[3] USN occurs as a result of lesions at the parietooccipital junction in the right hemisphere.[4] But, lesions in the occipital and frontal lobes, the thalamus, and putamen can also produce this symptom. Valler et al., (1988) suggested that the right fronto-temporo-parietal cortex could be the site of the lesion responsible for the USN.[5] Mesulam suggested that the posterior
parietal, frontal, and reticular components provide an integrated network for the modulation of attention within the extrapersonal space.[6] The posterior parietal lobe is primarily responsible for creating a sensory representation of extrapersonal events, and for targeting specific stimuli. The frontal eye fields and adjacent premotor areas regulate visual scanning, orient the head and eyes to explore the environment, and initiate motor acts for limb movement. Each component region gives rise to a different clinical type of USN when damaged. Transient regional diffusion‑weighted MRI (DWI) hyperintensity has been reported in patients with status epilepticus. MRI abnormalities in our patient may have, in all probability, been due to NCSE, since the CSF examination and angiography were normal. To conclude, this patient had a left unilateral spatial motor neglect as an ictal manifestation with the EEG showing epileptiform discharges. The motor neglect disappeared following the administration of an appropriate antiepileptic drug. NCSE should be considered as an etiology for causing USN apart from the usual vascular lesions.
Rohan Mahale, Anish Mehta, R. Srinivasa Department of Neurology, Mathikere Sampangappa Ramaiah Medical College and Hospital, Bangalore, Karnataka, India
Neurology India / March 2015 / Volume 63 / Issue 2
E‑mail: [email protected]
263
[Downloaded free from http://www.neurologyindia.com on Thursday, May 07, 2015, IP: 61.16.135.116]
Letters to Editor
References 1. 2.
3.
4. 5.
6.
Kaplan PW. Nonconvulsive status epilepticus. Semin Neurol 1996;16:33‑40. Heilman KM, Watson RT, Valenstein E. Neglect and related disorders. In: Heilman KM, Valenstein E, editors. Clinical Neuropsychology. New York: Oxford University Press; 1993. p. 279‑336. Mesulam MM. Attention, confusional states, and neglect. In: Mesulam MM, editor. Principles of Behavioral Neurology. Philadelphia: FA Davis Co; 1985. p. 125‑68. Kubo K. Localisation of unilateral spatial neglect. High Brain Funct Res 1989;9:106‑11. Vallar G, Perani D. The anatomy of unilateral neglect after right hemisphere stroke lesions. A clinical/CT‑scan correlation study in man. Neuropsychologia 1986;24:609‑22. Mesulam MM. Spatial attention and neglect: Parietal, frontal and cingulate contributions to the mental representation and attentional targeting of salient extrapersonal events. Philos Trans R Soc Lond B 1999;354:1325‑46. Access this article online
Website:
Quick Response Code
www.neurologyindia.com DOI: 10.4103/0028-3886.156301 PMID: xxxxx
Bilateral ptosis after burr hole evacuation of a chronic subdural hematoma Sir, The midline unpaired caudal nucleus of the third nerve lies in the midbrain, and supplies both the levators. Destructive lesions in this area or in the periaqueductal gray matter can lead to an isolated occurrence of bilateral ptosis. We present a unique case of a remote site hematoma in the midbrain following burr hole evacuation of a chronic subdural hematoma (CSDH) that caused bilateral ptosis. A 55‑year‑old man presented to our emergency department with a history of progressive weakness of the right upper and lower limb, aphasia, increased somnolence, and headache since the last 3 days. He was a known alcoholic for the past 30 years. The relatives recollected that the patient had a trivial fall a month ago and sustained a minor injury to the head.
and parietal burr hole drainage under general anesthesia. Around 100 ml of altered blood was evacuated slowly. Gentle saline irrigation was used to remove any remnant clots and blood products. Hemostasis was achieved and the wound closed without a drain. His neurological examination in the immediate postoperative period revealed bilateral ptosis with normal extraocular movements, and bilaterally equal and reactive pupils [Figure 3]. The patient's sensorium and right limb weakness also improved. Based on the clinical findings we inferred the lesion to be at the site of the unpaired midline nucleus of the third nerve that supplies the levators. This was further confirmed by a CT which revealed a 1 × 1.2 cm sized hematoma in the periaqueductal region of the midbrain [Figure 2]. The patient was treated conservatively and made a gradual recovery. At 6 months follow‑up, he shown a remarkable improvement except for the persistence of a mild bilateral ptosis. An intracerebral hematoma is a rare complication that they may develop following the evacuation of a CSDH. There is very little published data regarding this entity. Kotwica and Brzeziński[1] and Richter et al.,[2] have reported an incidence of 0.7–5%. There is no previous report of an intracerebral hematoma developing in the midbrain after evacuation of a CSDH in the published English literature. The oculomotor nerve arises from the oculomotor nuclear complex in the midbrain and conveys motor fibers to extraocular muscles and levators of the eyelid, and parasympathetic fibers to the pupil. The nucleus is situated in the periaqueductal gray matter just anterior to the aqueduct of Sylvius, at the level of the superior colliculi. Each oculomotor nucleus consists of multiple adjacent subnuclei that innervate specific ocular muscles. The paired lateral nuclei are the largest and are situated anterior and lateral to others; their medial portions are fused into an unpaired mass. A single midline structure, the central caudal nucleus, supplies the levator palpebrae muscles on both sides. The periaqueductal gray matter is also involved influencing the eyelid function; its destructive lesions may cause ptosis.[3] The symptoms of an intracerebral hematoma may occur either immediately or several days after evacuation of a CSDH. The hematoma can either be adjacent to the location of the CSDH or at a remote site, as occured in our case.
The underlying pathophysiology of this entity has not been clearly elucidated. Evaluation of blood flow changes in His computed tomography (CT) revealed a left‑sided, fronto-temporo-parietal CSDH, with a moderate midline patients with a CSDH using positron emission tomography has shift [Figure 1]. The routine preoperative hematological and demonstrated decreased blood flow at remote sites like the biochemical parameters were normal. We performed a frontal thalamus and the basal ganglia that is followed postoperatively Neurology India / March 2015 / Volume 63 / Issue 2 264
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
