Letters to Editor
References 1. Cho YL, Liu HN, Huang TP, Tarng DC. Uremic pruritus: Roles of parathyroid hormone and substance P. J Am Acad Dermatol 1997;36:538‑43. 2. Elsone L, Townsend T, Mutch K, Das K, Boggild M, Nurmikko T, et al. Neuropathic pruritus (itch) in neuromyelitisoptica. Mult Scler 2013;19:475‑9. 3. Sang‑Soo L, Hyung‑Suk L, Shin‑Hye B. Paroxysmal pruritus as the first relapsing symptom of neuromyelitisoptica. Neurology Asia 2010;15:185‑7. 4. Osterman PO, Westerberg CE. Paroxysmal attacks in multiple sclerosis. Brain 1975;98:189‑202. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.137020
Disease Rating Scale (UPDRS) score was 36. There were no other risk factors or features for seconday Porkinsonism. Computed tomography (CT) revealed multilobular cysts without calcification in bilateral basal ganglia, [Figure 1a]. Magnetic resonance imaging (MRI) demonstrated multilobular cysts in the basal ganglia, periventricular white matter, and centrum semiovale bilaterally. The cysts were isointense to cerebrospinal fluid (CSF) on all MRI sequences [Figure 1b‑f] with no contrast enhancement [Figure 1c]. There was no hydrocephalus, but mass effect was evident in the tissues adjacent to the cysts [Figures 1b, d‑f]. MR‑angiography was negative for vascular malformations. Comprehensive work‑up for any central nervous system infections was negative. Based on this data a final diagnosis of giant perivascular space was considered. She was put on dopamine agonist, pramipexol 3 × 0.125 mg initialy and was gradually titrated to 3 × 1 mg. At 10 months of follow‑up, she had significant improvement in her symptoms and UPDRS score was 11.
Sir, Virchow‑Robin or perivascular spaces (PVSs) are pia‑mater lined zones that surround vessels penetrating in and out of brain parenchyma. Virchow‑Robin spaces with diameter of >1.5 cm3 are termed as giant variants.[1] Giant PVS can be misdiagnosed as cystic lacunar infarcts, cystic tumors, and cystic infections especially when there are associated neurologic symptoms. We presented a patient with bilateral basal ganglia giant PVS and Parkinsonism.
Perivascular spaces are fluid filled spaces around vascular structures. Rudolf Virchow (German pathologist) and Charles Philippe Robin (French anatomist) described PVS in the years 1851 and 1859, respectively.[2] Structure of PVS differs whether they accompany arterial or venous vessels and whether their location is superficial or deeper in the brain. [3] Perivascular spaces are too thin in diameter (only 1-2 mm), however they have more easily been detected after invention of high resolution T2‑weighted MRI. Although there have been numerous arguments about nature of the fluid in PVS, today it has been accepted that these spaces function as drainage pathways for brain interstitial fluid and have some immune defense mechanisms by harboring microglia inside.[3] On MRI, PVS dilations are isointense to CSF in imaging sequences with no contrast enhancement.[3‑5] They are well circumscribed and follow lenticulostriate arteries in anterior perforated substance, perforating arteries in the white matter and penetrating branches of collicular and accessory collicular arteries in midbrain.[4,5]
A 54‑year‑old right-handed woman presented with asymmetric tremor, markedly in the right hand and difficulty to walk for 1 year duration. Neurological examination revealed mild facial masking, decreased blink rate, hypophonia, decreased arm swing on the right, 4–6 Hz bilateral resting tremor and cogwheel rigidity distinct on the right side, bradykinesia, and difficulty in walking. Handwriting was small and became smaller as she continued to write. Pull test was positive without recovery. Total Unified Parkinson’s
Differential for PVS includes cystic lacunar infarctions, cystic tumors and cystic infections.[3] Cystic lacunar infarcts have similar image intensities like dilated PVS as they contain water. Cystic infarctions are more common in white matter, whereas dilated PVS present usually in basal ganglia. Cystic infarctions have slit‑like or ovoid shape, dilated PVS are round or linear.[6] On CT image, dilated PVS do not have calcifications, whereas they may be a feature of parasitic infestations. As in our case, laboratory tests also help to differentiate
Received: 10-06-2014 Review completed: 26-06-2014 Accepted: 15‑06‑2014
Giant dilations of perivascular spaces in deep brain locations: A cause for parkinsonism?
Neurology India | May-Jun 2014 | Vol 62 | Issue 3
327
Letters to Editor
a
b
c
d
e
f
Figure 1: Computed tomography shows hypodense cystic areas in both basal ganglia. Neither cyst has calcification (a). Fluid-attenuated inversion recovery (FLAIR) and T2-weighted magnetic resonance images reveal multilobular cysts that are isointense to cerebrospinal fluid and located in the basal ganglia, periventricular white matter, and centrum semiovale. Note the pericystic edema on the FLAIR images (b,d-f). On T1-weighted scans, cysts do not enhance with intravenous gadolinium (c)
dilated PVS from infections. As a confounding matter, hyperintensity observed around some dilated PVS, which makes us think tumor diagnosis, sometimes delays the diagnosis. However, these fields can be distinguished as gliotic regions with new MRI technologies (MR spectroscopy).
clinical findings regressed gradually. In this patient, one cannot exclude the possiblity of occurrence of the two diseases in the same patient. However, clinical rarity of the condition prevents large‑scale prospective studies to be conducted.
Presenting clinical features of giant PVS depend on the location of the PVS. Giant PVS present near CSF pathways like in mesencephalon and brain stem, may present with hydrocephalus.[3] Hydrocephalus develops insidiously and the clinical presentation may mimic normal‑pressure‑hydrocephalus. Management with CSF diversion surgeries (V‑P shunt, cystoperitoneal shunt, ventriculocisternostomy) alleviates signs and symptoms even though cyst size generally does not change. [3] There has been case reports relating seizure disorder to PVS; however, the relationship is uncertain.[3] Bastos et al. described a 64‑year‑old man with PVS dilation in hypocampal region, who presented with temporal lobe seizure. [7] Patient with PVS and seizure disorder should be evaluated comprehensively before the causal relation between them is considered Multiple enlarged PVS in striatum, or état criblé, has been related with arteriosclerotic Parkinsonism.[3] However, the underlying mechanism has not been outlined, yet. An explanation in this patient can be the location of giant PVS in the basal ganglia. Bilateral compression of extrapyramidal pathways might have resulted in Parkinsonism signs and symptoms. After dopamine agonist therapy,
Baran Yılmaz, Zafer Orkun Toktaş, Murat Şakir Ekşi1, Hayal Ergin Toktaş2, Füsun Mayda Domaç2, Türker Kılıç
328
Departments of Neurosurgery, Bahcesehir University Medical School, Istanbul, 2Neurology, Erenkoy Mental Research and Training Hospital, Istanbul, Turkey, 1 Department of Orthopedics‑Spine Center, University of California, San Francisco, California, USA E‑mail: [email protected]
References 1. 2. 3. 4. 5. 6.
Ahmad FU, Garg A, Singh M, Mishra NK. Giant mesencephalothalamic virchow‑robin spaces causing obstructive hydrocephalus. A case report. Neuroradiol J 2007;20:303‑6. Kwee RM, Kwee TC. Virchow‑Robin spaces at MR imaging. Radiographics 2007;27:1071‑86. House P, Salzman KL, Osborn AG, MacDonald JD, Jensen RL, Couldwell WT. Surgical considerations regarding giant dilations of the perivascular spaces. J Neurosurg 2004;100:820‑4. Jhawar SS, Garewal SS, Bhargava P, Nittala PP. Dilated Virchow Robin spaces mimicking cystic neoplasm of cingulated gyrus. Neurol India 2012;60:136‑7. Kanamalla US, Calabro F, Jinkins JR. Cavernous dilatation of mesencephalic Virchow‑Robin spaces with obstructive hydrocephalus. Neuroradiology 2000;42:881‑4. Bokura H, Kobayashi S, Yamaguchi S. Distinguishing silent lacunar infarction from enlarged Virchow‑Robin spaces: A magnetic resonance Neurology India | May-Jun 2014 | Vol 62 | Issue 3
Letters to Editor
imaging and pathological study. J Neurol 1998;245:116‑22. 7. Bastos AC, Andermann F, Melancon D, Cendes F, Guberman A, Dubeau F, et al. Late‑onset temporal lobe epilepsy and dilatation of the hippocampal sulcus by an enlarged Virchow‑Robin space. Neurology 1998;50:784‑7.
abuse[5] and vasculopathies.[5,6] The surgical bed and the immediately surrounding areas are most often the site of this complication. However, there can be intracranial bleed away from the operative site “remote site bleed” following a neurosurgical procedure.
Access this article online
Remote site bleed following removal of a space occupying lesion or drainage of cerebrospinal fluid (CSF) is a rare and dreaded complication. It carries significant morbidity and mortality. The location of remote site bleed can be epidural, subdural, or intracerebral [7,8] and can be supratentorial or infratentorial following a supratentorial or infratentorial surgery. van Gehuchten in 1937 described the first case of remote site bleed, a pontine hemorrhage secondary to a subtemporal decompression for a temporal lobe meningioma.[9] This report describes six patients with remote site bleed following a neurosurgical procedure and reviewed the published literature in this regard.
Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.137022
Received: 03‑06‑2014 Review completed: 10-06-2014 Accepted: 10‑06‑2014
Remote site intracranial hemorrhage: Our experience and review of literature Sir, Hemorrhage within the surgical bed frequently complicates a neurosurgical procedure. Postoperative hematoma in the absence of risk factors is reported to occur in about 0.6-1.4% of cases.[1,2] Known risk factors are coagulopathies and anticoagulant therapy,[3,4] alcohol
Remote site bleed is defined as intracranial bleed/hematoma at a site away from the primary surgery site. We retrospectively analyzed data of patients who underwent cranial neurosurgical procedure at our center. Six patients with remote site bleed in the post‑operative period were selected and their case records were reviewed [Table 1]. The demographic data, diagnoses, surgical procedure performed, cause of bleed, and final outcomes were reviewed. Patients with antecedent or postoperative coagulopathy were excluded from this study.
Table 1: Summary of our cases
Age/ Symptoms gender
Diagnosis
Procedure performed
Site of bleed
Type of bleed
Redo surgery
28 year/ Female
Headache, blurring of vision
Craniotomy and marsupialization
Supratentorial (Bilateral frontal)
EDH
50 year/ Female
Hearing loss, facial paresis, gait ataxia
Rt sylvian Fissure arachnoid cyst Lt vestibular schwannoma
Supratentorial (Diffuse)
SAH
20 year/ Male
Lt FTP acute subdural hematoma and underwent Lt FTP decompressive craniectomy, presented with skin flap bulge Hearing loss, facial paresis, gait ataxia
Lt FTP sudural hygroma
Lt RMSOC and tumor excision Intraoperative‑ Cerebellar bulge +, Ventriculostomy done Tapping of subdural fluid
Craniotomy and evacuation of hematoma VP shunt
Supratentorial (Diffuse)
Intraparenchymal None
3
Rt vestibular schwannoma
RtRMSOC and tumor excision
Supratentorial (Lt parieto‑temporal)
EDH
5
Cerebellar hematoma
MLSOC and evacuation of hematoma Lt frontal craniotomy and excision of tumor
Supratentorial (left fronto parieto temporal) Supratentorial (Rt frontal)
SDH
38 year/ Female 64 year/ Male 13 year/ Female
Sudden onset loss of consciousness GCS E2VtM4 Headache, blurring of vision
Lt lateral ventricular neurocytoma
EDH
Craniotomy and evacuation of hematoma Craniotomy and evacuation of hematoma Craniotomy and evacuation of hematoma
GOSat 6 months 5
4
4
5
GOS - Glasgow outcome score; EDH - Extradural hematoma; RMSOC - Retromastoid suboccipital craniotomy; SAH - Subarachnoid hemorrhage; FTP - Fronto‑ temporo‑parietal; MLSOC - Midline suboccipital craniotomy; SDH - Subdural hematoma; F - Female; M - Male
Neurology India | May-Jun 2014 | Vol 62 | Issue 3
329
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.
References 1. Cho YL, Liu HN, Huang TP, Tarng DC. Uremic pruritus: Roles of parathyroid hormone and substance P. J Am Acad Dermatol 1997;36:538‑43. 2. Elsone L, Townsend T, Mutch K, Das K, Boggild M, Nurmikko T, et al. Neuropathic pruritus (itch) in neuromyelitisoptica. Mult Scler 2013;19:475‑9. 3. Sang‑Soo L, Hyung‑Suk L, Shin‑Hye B. Paroxysmal pruritus as the first relapsing symptom of neuromyelitisoptica. Neurology Asia 2010;15:185‑7. 4. Osterman PO, Westerberg CE. Paroxysmal attacks in multiple sclerosis. Brain 1975;98:189‑202. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.137020
Disease Rating Scale (UPDRS) score was 36. There were no other risk factors or features for seconday Porkinsonism. Computed tomography (CT) revealed multilobular cysts without calcification in bilateral basal ganglia, [Figure 1a]. Magnetic resonance imaging (MRI) demonstrated multilobular cysts in the basal ganglia, periventricular white matter, and centrum semiovale bilaterally. The cysts were isointense to cerebrospinal fluid (CSF) on all MRI sequences [Figure 1b‑f] with no contrast enhancement [Figure 1c]. There was no hydrocephalus, but mass effect was evident in the tissues adjacent to the cysts [Figures 1b, d‑f]. MR‑angiography was negative for vascular malformations. Comprehensive work‑up for any central nervous system infections was negative. Based on this data a final diagnosis of giant perivascular space was considered. She was put on dopamine agonist, pramipexol 3 × 0.125 mg initialy and was gradually titrated to 3 × 1 mg. At 10 months of follow‑up, she had significant improvement in her symptoms and UPDRS score was 11.
Sir, Virchow‑Robin or perivascular spaces (PVSs) are pia‑mater lined zones that surround vessels penetrating in and out of brain parenchyma. Virchow‑Robin spaces with diameter of >1.5 cm3 are termed as giant variants.[1] Giant PVS can be misdiagnosed as cystic lacunar infarcts, cystic tumors, and cystic infections especially when there are associated neurologic symptoms. We presented a patient with bilateral basal ganglia giant PVS and Parkinsonism.
Perivascular spaces are fluid filled spaces around vascular structures. Rudolf Virchow (German pathologist) and Charles Philippe Robin (French anatomist) described PVS in the years 1851 and 1859, respectively.[2] Structure of PVS differs whether they accompany arterial or venous vessels and whether their location is superficial or deeper in the brain. [3] Perivascular spaces are too thin in diameter (only 1-2 mm), however they have more easily been detected after invention of high resolution T2‑weighted MRI. Although there have been numerous arguments about nature of the fluid in PVS, today it has been accepted that these spaces function as drainage pathways for brain interstitial fluid and have some immune defense mechanisms by harboring microglia inside.[3] On MRI, PVS dilations are isointense to CSF in imaging sequences with no contrast enhancement.[3‑5] They are well circumscribed and follow lenticulostriate arteries in anterior perforated substance, perforating arteries in the white matter and penetrating branches of collicular and accessory collicular arteries in midbrain.[4,5]
A 54‑year‑old right-handed woman presented with asymmetric tremor, markedly in the right hand and difficulty to walk for 1 year duration. Neurological examination revealed mild facial masking, decreased blink rate, hypophonia, decreased arm swing on the right, 4–6 Hz bilateral resting tremor and cogwheel rigidity distinct on the right side, bradykinesia, and difficulty in walking. Handwriting was small and became smaller as she continued to write. Pull test was positive without recovery. Total Unified Parkinson’s
Differential for PVS includes cystic lacunar infarctions, cystic tumors and cystic infections.[3] Cystic lacunar infarcts have similar image intensities like dilated PVS as they contain water. Cystic infarctions are more common in white matter, whereas dilated PVS present usually in basal ganglia. Cystic infarctions have slit‑like or ovoid shape, dilated PVS are round or linear.[6] On CT image, dilated PVS do not have calcifications, whereas they may be a feature of parasitic infestations. As in our case, laboratory tests also help to differentiate
Received: 10-06-2014 Review completed: 26-06-2014 Accepted: 15‑06‑2014
Giant dilations of perivascular spaces in deep brain locations: A cause for parkinsonism?
Neurology India | May-Jun 2014 | Vol 62 | Issue 3
327
Letters to Editor
a
b
c
d
e
f
Figure 1: Computed tomography shows hypodense cystic areas in both basal ganglia. Neither cyst has calcification (a). Fluid-attenuated inversion recovery (FLAIR) and T2-weighted magnetic resonance images reveal multilobular cysts that are isointense to cerebrospinal fluid and located in the basal ganglia, periventricular white matter, and centrum semiovale. Note the pericystic edema on the FLAIR images (b,d-f). On T1-weighted scans, cysts do not enhance with intravenous gadolinium (c)
dilated PVS from infections. As a confounding matter, hyperintensity observed around some dilated PVS, which makes us think tumor diagnosis, sometimes delays the diagnosis. However, these fields can be distinguished as gliotic regions with new MRI technologies (MR spectroscopy).
clinical findings regressed gradually. In this patient, one cannot exclude the possiblity of occurrence of the two diseases in the same patient. However, clinical rarity of the condition prevents large‑scale prospective studies to be conducted.
Presenting clinical features of giant PVS depend on the location of the PVS. Giant PVS present near CSF pathways like in mesencephalon and brain stem, may present with hydrocephalus.[3] Hydrocephalus develops insidiously and the clinical presentation may mimic normal‑pressure‑hydrocephalus. Management with CSF diversion surgeries (V‑P shunt, cystoperitoneal shunt, ventriculocisternostomy) alleviates signs and symptoms even though cyst size generally does not change. [3] There has been case reports relating seizure disorder to PVS; however, the relationship is uncertain.[3] Bastos et al. described a 64‑year‑old man with PVS dilation in hypocampal region, who presented with temporal lobe seizure. [7] Patient with PVS and seizure disorder should be evaluated comprehensively before the causal relation between them is considered Multiple enlarged PVS in striatum, or état criblé, has been related with arteriosclerotic Parkinsonism.[3] However, the underlying mechanism has not been outlined, yet. An explanation in this patient can be the location of giant PVS in the basal ganglia. Bilateral compression of extrapyramidal pathways might have resulted in Parkinsonism signs and symptoms. After dopamine agonist therapy,
Baran Yılmaz, Zafer Orkun Toktaş, Murat Şakir Ekşi1, Hayal Ergin Toktaş2, Füsun Mayda Domaç2, Türker Kılıç
328
Departments of Neurosurgery, Bahcesehir University Medical School, Istanbul, 2Neurology, Erenkoy Mental Research and Training Hospital, Istanbul, Turkey, 1 Department of Orthopedics‑Spine Center, University of California, San Francisco, California, USA E‑mail: [email protected]
References 1. 2. 3. 4. 5. 6.
Ahmad FU, Garg A, Singh M, Mishra NK. Giant mesencephalothalamic virchow‑robin spaces causing obstructive hydrocephalus. A case report. Neuroradiol J 2007;20:303‑6. Kwee RM, Kwee TC. Virchow‑Robin spaces at MR imaging. Radiographics 2007;27:1071‑86. House P, Salzman KL, Osborn AG, MacDonald JD, Jensen RL, Couldwell WT. Surgical considerations regarding giant dilations of the perivascular spaces. J Neurosurg 2004;100:820‑4. Jhawar SS, Garewal SS, Bhargava P, Nittala PP. Dilated Virchow Robin spaces mimicking cystic neoplasm of cingulated gyrus. Neurol India 2012;60:136‑7. Kanamalla US, Calabro F, Jinkins JR. Cavernous dilatation of mesencephalic Virchow‑Robin spaces with obstructive hydrocephalus. Neuroradiology 2000;42:881‑4. Bokura H, Kobayashi S, Yamaguchi S. Distinguishing silent lacunar infarction from enlarged Virchow‑Robin spaces: A magnetic resonance Neurology India | May-Jun 2014 | Vol 62 | Issue 3
Letters to Editor
imaging and pathological study. J Neurol 1998;245:116‑22. 7. Bastos AC, Andermann F, Melancon D, Cendes F, Guberman A, Dubeau F, et al. Late‑onset temporal lobe epilepsy and dilatation of the hippocampal sulcus by an enlarged Virchow‑Robin space. Neurology 1998;50:784‑7.
abuse[5] and vasculopathies.[5,6] The surgical bed and the immediately surrounding areas are most often the site of this complication. However, there can be intracranial bleed away from the operative site “remote site bleed” following a neurosurgical procedure.
Access this article online
Remote site bleed following removal of a space occupying lesion or drainage of cerebrospinal fluid (CSF) is a rare and dreaded complication. It carries significant morbidity and mortality. The location of remote site bleed can be epidural, subdural, or intracerebral [7,8] and can be supratentorial or infratentorial following a supratentorial or infratentorial surgery. van Gehuchten in 1937 described the first case of remote site bleed, a pontine hemorrhage secondary to a subtemporal decompression for a temporal lobe meningioma.[9] This report describes six patients with remote site bleed following a neurosurgical procedure and reviewed the published literature in this regard.
Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.137022
Received: 03‑06‑2014 Review completed: 10-06-2014 Accepted: 10‑06‑2014
Remote site intracranial hemorrhage: Our experience and review of literature Sir, Hemorrhage within the surgical bed frequently complicates a neurosurgical procedure. Postoperative hematoma in the absence of risk factors is reported to occur in about 0.6-1.4% of cases.[1,2] Known risk factors are coagulopathies and anticoagulant therapy,[3,4] alcohol
Remote site bleed is defined as intracranial bleed/hematoma at a site away from the primary surgery site. We retrospectively analyzed data of patients who underwent cranial neurosurgical procedure at our center. Six patients with remote site bleed in the post‑operative period were selected and their case records were reviewed [Table 1]. The demographic data, diagnoses, surgical procedure performed, cause of bleed, and final outcomes were reviewed. Patients with antecedent or postoperative coagulopathy were excluded from this study.
Table 1: Summary of our cases
Age/ Symptoms gender
Diagnosis
Procedure performed
Site of bleed
Type of bleed
Redo surgery
28 year/ Female
Headache, blurring of vision
Craniotomy and marsupialization
Supratentorial (Bilateral frontal)
EDH
50 year/ Female
Hearing loss, facial paresis, gait ataxia
Rt sylvian Fissure arachnoid cyst Lt vestibular schwannoma
Supratentorial (Diffuse)
SAH
20 year/ Male
Lt FTP acute subdural hematoma and underwent Lt FTP decompressive craniectomy, presented with skin flap bulge Hearing loss, facial paresis, gait ataxia
Lt FTP sudural hygroma
Lt RMSOC and tumor excision Intraoperative‑ Cerebellar bulge +, Ventriculostomy done Tapping of subdural fluid
Craniotomy and evacuation of hematoma VP shunt
Supratentorial (Diffuse)
Intraparenchymal None
3
Rt vestibular schwannoma
RtRMSOC and tumor excision
Supratentorial (Lt parieto‑temporal)
EDH
5
Cerebellar hematoma
MLSOC and evacuation of hematoma Lt frontal craniotomy and excision of tumor
Supratentorial (left fronto parieto temporal) Supratentorial (Rt frontal)
SDH
38 year/ Female 64 year/ Male 13 year/ Female
Sudden onset loss of consciousness GCS E2VtM4 Headache, blurring of vision
Lt lateral ventricular neurocytoma
EDH
Craniotomy and evacuation of hematoma Craniotomy and evacuation of hematoma Craniotomy and evacuation of hematoma
GOSat 6 months 5
4
4
5
GOS - Glasgow outcome score; EDH - Extradural hematoma; RMSOC - Retromastoid suboccipital craniotomy; SAH - Subarachnoid hemorrhage; FTP - Fronto‑ temporo‑parietal; MLSOC - Midline suboccipital craniotomy; SDH - Subdural hematoma; F - Female; M - Male
Neurology India | May-Jun 2014 | Vol 62 | Issue 3
329
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.
