- Email: [email protected]
Hemichorea, an unusual complication of ventriculoperitoneal shunt
Case Report / Journal of Clinical Neuroscience 15 (2008) 599–601
Physicians should be aware of the possibility of DP when prescribing TPM. We recommend careful monitoring of these patients and discontinuation of TPM if DP develops. References 1. Lyell A. Delusions of parasitosis. Br J Dermatol 1983;108:485–99. 2. Trabert W. 100 years of delusional parasitosis. Meta-analysis of 1,223 case reports. Psychopathology 1995;28:238–46.
599
3. Mula M, Trimble MR, Lhatoo SD, et al. Topiramate and psychiatric adverse events in patients with epilepsy. Epilepsia 2003;44: 659–63. 4. Trimble MR. New antiepileptic drugs and psychopathology. Neuropsychobiology 1998;38:149–51. 5. De Leon J, Antelo RE, Simpson G. Delusion of parasitosis or chronic tactile hallucinosis: hypothesis about their brain physiopathology. Compr Psychiatry 1992;33:25–33. 6. Berrios GE. Delusional parasitosis and physical disease. Compr Psychiatry 1985;26:395–403.
doi:10.1016/j.jocn.2006.12.017
Hemichorea, an unusual complication of ventriculoperitoneal shunt Likhith M. Alakandy *, Ramanathan Venkatachalam Iyer, Aprajay Golash Department of Neurosurgery, Royal Preston Hospital, Sharoe Green Lane, Fulwood, Preston PR2 9HT, Lancashire, UK Received 28 October 2006; accepted 6 December 2006
Abstract We report an unusual case of left hemichorea in a 24-year-old male patient that followed the insertion of a right ventriculoperitoneal shunt. This completely resolved after removal of the shunt. A review of the available literature and the possible aetiopathogenesis is discussed. Ó 2007 Elsevier Ltd. All rights reserved. Keywords: Hemichorea; Dyskinaesia; Ventriculoperitoneal shunt
1. Introduction Movement disorders are extremely rare complications of ventriculoperitoneal (VP) shunts. Contralateral parkinsonism and ballism have been reported following the insertion of ventricular catheters,1–3 and in all these patients, the symptoms resolved after removal of the shunt. Focal lesions in the basal ganglia typically produce contralateral chorea or hemiballism, whereas generalised injury may cause generalised chorea or ballism. The exact mechanism of reversible dyskinesia following malposition of ventricular catheters is not known. In this report, we describe such a patient presenting with hemichorea as a complication of VP shunt and discuss the possible aetiopathogenesis. 2. Case report A 24-year-old male patient presented at the age of 16 with a 5-month history of deteriorating visual acuity, diplopia and episodic loss of consciousness. He had papillo*
Corresponding author. Tel.: +44 7970741793; fax: +44 141 201 2020. E-mail addresses: [email protected] (L.M. Alakandy).
edema and bilateral sixth cranial nerve palsies. There was a history of meningococcal meningitis at the age of 2 years from which he had recovered completely without any residual deficits. CT scan showed obstructive hydrocephalus and aqueduct stenosis, presumably as a result of childhood meningitis. A right-sided medium pressure VP shunt was inserted following which there was satisfactory clinical improvement. He presented again 7 years later with a 3week history of headaches, diplopia and blurring of vision. Radiological examination of the shunt showed fracture of the shunt tube in the chest wall and distal migration of the peritoneal end. CT scan of the brain showed ventriculomegaly. The shunt was revised on the right side and he had complete resolution of his headaches and visual disturbance. Two weeks after revision of the shunt, he developed a choreiform movement of the left side of his body. These movements were rapid, irregular and non-purposeful, predominantly involving the upper limb. CT scan revealed the shunt catheter to be traversing the right basal ganglia with its tip in the third ventricle (Figs. 1 and 2). He refused to have the shunt revised and commenced medical treatment, which failed to control the dyskinesia. A CT scan 4 weeks
600
Case Report / Journal of Clinical Neuroscience 15 (2008) 599–601
later showed a right-sided chronic subdural haematoma in addition to a reduction in the size of the ventricles. The hemichorea persisted but he had no symptoms relating to the subdural haematoma. He agreed to surgical intervention. The shunt was removed and the subdural haematoma evacuated through a right temporal burr-hole. The choreiform movements completely resolved in a few days following this and to date he has not required reinsertion of the shunt. 3. Discussion
Fig. 1. Axial CT scan showing the shunt to be traversing the right thalamus.
Fig. 2. Axial CT scan showing the tip of the shunt in the third ventricle.
The most common lesions associated with hyperkinetic movement disorders are ischaemic stroke and multiple sclerosis.4,5 They can also rarely present as complications of a shunt. In our patient, local trauma or irritation might seem to be the obvious explanation for this. However, this is not always the case as highlighted in a report by Walker et al.2 He described a 68-year-old female patient who developed bilateral ballism following insertion of a right frontal VP shunt for normal pressure hydrocephalus. The position of the shunt catheter tip was confirmed to be intraventricular. On removal of the shunt, the ballism resolved after a few days. She later underwent another VP shunt, this time through a right parietal burr-hole and developed the same movement disorder, which resolved a few weeks after removal of the shunt. Cheshire et al.1 reported a 28-yearold female patient who developed unilateral tremor, rigidity and paraesthesia following administration of methotrexate via an Ommaya reservoir that was inadvertently placed too deep into the contralateral mesencephalon. Gooskens et al.3 reported a young girl, who developed voluntary movement myoclonus of the contralateral hand as a result of a granulomatous reaction to a shunt tip in the thalamus. These two reports suggest disruption of the neuronal network within the basal ganglia. Chorea, which is characterised by abrupt involuntary movements, is known to result from dysfunction within the complex network interconnecting motor cortical areas and the basal ganglia. Focal damage to the caudate nucleus, putamen, globus pallidus as well as associated structures like the subthalamic nucleus and substantia nigra can all lead to choreic disorders.6,7 In the last two decades, there has been considerable progress in understanding of the pathways responsible for unwanted movements. From primate models of experimental hyperkinetic syndromes, it can be suggested that contralateral hyperkinetic disorders result from disinhibition of the thalamic nuclei due to a reduction of tonic inhibitory output from the globus pallidus interna. This may result in the inability to suppress unwanted responses to the cortical sensory stimuli, resulting in chorea.8 Other movement disorders, including parkinsonism, have been reported to occur as a result of obstructive hydrocephalus.9 However, this is not always reversible. Apart from local damage due to a malpositioned shunt catheter, damage to the basal ganglia in hydrocephalus may be a result of direct pressure or chronic impairment of
Case Report / Journal of Clinical Neuroscience 15 (2008) 601–603
blood flow. When this is the case, the symptoms tend to improve with time after removal of the offending cause.10,11 However, in our patient the hemichorea did not appear for 2 weeks after the shunt was placed, and persisted for a few days after the shunt was removed. This makes local irritation an unlikely aetiology, where the symptoms should have manifested immediately. Decreased perfusion is also unlikely to be a valid explanation as the shunt was over-draining, as evidenced by the development of chronic subdural haematoma at his later presentation. Our patient has remained symptom-free at follow-up without a shunt. No other cause of the movement disorder was found. 4. Conclusion We report hemichorea as an extremely rare complication of ventriculoperitoneal shunts, and in the absence of structural damage appears to be reversible. The mechanism of this phenomenon is unknown. References
601
2. Walker FO, Hunt VP. Ballism: an association with ventriculoperitoneal shunting. Neurology 1990;40:1004. 3. Gooskens RH, Kaiser MC, Veiga-Pires JA. Computerised tomography in the management of complications due to ventricular shunting. Neuroradiology 1981;21:47–9. 4. O’Brien CF, Chorea. In: Jankovic J, Tolosa E, editors. Parkinson’s Disease and Movement Disorders. 3rd edition. Philadelphia: Lippincott Willimas and Wilkins; 1998. p. 357–64. 5. Alarcon F, Zijlmans JC, Duenas G, et al. Post-stroke movement disorders: report of 56 patients. J Neurol Neurosurg Psychiatry 2004;75:1568–74. 6. Slevin JT, Sparks DL, Dempsey RJ, et al. Altered striatal dopaminergic metabolism 36 hours after unilateral trauma to the human mesencephalon. Neurology 1987;37:322–5. 7. Bhatia KP, Marsden CD. The behavioural and motor consequences of focal lesions of the basal ganglia in man. Brain 1994;117:859–76. 8. DeLong MR. Primate models of movement disorders of basal ganglia origin. Trends Neurosci 1990;13:281–5. 9. Zeidler M, Dorman PJ, Ferguson IT, et al. Parkinsonism associated with obstructive hydrocephalus due to idiopathic aqueductal stenosis. J Neurol Neurosurg Psychiatry 1998;64:657–9. 10. Jankovic J, Newmark M, Peter P. Parkinsonism and acquired hydrocephalus. Move Disord 1986;1:59–64. 11. Shahar E, Lambert R, Hwang PA, et al. Obstructive hydrocephalus induced parkinsonism I: decreased basal ganglia regional blood flow. Pediatr Neurol 1988;4:117–9.
1. Cheshire WP, Ehle AL. Hemiparkinsonism as a complication of an Ommaya reservoir. Case report. J Neurosurg 1990;73:774–6. doi:10.1016/j.jocn.2006.12.016
Glioblastoma multiforme of the conus medullaris with holocordal spread Vivek Bonde, Srikant Balasubramaniam *, Atul Goel Department of Neurosurgery, R. No. 273, Seth G.S. Medical College and King Edward VII Memorial Hospital, Parel, Mumbai, Maharashtra 400 012, India Received 15 October 2006; accepted 1 December 2006
Abstract Glioblastoma multiforme of the conus medullaris is rare. It spreads both locally to contiguous structures and also by cerebrospinal seeding. The disease is aggressive and the ultimate prognosis is dismal. We report a case of conus medullaris glioblastoma multiforme with involvement of the holocord. In spite of excision of the conus medullaris tumor followed by radiation, the disease continued to progress rapidly. We discuss the rarity of the disease and the related literature with regard to treatment modalities. Ó 2007 Elsevier Ltd. All rights reserved. Keywords: Holocord; Astrocytoma; Glioblastoma multiforme; Intramedullary; Conus medullaris
1. Introduction Astrocytoma is the most common primary tumour of the central nervous system.1 Intramedullary astrocytomas *
Corresponding author. Tel.: +91 22 24129884; fax: +91 22 24143435. E-mail addresses: [email protected] (S. Balasubramaniam).
of the spinal cord are not as common as intracranial astrocytomas. Intramedullary astrocytomas are generally of low grade,1,2 and high-grade astrocytomas are comparatively rare in the spinal cord. The ratio of low-grade to highgrade astrocytomas in the spinal cord is about 3:1.2,3 Glioblastoma multiforme accounts for about 1–3% of all
Physicians should be aware of the possibility of DP when prescribing TPM. We recommend careful monitoring of these patients and discontinuation of TPM if DP develops. References 1. Lyell A. Delusions of parasitosis. Br J Dermatol 1983;108:485–99. 2. Trabert W. 100 years of delusional parasitosis. Meta-analysis of 1,223 case reports. Psychopathology 1995;28:238–46.
599
3. Mula M, Trimble MR, Lhatoo SD, et al. Topiramate and psychiatric adverse events in patients with epilepsy. Epilepsia 2003;44: 659–63. 4. Trimble MR. New antiepileptic drugs and psychopathology. Neuropsychobiology 1998;38:149–51. 5. De Leon J, Antelo RE, Simpson G. Delusion of parasitosis or chronic tactile hallucinosis: hypothesis about their brain physiopathology. Compr Psychiatry 1992;33:25–33. 6. Berrios GE. Delusional parasitosis and physical disease. Compr Psychiatry 1985;26:395–403.
doi:10.1016/j.jocn.2006.12.017
Hemichorea, an unusual complication of ventriculoperitoneal shunt Likhith M. Alakandy *, Ramanathan Venkatachalam Iyer, Aprajay Golash Department of Neurosurgery, Royal Preston Hospital, Sharoe Green Lane, Fulwood, Preston PR2 9HT, Lancashire, UK Received 28 October 2006; accepted 6 December 2006
Abstract We report an unusual case of left hemichorea in a 24-year-old male patient that followed the insertion of a right ventriculoperitoneal shunt. This completely resolved after removal of the shunt. A review of the available literature and the possible aetiopathogenesis is discussed. Ó 2007 Elsevier Ltd. All rights reserved. Keywords: Hemichorea; Dyskinaesia; Ventriculoperitoneal shunt
1. Introduction Movement disorders are extremely rare complications of ventriculoperitoneal (VP) shunts. Contralateral parkinsonism and ballism have been reported following the insertion of ventricular catheters,1–3 and in all these patients, the symptoms resolved after removal of the shunt. Focal lesions in the basal ganglia typically produce contralateral chorea or hemiballism, whereas generalised injury may cause generalised chorea or ballism. The exact mechanism of reversible dyskinesia following malposition of ventricular catheters is not known. In this report, we describe such a patient presenting with hemichorea as a complication of VP shunt and discuss the possible aetiopathogenesis. 2. Case report A 24-year-old male patient presented at the age of 16 with a 5-month history of deteriorating visual acuity, diplopia and episodic loss of consciousness. He had papillo*
Corresponding author. Tel.: +44 7970741793; fax: +44 141 201 2020. E-mail addresses: [email protected] (L.M. Alakandy).
edema and bilateral sixth cranial nerve palsies. There was a history of meningococcal meningitis at the age of 2 years from which he had recovered completely without any residual deficits. CT scan showed obstructive hydrocephalus and aqueduct stenosis, presumably as a result of childhood meningitis. A right-sided medium pressure VP shunt was inserted following which there was satisfactory clinical improvement. He presented again 7 years later with a 3week history of headaches, diplopia and blurring of vision. Radiological examination of the shunt showed fracture of the shunt tube in the chest wall and distal migration of the peritoneal end. CT scan of the brain showed ventriculomegaly. The shunt was revised on the right side and he had complete resolution of his headaches and visual disturbance. Two weeks after revision of the shunt, he developed a choreiform movement of the left side of his body. These movements were rapid, irregular and non-purposeful, predominantly involving the upper limb. CT scan revealed the shunt catheter to be traversing the right basal ganglia with its tip in the third ventricle (Figs. 1 and 2). He refused to have the shunt revised and commenced medical treatment, which failed to control the dyskinesia. A CT scan 4 weeks
600
Case Report / Journal of Clinical Neuroscience 15 (2008) 599–601
later showed a right-sided chronic subdural haematoma in addition to a reduction in the size of the ventricles. The hemichorea persisted but he had no symptoms relating to the subdural haematoma. He agreed to surgical intervention. The shunt was removed and the subdural haematoma evacuated through a right temporal burr-hole. The choreiform movements completely resolved in a few days following this and to date he has not required reinsertion of the shunt. 3. Discussion
Fig. 1. Axial CT scan showing the shunt to be traversing the right thalamus.
Fig. 2. Axial CT scan showing the tip of the shunt in the third ventricle.
The most common lesions associated with hyperkinetic movement disorders are ischaemic stroke and multiple sclerosis.4,5 They can also rarely present as complications of a shunt. In our patient, local trauma or irritation might seem to be the obvious explanation for this. However, this is not always the case as highlighted in a report by Walker et al.2 He described a 68-year-old female patient who developed bilateral ballism following insertion of a right frontal VP shunt for normal pressure hydrocephalus. The position of the shunt catheter tip was confirmed to be intraventricular. On removal of the shunt, the ballism resolved after a few days. She later underwent another VP shunt, this time through a right parietal burr-hole and developed the same movement disorder, which resolved a few weeks after removal of the shunt. Cheshire et al.1 reported a 28-yearold female patient who developed unilateral tremor, rigidity and paraesthesia following administration of methotrexate via an Ommaya reservoir that was inadvertently placed too deep into the contralateral mesencephalon. Gooskens et al.3 reported a young girl, who developed voluntary movement myoclonus of the contralateral hand as a result of a granulomatous reaction to a shunt tip in the thalamus. These two reports suggest disruption of the neuronal network within the basal ganglia. Chorea, which is characterised by abrupt involuntary movements, is known to result from dysfunction within the complex network interconnecting motor cortical areas and the basal ganglia. Focal damage to the caudate nucleus, putamen, globus pallidus as well as associated structures like the subthalamic nucleus and substantia nigra can all lead to choreic disorders.6,7 In the last two decades, there has been considerable progress in understanding of the pathways responsible for unwanted movements. From primate models of experimental hyperkinetic syndromes, it can be suggested that contralateral hyperkinetic disorders result from disinhibition of the thalamic nuclei due to a reduction of tonic inhibitory output from the globus pallidus interna. This may result in the inability to suppress unwanted responses to the cortical sensory stimuli, resulting in chorea.8 Other movement disorders, including parkinsonism, have been reported to occur as a result of obstructive hydrocephalus.9 However, this is not always reversible. Apart from local damage due to a malpositioned shunt catheter, damage to the basal ganglia in hydrocephalus may be a result of direct pressure or chronic impairment of
Case Report / Journal of Clinical Neuroscience 15 (2008) 601–603
blood flow. When this is the case, the symptoms tend to improve with time after removal of the offending cause.10,11 However, in our patient the hemichorea did not appear for 2 weeks after the shunt was placed, and persisted for a few days after the shunt was removed. This makes local irritation an unlikely aetiology, where the symptoms should have manifested immediately. Decreased perfusion is also unlikely to be a valid explanation as the shunt was over-draining, as evidenced by the development of chronic subdural haematoma at his later presentation. Our patient has remained symptom-free at follow-up without a shunt. No other cause of the movement disorder was found. 4. Conclusion We report hemichorea as an extremely rare complication of ventriculoperitoneal shunts, and in the absence of structural damage appears to be reversible. The mechanism of this phenomenon is unknown. References
601
2. Walker FO, Hunt VP. Ballism: an association with ventriculoperitoneal shunting. Neurology 1990;40:1004. 3. Gooskens RH, Kaiser MC, Veiga-Pires JA. Computerised tomography in the management of complications due to ventricular shunting. Neuroradiology 1981;21:47–9. 4. O’Brien CF, Chorea. In: Jankovic J, Tolosa E, editors. Parkinson’s Disease and Movement Disorders. 3rd edition. Philadelphia: Lippincott Willimas and Wilkins; 1998. p. 357–64. 5. Alarcon F, Zijlmans JC, Duenas G, et al. Post-stroke movement disorders: report of 56 patients. J Neurol Neurosurg Psychiatry 2004;75:1568–74. 6. Slevin JT, Sparks DL, Dempsey RJ, et al. Altered striatal dopaminergic metabolism 36 hours after unilateral trauma to the human mesencephalon. Neurology 1987;37:322–5. 7. Bhatia KP, Marsden CD. The behavioural and motor consequences of focal lesions of the basal ganglia in man. Brain 1994;117:859–76. 8. DeLong MR. Primate models of movement disorders of basal ganglia origin. Trends Neurosci 1990;13:281–5. 9. Zeidler M, Dorman PJ, Ferguson IT, et al. Parkinsonism associated with obstructive hydrocephalus due to idiopathic aqueductal stenosis. J Neurol Neurosurg Psychiatry 1998;64:657–9. 10. Jankovic J, Newmark M, Peter P. Parkinsonism and acquired hydrocephalus. Move Disord 1986;1:59–64. 11. Shahar E, Lambert R, Hwang PA, et al. Obstructive hydrocephalus induced parkinsonism I: decreased basal ganglia regional blood flow. Pediatr Neurol 1988;4:117–9.
1. Cheshire WP, Ehle AL. Hemiparkinsonism as a complication of an Ommaya reservoir. Case report. J Neurosurg 1990;73:774–6. doi:10.1016/j.jocn.2006.12.016
Glioblastoma multiforme of the conus medullaris with holocordal spread Vivek Bonde, Srikant Balasubramaniam *, Atul Goel Department of Neurosurgery, R. No. 273, Seth G.S. Medical College and King Edward VII Memorial Hospital, Parel, Mumbai, Maharashtra 400 012, India Received 15 October 2006; accepted 1 December 2006
Abstract Glioblastoma multiforme of the conus medullaris is rare. It spreads both locally to contiguous structures and also by cerebrospinal seeding. The disease is aggressive and the ultimate prognosis is dismal. We report a case of conus medullaris glioblastoma multiforme with involvement of the holocord. In spite of excision of the conus medullaris tumor followed by radiation, the disease continued to progress rapidly. We discuss the rarity of the disease and the related literature with regard to treatment modalities. Ó 2007 Elsevier Ltd. All rights reserved. Keywords: Holocord; Astrocytoma; Glioblastoma multiforme; Intramedullary; Conus medullaris
1. Introduction Astrocytoma is the most common primary tumour of the central nervous system.1 Intramedullary astrocytomas *
Corresponding author. Tel.: +91 22 24129884; fax: +91 22 24143435. E-mail addresses: [email protected] (S. Balasubramaniam).
of the spinal cord are not as common as intracranial astrocytomas. Intramedullary astrocytomas are generally of low grade,1,2 and high-grade astrocytomas are comparatively rare in the spinal cord. The ratio of low-grade to highgrade astrocytomas in the spinal cord is about 3:1.2,3 Glioblastoma multiforme accounts for about 1–3% of all