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3. 4.
Adachi K, Yoshida K, Miwa T, Ikeda E, Kawase T. Olfactory schwannoma. Acta Neurochir (Wien) 2007;149:605-10. Yako K, Morita A, Ueki K, Kirino T. Subfrontal schwannoma. Acta Neurochir (Wien) 2005;147:655-7. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128353
Received: 08-01-2014 Review completed: 08-01-2014 Accepted: 31-01-2014
Intravenous immunoglobulin induced meningoencephalitis Sir, Adverse effects to intravenous immunoglobulin (IVIG) are uncommon and aseptic meningitis and encephalopathy have been rarely reported with IVIG.[1-6] We here report a case of IVIG induced meningoencephalitis. A 43-year-old female patient was evaluated for worsening of chronic headache following a respiratory tract infection. Magnetic resonance imaging (MRI) brain was normal. Cerebrospinal fluid (CSF) analysis showed mild elevated proteins (74 mg/dl) with normal sugar and cell (2/cumm) count. She subsequently developed post-lumbar puncture headache and left facial paresis and was started on steroids. Five days later, she presented with difficulty in walking and paraparesis with normal bowel and bladder function. Examination revealed left lower motor neuron facial paresis, quadriparesis (Grade 4/5), sluggish/absent deep tendon reflexes and bilaterally
a
down going plantar response. She remained afebrile. Blood investigations including electrolytes were normal. Nerve conduction study showed motor demyelinating polyneuropathy consistent with acute inflammatory demyelinating polyneuropathy. She worsened in weakness over the next 48 h requiring assistance to ambulate and was started on IVIG (2 g/kg over 5 days). After receiving 55 g of IVIG, she developed altered mental state, right hemineglect and right sided paucity of movements. She remained afebrile and had no meningeal signs. Repeat MRI brain including diffusion sequences was normal. Electroencephalogram (EEG) showed left hemispheric slowing, high amplitude rhythmic delta activity [Figure 1a]. Repeat CSF showed elevated proteins (129 mg %) normal sugar (62 mg/dl vs. blood glucose 129 mg/dl) and 16 cells/cumm (predominant lymphocytes). Gram stain was normal and culture grew no organism. CSF serology for common virus and bacterial infections was negative. Liver functions, ammonia and electrolytes were also normal. Provisional diagnosis of IVIG induced meningoencephalitis was considered and IVIG was discontinued. She improved gradually in sensorium over the next 48 h and EEG repeated 5 days later was normal [Figure 1b]. Motor weakness recovered completely by 4 weeks. She remains asymptomatic and is under follow-up. Adverse effects with the use IVIG are very rare and reported in less than 5% of patients.[1] The most common adverse effects occur soon after or during infusions and include headache, chills, myalgia, wheezing, tachycardia, low backache, nausea and hypotension. The other serious adverse effects include anaphylactic reactions, thromboembolic events and acute renal failure. Only a few cases of aseptic meningitis with IVIG have been reported.[2,3] History of migraine regardless of the type of commercial preparation or the infusion rate increases the risk for aseptic meningitis.[3] Majority of patients
b
Figure 1: Electroencephalogram (EEG) in bipolar anterior-posterior montage showing left hemispheric attenuation and high amplitude rhythmic delta activity during the initial diagnosis of menigoencephalitis (a - arrows). Five days later, EEG shows resolution of the abnormalities with appearance of symmetrical background activity (b - arrows) (filter - 1-70 Hz)
98
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
[Downloaded free from http://www.neurologyindia.com on Thursday, March 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for this journal Letters to Editor
develop signs and symptoms within 48 h of IVIG infusion. Very rarely IVIG may cause posterior reversible encephalopathy.[4] The exact mechanism of IVIG induced aseptic meningitis/ encephalopathy is not known. Immunoglobulin G can penetrate the meninges and brain parenchyma and usually remains in the CSF for 48 h post-termination of infusion. Several factors have been implicated in the pathogenesis of meningitis/encephalopathy: Direct inflammatory response to the IVIG, cerebrovascular reactivity, hypersensitivity response and cytokines release.[5] Wada et al. demonstrated the excitotoxic effect of IVIG, increase in glutamate and glutamine complex peak on MR spectroscopy in acute encephalopathy following IVIG.[6]
Conclusion Meningoencephalitis can occur as a very rare self-limiting complication of IVIG treatment.
Javeria Nooraine, Rajesh B. Iyer, S. Raghavendra Department of Neurology, Vikram Hospital, Bengaluru, Karnataka, India E-mail: [email protected]
References 1. 2. 3. 4. 5. 6.
Duhem C, Dicato MA, Ries F. Side-effects of intravenous immune globulins. Clin Exp Immunol 1994;97 Suppl 1:79-83. Kaarthigeyan K, Burli VV. Aseptic meningitis following intravenous immunoglobulin therapy of common variable immunodeficiency. J Pediatr Neurosci 2011;6:160-1. Sekul EA, Cupler EJ, Dalakas MC. Aseptic meningitis associated with high-dose intravenous immunoglobulin therapy: Frequency and risk factors. Ann Intern Med 1994;121:259-62. Voltz R, Rosen FV, Yousry T, Beck J, Hohlfeld R. Reversible encephalopathy with cerebral vasospasm in a Guillain-Barré syndrome patient treated with intravenous immunoglobulin. Neurology 1996;46:250-1. Preminger-Shapiro R, Nussinovitch M, Soen G, Varsano I. Aseptic meningitis: A frequent side-effect of intravenous immunoglobulin? Eur J Pediatr 1995;154:866-7. Wada A, Yoshida R, Oda K, Fukuba E, Uchida N, Kitagaki H. Acute encephalopathy associated with intravenous immunoglobulin therapy. AJNR Am J Neuroradiol 2005;26:2311-5.
Sixth nerve schwannoma Sir, Schwannomas represent approximately 7-10% of all primary intracranial tumors. The most frequent are in eight and fifth cranial nerves.[1] Schwannoma arising from the sixth cranial nerve is exceedingly rare. Due to its close proximity to fifth and eight nerves and similar clinical and radiological findings, it is difficult to diagnose sixth nerve schwannoma preoperatively.[2,3] A 42-year-old male presented with a history of diplopia on the left lateral gaze and intermittent headache for 6 months. There was no family history suggestive of neurofibromatosis. Neurological examination revealed left 6th nerve paralysis [Figure 1a]. Magnetic resonance imaging (MRI) brain revealed a left cerebellopontine angle lesion iso to hypointense on T1-WI [Figure 2a] hyperintense on T2-WI [Figure 2b] enhancing heterogeneously on contrast [Figure 2c]. The internal acoustic meatus was of normal size. A small enhancing component of the tumor was seen going toward the Dorello’s canal [Figure 2c arrow head]. Patient underwent left retromastoid suboccipital craniotomy and gross total excision of tumor under intraoperative cranial nerve monitoring. The tumor was arising from 6th cranial nerve. He had an uneventful recovery. At 3 months follow-up, the MRI brain was normal [Figure 2d] and sixth nerve paralysis improved to normal [Figure 1b]. Histopathological examination revealed a cellular tumor composed of spindle-shaped cells with oval-to-spindle nuclei and a moderate amount of cytoplasm. The tumor cells were arranged in palisading pattern and formed verocay bodies [Figure 3]. All the cranial nerves except first and second have covering myelin sheath composed of Schwann cells and are potential sites for developing schwannoma. Most common location of intracranial schwannoma is in the cerebello-pontine angle where they arise from the vestibular division of the eighth nerve. Other common
Access this article online Quick Response Code:
Website: www.neurologyindia.com
a
PMID: *** DOI: 10.4103/0028-3886.128354
b Received: 09-01-2014 Review completed: 09-01-2014 Accepted: 02-02-2014 Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
Figure 1: (a) Pre-operative and (b) post-operative clinical photograph
99
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.
3. 4.
Adachi K, Yoshida K, Miwa T, Ikeda E, Kawase T. Olfactory schwannoma. Acta Neurochir (Wien) 2007;149:605-10. Yako K, Morita A, Ueki K, Kirino T. Subfrontal schwannoma. Acta Neurochir (Wien) 2005;147:655-7. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128353
Received: 08-01-2014 Review completed: 08-01-2014 Accepted: 31-01-2014
Intravenous immunoglobulin induced meningoencephalitis Sir, Adverse effects to intravenous immunoglobulin (IVIG) are uncommon and aseptic meningitis and encephalopathy have been rarely reported with IVIG.[1-6] We here report a case of IVIG induced meningoencephalitis. A 43-year-old female patient was evaluated for worsening of chronic headache following a respiratory tract infection. Magnetic resonance imaging (MRI) brain was normal. Cerebrospinal fluid (CSF) analysis showed mild elevated proteins (74 mg/dl) with normal sugar and cell (2/cumm) count. She subsequently developed post-lumbar puncture headache and left facial paresis and was started on steroids. Five days later, she presented with difficulty in walking and paraparesis with normal bowel and bladder function. Examination revealed left lower motor neuron facial paresis, quadriparesis (Grade 4/5), sluggish/absent deep tendon reflexes and bilaterally
a
down going plantar response. She remained afebrile. Blood investigations including electrolytes were normal. Nerve conduction study showed motor demyelinating polyneuropathy consistent with acute inflammatory demyelinating polyneuropathy. She worsened in weakness over the next 48 h requiring assistance to ambulate and was started on IVIG (2 g/kg over 5 days). After receiving 55 g of IVIG, she developed altered mental state, right hemineglect and right sided paucity of movements. She remained afebrile and had no meningeal signs. Repeat MRI brain including diffusion sequences was normal. Electroencephalogram (EEG) showed left hemispheric slowing, high amplitude rhythmic delta activity [Figure 1a]. Repeat CSF showed elevated proteins (129 mg %) normal sugar (62 mg/dl vs. blood glucose 129 mg/dl) and 16 cells/cumm (predominant lymphocytes). Gram stain was normal and culture grew no organism. CSF serology for common virus and bacterial infections was negative. Liver functions, ammonia and electrolytes were also normal. Provisional diagnosis of IVIG induced meningoencephalitis was considered and IVIG was discontinued. She improved gradually in sensorium over the next 48 h and EEG repeated 5 days later was normal [Figure 1b]. Motor weakness recovered completely by 4 weeks. She remains asymptomatic and is under follow-up. Adverse effects with the use IVIG are very rare and reported in less than 5% of patients.[1] The most common adverse effects occur soon after or during infusions and include headache, chills, myalgia, wheezing, tachycardia, low backache, nausea and hypotension. The other serious adverse effects include anaphylactic reactions, thromboembolic events and acute renal failure. Only a few cases of aseptic meningitis with IVIG have been reported.[2,3] History of migraine regardless of the type of commercial preparation or the infusion rate increases the risk for aseptic meningitis.[3] Majority of patients
b
Figure 1: Electroencephalogram (EEG) in bipolar anterior-posterior montage showing left hemispheric attenuation and high amplitude rhythmic delta activity during the initial diagnosis of menigoencephalitis (a - arrows). Five days later, EEG shows resolution of the abnormalities with appearance of symmetrical background activity (b - arrows) (filter - 1-70 Hz)
98
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
[Downloaded free from http://www.neurologyindia.com on Thursday, March 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for this journal Letters to Editor
develop signs and symptoms within 48 h of IVIG infusion. Very rarely IVIG may cause posterior reversible encephalopathy.[4] The exact mechanism of IVIG induced aseptic meningitis/ encephalopathy is not known. Immunoglobulin G can penetrate the meninges and brain parenchyma and usually remains in the CSF for 48 h post-termination of infusion. Several factors have been implicated in the pathogenesis of meningitis/encephalopathy: Direct inflammatory response to the IVIG, cerebrovascular reactivity, hypersensitivity response and cytokines release.[5] Wada et al. demonstrated the excitotoxic effect of IVIG, increase in glutamate and glutamine complex peak on MR spectroscopy in acute encephalopathy following IVIG.[6]
Conclusion Meningoencephalitis can occur as a very rare self-limiting complication of IVIG treatment.
Javeria Nooraine, Rajesh B. Iyer, S. Raghavendra Department of Neurology, Vikram Hospital, Bengaluru, Karnataka, India E-mail: [email protected]
References 1. 2. 3. 4. 5. 6.
Duhem C, Dicato MA, Ries F. Side-effects of intravenous immune globulins. Clin Exp Immunol 1994;97 Suppl 1:79-83. Kaarthigeyan K, Burli VV. Aseptic meningitis following intravenous immunoglobulin therapy of common variable immunodeficiency. J Pediatr Neurosci 2011;6:160-1. Sekul EA, Cupler EJ, Dalakas MC. Aseptic meningitis associated with high-dose intravenous immunoglobulin therapy: Frequency and risk factors. Ann Intern Med 1994;121:259-62. Voltz R, Rosen FV, Yousry T, Beck J, Hohlfeld R. Reversible encephalopathy with cerebral vasospasm in a Guillain-Barré syndrome patient treated with intravenous immunoglobulin. Neurology 1996;46:250-1. Preminger-Shapiro R, Nussinovitch M, Soen G, Varsano I. Aseptic meningitis: A frequent side-effect of intravenous immunoglobulin? Eur J Pediatr 1995;154:866-7. Wada A, Yoshida R, Oda K, Fukuba E, Uchida N, Kitagaki H. Acute encephalopathy associated with intravenous immunoglobulin therapy. AJNR Am J Neuroradiol 2005;26:2311-5.
Sixth nerve schwannoma Sir, Schwannomas represent approximately 7-10% of all primary intracranial tumors. The most frequent are in eight and fifth cranial nerves.[1] Schwannoma arising from the sixth cranial nerve is exceedingly rare. Due to its close proximity to fifth and eight nerves and similar clinical and radiological findings, it is difficult to diagnose sixth nerve schwannoma preoperatively.[2,3] A 42-year-old male presented with a history of diplopia on the left lateral gaze and intermittent headache for 6 months. There was no family history suggestive of neurofibromatosis. Neurological examination revealed left 6th nerve paralysis [Figure 1a]. Magnetic resonance imaging (MRI) brain revealed a left cerebellopontine angle lesion iso to hypointense on T1-WI [Figure 2a] hyperintense on T2-WI [Figure 2b] enhancing heterogeneously on contrast [Figure 2c]. The internal acoustic meatus was of normal size. A small enhancing component of the tumor was seen going toward the Dorello’s canal [Figure 2c arrow head]. Patient underwent left retromastoid suboccipital craniotomy and gross total excision of tumor under intraoperative cranial nerve monitoring. The tumor was arising from 6th cranial nerve. He had an uneventful recovery. At 3 months follow-up, the MRI brain was normal [Figure 2d] and sixth nerve paralysis improved to normal [Figure 1b]. Histopathological examination revealed a cellular tumor composed of spindle-shaped cells with oval-to-spindle nuclei and a moderate amount of cytoplasm. The tumor cells were arranged in palisading pattern and formed verocay bodies [Figure 3]. All the cranial nerves except first and second have covering myelin sheath composed of Schwann cells and are potential sites for developing schwannoma. Most common location of intracranial schwannoma is in the cerebello-pontine angle where they arise from the vestibular division of the eighth nerve. Other common
Access this article online Quick Response Code:
Website: www.neurologyindia.com
a
PMID: *** DOI: 10.4103/0028-3886.128354
b Received: 09-01-2014 Review completed: 09-01-2014 Accepted: 02-02-2014 Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
Figure 1: (a) Pre-operative and (b) post-operative clinical photograph
99
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.
