[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189] || Click here to download free Android application fo journal Letters to Editor
but arachnoid plain was well maintained, so dissecting out the tumor from carotid was easy. After substantial debulking of the mass, the third nerve appeared from the interpeduncular cistern as a bundle and ran straight into the base of the mass. Complete removal of the tumor was done [Figure 3]. Preservation of nerve was tried but could not be preserved and was sacrificed. Postoperatively, the patient developed complete third nerve paralysis on the left side. Histopathology and immunohistochemistry confirmed the diagnosis of schwannoma [Figure 4]. During one year follow‑up, her hormonal status had improved without radiological evidence of recurrence.
space where these slow‑growing tumors expand to a large size before they produce symptoms. Preserving function of the third nerve in these large tumors can be a challenge (whether complete or partial resections done), as confirmed in previous reports.[5,6]
Kovacs, [3] who in 1927, described an isolated oculomotor nerve schwannoma observed during an autopsy, was probably the first to report such a tumor. Celli et al [4] divided oculomotor cranial nerve schwannomas into three groups: (1) Cisternal, (2) cisternocavernous and (3) cavernous lesions. This classification was based on the preferred extension of these tumors. Our case belongs to the cisternal type. The explanation for this large tumor could be that the ventral cistern of the brainstem is a potential
References
Satya Bhusan Senapati, Sudhansu Sekhar Mishra, Srikanta Das, Deepak Kumar Parida Department of Neurosurgery, Shrirama Chandra Bhanja Medical College and Hospital, Cuttack, Odisha, India E‑mail: [email protected]
1. Saetia K, Larbcharoensub N, Wetchagama N.Oculomotor nerve schwannoma: A case report and review of the literature. J Med Assoc Thai 2011;94:1002‑7. 2. Prabhu SS, Bruner JM. Large oculomotor schwannoma presenting as a parasellar mass: A case report and literature review. Surg Neurol Int 2010;1:15. 3. Kovacs W. Ueber ein solitares neuinom des nervus oculomotorius. Zentralbl Allg Pathol 1927;40:518‑22. 4. Celli P, Ferrante L, Acqui M, Mastronardi L, Fortuna A, Palma L. Neurinoma of the third, fourth, and sixth cranial nerves: A survey and report of a new fourth nerve case. Surg Neurol 1992;38:216‑24. 5. Kachhara R, Nair S, Radhakrishnan VV. Oculomotor nerve neurinoma: Report of two cases. Acta Neurochir (Wien) 1998;140:1147‑51. 6. Mehta VS, Singh RV, Misra NK, Choudhary C. Schwannoma of the oculomotor nerve. Br J Neurosurg 1990;4:69‑72. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.144459
a
b
Figure 3: Postoperative axial and coronal contrast CT scans showing a complete resection of the lesion
Received: 29-07-2014 Review completed: 03-10-2014 Accepted: 03-10-2014
Pilomyxoid astrocytoma in an elderly patient: A case report and review of literature Sir, Pilomyxoid astrocytomas (PMAs) are considered as the infantile variant of pilocytic astrocytoma (PA) with a more aggressive course. The occurrence of these tumours in the elderly population is very rare. We report a case of PMA of the thalamic region in a 62‑year‑old male. To the best of our knowledge, this is the third reported case in literature of PMA in an individual aged > 60 years. Figure 4: Histopathology showing compact Antoni A and loose Antoni B tissue, supporting schwannoma
Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5
Our patient presented with sudden onset of memory disturbances and behavioural alterations of 1‑week 549
[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for journal Letters to Editor
duration. On examination, he was conscious with stable vital parameters. There was no deficit. Systemic examination and routine investigations were within normal limits. MRI brain showed a heterogeneously enhancing mass lesion in the right thalamic region [Figure 1]. The patient underwent pericoronal parasagittal craniotomy and gross total resection of the mass. Postoperative period was unremarkable. Microscopic examination of the submitted tissue showed a predominantly myxoid neoplasm containing monomorphic population of bipolar cells arranged in a predominant angiocentric pattern [Figure 2]. No Rosenthal fibres or eosinophilic granular bodies were seen. Glomeruloid vascular tufts were seen; however, no areas of necrosis or increased mitotic activity were noted. Ki‑67 index was 4%. A diagnosis of PMA, WHO grade II, was established. There was no evidence of dissemination into cerebrospinal fluid. Hence, no further treatment was given, and the patient was kept under regular follow‑up.
The term ‘PMA’was first introduced by Tihan et al., in 1999 as a more aggressive variant of PA.[1] It was included as a separate entity in the 2007 WHO Classification of Tumours of the Central Nervous System. Both PMA and PA shows predilection for optico-chiasmatic and hypothalamic regions The mean age of the patients with PMA at the time of diagnosis is 18 months, whereas that of patients with PA is 58 months.[2] Komotar et al., first reported a case of PMA outside the paediatric age group in a 28‑year‑old male.[3] Since then, there have been a few exceptional case reports of PMA in adults. We found only two case reports of PMA in individuals aged > 60 years. Skovrlj et al.[2] reported a case of PMA of the fourth ventricle in a 72‑year‑old male and Toyoda et al.[4] described PMA of the basal cistern in a 77‑year‑ old male. Komotar et al. compared the long-term outcomes in age and location matched patients diagnosed with PMA and PA and found higher rates of local recurrence, cerebrospinal fluid dissemination and shorter survival times for patients with PMA than those with PA.[5] Currently, there are no consensus guidelines on the management of PMAs. Wherever possible, gross total resection (GTR) is the primary treatment strategy, as GTR has been shown to be the most reliable predictor of outcome in children with low‑grade gliomas where surgery can be performed without excessive morbidity.[2] Owing to the young age of patients diagnosed with PMA, adjuvant chemo‑or radiotherapy is restricted to cases with subtotal excision or recurrence.[5] Taking this into consideration, our patient also did not receive any adjuvant therapy after GTR and is being kept under regular follow‑up.
Figure 1: MRI brain shows a heterogeneously enhancing mass lesion in the right thalamic region
Reporting of more such cases is needed to define the epidemiology of these tumours and to formulate management guidelines in the adult patients who can withstand a more aggressive treatment.
R. Amita, S. Sandhyamani, Suresh Nair1, T. R. Kapilamoorthy2
a
Departments of Pathology, 1Neurosurgery, 2Imaging Sciences and Interventional Radiology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India E‑mail: [email protected]
b
References 1. 2. c Figure 2: (a) A glial neoplasm, with abundant myxoid stroma. (b) Bipolar cells arranged in an angiocentric pattern. (c) Glomeruloid vascular tufts. (a, b and c: H and E, 0078100)
550
3.
Omura T, Nawashiro H, Osada H, Shima K, Tsuda H, Shinsuke A. Pilomyxoid astrocytoma of the fourth ventricle in an adult. Acta Neurochir (Wien) 2008;150:1203‑6. Skovrlj B, Pain M, Bederson JB, Fowkes M. Pilomyxoid astrocytoma of the cerebellar vermis in an elderly patient. Surg Neurol Int 2014;5:29. Komotar RJ, Mocco J, Zacharia BE, Wilson DA, Kim PY, Canoll PD, et al. Astrocytoma with pilomyxoid features presenting in an adult. Neuropathology 2006;26:89‑93. Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5
[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for journal Letters to Editor
and treated early.[1] It is extremely difficult to diagnosis this fungal infection even with newer radiological modalities.
4. Toyoda K, Tsutsumi K, Ono T, Takahata H, Toda K, Ito M, et al. Pilomyxoid astrocytoma presenting in an elderly patient: A case report. No Shinkei Geka 2009;37:1123‑8. 5. Komotar RJ, Mocco J, Jones JE, Zacharia BE, Tihan T, Feldstein NA, et al. Pilomyxoid astrocytoma: Diagnosis, prognosis, and management. Neurosurg Focus 2005;18:E7.
A 42‑year‑old male, cook by profession and apparently immunocompetent presented with headache and altered sensorium of 2 weeks duration. Examination was unremarkable except for bilateral mild papilledema. Magnetic resonance imaging (MRI) showed a T1‑weighted hypointense lesion in right parietal region. The lesion was intensively contrast enhancing with multiple small lesions surrounding the main lesion [Figure 1]. It was hyperintense on T2‑weighted sequence and there was restriction on diffusion‑weighted imaging (DWI) [Figure 1]. In view of altered sensorium he was taken up for surgery with a preoperative diagnosis of high grade malignancy. During surgery, aspiration of the lesion showed pus. The abscess wall was removed in total after defining a clear plane of cleavage between brain and abscess. The wall of abscess had multiple blackish spots. The frozen section of abscess wall revealed granulomas and multinucleated giant cells containing pigmented fungi. Loading dose of amphotericin B was given intraoperatively. Biopsy report confirmed the diagnosis of fungal infection, pheohyphomycosis [Figure 2]. Patient has received 1,500 mg. of amphotericin B and is on oral voriconazole, doing well at follow‑up.
Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.144460
Received: 04-09-2014 Review completed: 14-10-2014 Accepted: 03-10-2014
A case of cerebral pheohyphomycosis in an immunocompetent patient: Emphasis on intraoperative findings Sir, Cerebral pheohyphomycosis is a fungal infection caused by dematiaceous septate mycelial elements and is associated with a very high mortality unless diagnosed
a
e
The term pheohyphomycosis means dark pigmented fungal hyphae.[2] This infection is caused in majority of cases by a single order Chaetothyriales. About 50% patients
c
b
f
d
g
h
Figure 1: (a) NCCT head showing hypodense lesion in right parietooccipital region with surrounding perilesional edema. (b) MRI plain T1WI axial showing hypointense lesion and (c) ring enhancement on contrast. (d) MRI T2WI axial showing hyperintense lesion with perilesional edema. (e) MRI contrast T1WI sagittal showing ring enhancing lesion and a few satellite lesions. (f) MRI FLAIR coronal image showing almost isointense lesion with surrounding edema. (g) MRI DWI showing restriction of diffusion and (h) postoperative NCCT head showing complete excision of lesion. NCCT = Noncontrast computerized tomography, MRI = magnetic resonance imaging, T1WI = T1-weighted imaging, T2WI = T2-weighted imaging, FLAIR = fluid-attenuated inversion recovery, DWI = diffusion-weighted imaging
Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5
551
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.
but arachnoid plain was well maintained, so dissecting out the tumor from carotid was easy. After substantial debulking of the mass, the third nerve appeared from the interpeduncular cistern as a bundle and ran straight into the base of the mass. Complete removal of the tumor was done [Figure 3]. Preservation of nerve was tried but could not be preserved and was sacrificed. Postoperatively, the patient developed complete third nerve paralysis on the left side. Histopathology and immunohistochemistry confirmed the diagnosis of schwannoma [Figure 4]. During one year follow‑up, her hormonal status had improved without radiological evidence of recurrence.
space where these slow‑growing tumors expand to a large size before they produce symptoms. Preserving function of the third nerve in these large tumors can be a challenge (whether complete or partial resections done), as confirmed in previous reports.[5,6]
Kovacs, [3] who in 1927, described an isolated oculomotor nerve schwannoma observed during an autopsy, was probably the first to report such a tumor. Celli et al [4] divided oculomotor cranial nerve schwannomas into three groups: (1) Cisternal, (2) cisternocavernous and (3) cavernous lesions. This classification was based on the preferred extension of these tumors. Our case belongs to the cisternal type. The explanation for this large tumor could be that the ventral cistern of the brainstem is a potential
References
Satya Bhusan Senapati, Sudhansu Sekhar Mishra, Srikanta Das, Deepak Kumar Parida Department of Neurosurgery, Shrirama Chandra Bhanja Medical College and Hospital, Cuttack, Odisha, India E‑mail: [email protected]
1. Saetia K, Larbcharoensub N, Wetchagama N.Oculomotor nerve schwannoma: A case report and review of the literature. J Med Assoc Thai 2011;94:1002‑7. 2. Prabhu SS, Bruner JM. Large oculomotor schwannoma presenting as a parasellar mass: A case report and literature review. Surg Neurol Int 2010;1:15. 3. Kovacs W. Ueber ein solitares neuinom des nervus oculomotorius. Zentralbl Allg Pathol 1927;40:518‑22. 4. Celli P, Ferrante L, Acqui M, Mastronardi L, Fortuna A, Palma L. Neurinoma of the third, fourth, and sixth cranial nerves: A survey and report of a new fourth nerve case. Surg Neurol 1992;38:216‑24. 5. Kachhara R, Nair S, Radhakrishnan VV. Oculomotor nerve neurinoma: Report of two cases. Acta Neurochir (Wien) 1998;140:1147‑51. 6. Mehta VS, Singh RV, Misra NK, Choudhary C. Schwannoma of the oculomotor nerve. Br J Neurosurg 1990;4:69‑72. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.144459
a
b
Figure 3: Postoperative axial and coronal contrast CT scans showing a complete resection of the lesion
Received: 29-07-2014 Review completed: 03-10-2014 Accepted: 03-10-2014
Pilomyxoid astrocytoma in an elderly patient: A case report and review of literature Sir, Pilomyxoid astrocytomas (PMAs) are considered as the infantile variant of pilocytic astrocytoma (PA) with a more aggressive course. The occurrence of these tumours in the elderly population is very rare. We report a case of PMA of the thalamic region in a 62‑year‑old male. To the best of our knowledge, this is the third reported case in literature of PMA in an individual aged > 60 years. Figure 4: Histopathology showing compact Antoni A and loose Antoni B tissue, supporting schwannoma
Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5
Our patient presented with sudden onset of memory disturbances and behavioural alterations of 1‑week 549
[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for journal Letters to Editor
duration. On examination, he was conscious with stable vital parameters. There was no deficit. Systemic examination and routine investigations were within normal limits. MRI brain showed a heterogeneously enhancing mass lesion in the right thalamic region [Figure 1]. The patient underwent pericoronal parasagittal craniotomy and gross total resection of the mass. Postoperative period was unremarkable. Microscopic examination of the submitted tissue showed a predominantly myxoid neoplasm containing monomorphic population of bipolar cells arranged in a predominant angiocentric pattern [Figure 2]. No Rosenthal fibres or eosinophilic granular bodies were seen. Glomeruloid vascular tufts were seen; however, no areas of necrosis or increased mitotic activity were noted. Ki‑67 index was 4%. A diagnosis of PMA, WHO grade II, was established. There was no evidence of dissemination into cerebrospinal fluid. Hence, no further treatment was given, and the patient was kept under regular follow‑up.
The term ‘PMA’was first introduced by Tihan et al., in 1999 as a more aggressive variant of PA.[1] It was included as a separate entity in the 2007 WHO Classification of Tumours of the Central Nervous System. Both PMA and PA shows predilection for optico-chiasmatic and hypothalamic regions The mean age of the patients with PMA at the time of diagnosis is 18 months, whereas that of patients with PA is 58 months.[2] Komotar et al., first reported a case of PMA outside the paediatric age group in a 28‑year‑old male.[3] Since then, there have been a few exceptional case reports of PMA in adults. We found only two case reports of PMA in individuals aged > 60 years. Skovrlj et al.[2] reported a case of PMA of the fourth ventricle in a 72‑year‑old male and Toyoda et al.[4] described PMA of the basal cistern in a 77‑year‑ old male. Komotar et al. compared the long-term outcomes in age and location matched patients diagnosed with PMA and PA and found higher rates of local recurrence, cerebrospinal fluid dissemination and shorter survival times for patients with PMA than those with PA.[5] Currently, there are no consensus guidelines on the management of PMAs. Wherever possible, gross total resection (GTR) is the primary treatment strategy, as GTR has been shown to be the most reliable predictor of outcome in children with low‑grade gliomas where surgery can be performed without excessive morbidity.[2] Owing to the young age of patients diagnosed with PMA, adjuvant chemo‑or radiotherapy is restricted to cases with subtotal excision or recurrence.[5] Taking this into consideration, our patient also did not receive any adjuvant therapy after GTR and is being kept under regular follow‑up.
Figure 1: MRI brain shows a heterogeneously enhancing mass lesion in the right thalamic region
Reporting of more such cases is needed to define the epidemiology of these tumours and to formulate management guidelines in the adult patients who can withstand a more aggressive treatment.
R. Amita, S. Sandhyamani, Suresh Nair1, T. R. Kapilamoorthy2
a
Departments of Pathology, 1Neurosurgery, 2Imaging Sciences and Interventional Radiology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India E‑mail: [email protected]
b
References 1. 2. c Figure 2: (a) A glial neoplasm, with abundant myxoid stroma. (b) Bipolar cells arranged in an angiocentric pattern. (c) Glomeruloid vascular tufts. (a, b and c: H and E, 0078100)
550
3.
Omura T, Nawashiro H, Osada H, Shima K, Tsuda H, Shinsuke A. Pilomyxoid astrocytoma of the fourth ventricle in an adult. Acta Neurochir (Wien) 2008;150:1203‑6. Skovrlj B, Pain M, Bederson JB, Fowkes M. Pilomyxoid astrocytoma of the cerebellar vermis in an elderly patient. Surg Neurol Int 2014;5:29. Komotar RJ, Mocco J, Zacharia BE, Wilson DA, Kim PY, Canoll PD, et al. Astrocytoma with pilomyxoid features presenting in an adult. Neuropathology 2006;26:89‑93. Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5
[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189] || Click here to download free Android application for journal Letters to Editor
and treated early.[1] It is extremely difficult to diagnosis this fungal infection even with newer radiological modalities.
4. Toyoda K, Tsutsumi K, Ono T, Takahata H, Toda K, Ito M, et al. Pilomyxoid astrocytoma presenting in an elderly patient: A case report. No Shinkei Geka 2009;37:1123‑8. 5. Komotar RJ, Mocco J, Jones JE, Zacharia BE, Tihan T, Feldstein NA, et al. Pilomyxoid astrocytoma: Diagnosis, prognosis, and management. Neurosurg Focus 2005;18:E7.
A 42‑year‑old male, cook by profession and apparently immunocompetent presented with headache and altered sensorium of 2 weeks duration. Examination was unremarkable except for bilateral mild papilledema. Magnetic resonance imaging (MRI) showed a T1‑weighted hypointense lesion in right parietal region. The lesion was intensively contrast enhancing with multiple small lesions surrounding the main lesion [Figure 1]. It was hyperintense on T2‑weighted sequence and there was restriction on diffusion‑weighted imaging (DWI) [Figure 1]. In view of altered sensorium he was taken up for surgery with a preoperative diagnosis of high grade malignancy. During surgery, aspiration of the lesion showed pus. The abscess wall was removed in total after defining a clear plane of cleavage between brain and abscess. The wall of abscess had multiple blackish spots. The frozen section of abscess wall revealed granulomas and multinucleated giant cells containing pigmented fungi. Loading dose of amphotericin B was given intraoperatively. Biopsy report confirmed the diagnosis of fungal infection, pheohyphomycosis [Figure 2]. Patient has received 1,500 mg. of amphotericin B and is on oral voriconazole, doing well at follow‑up.
Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.144460
Received: 04-09-2014 Review completed: 14-10-2014 Accepted: 03-10-2014
A case of cerebral pheohyphomycosis in an immunocompetent patient: Emphasis on intraoperative findings Sir, Cerebral pheohyphomycosis is a fungal infection caused by dematiaceous septate mycelial elements and is associated with a very high mortality unless diagnosed
a
e
The term pheohyphomycosis means dark pigmented fungal hyphae.[2] This infection is caused in majority of cases by a single order Chaetothyriales. About 50% patients
c
b
f
d
g
h
Figure 1: (a) NCCT head showing hypodense lesion in right parietooccipital region with surrounding perilesional edema. (b) MRI plain T1WI axial showing hypointense lesion and (c) ring enhancement on contrast. (d) MRI T2WI axial showing hyperintense lesion with perilesional edema. (e) MRI contrast T1WI sagittal showing ring enhancing lesion and a few satellite lesions. (f) MRI FLAIR coronal image showing almost isointense lesion with surrounding edema. (g) MRI DWI showing restriction of diffusion and (h) postoperative NCCT head showing complete excision of lesion. NCCT = Noncontrast computerized tomography, MRI = magnetic resonance imaging, T1WI = T1-weighted imaging, T2WI = T2-weighted imaging, FLAIR = fluid-attenuated inversion recovery, DWI = diffusion-weighted imaging
Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5
551
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.
