- Email: [email protected]
Giant cranionasal schwannoma
520
Case Reports / Journal of Clinical Neuroscience 17 (2010) 520–522
tissue plasminogen activator or anti-platelets agents may have suppressed further thrombosis during an endovascular maneuver, but this procedure carries a risk of re-bleeding following recanalization of the aneurysm, and hemorrhagic transformation in the superior trunk territory, which had already resulted in a complete infarction prior to the inferior trunk territory was affected. Balloon angioplasty at the M1–M2 bifurcation, close to the neck of the aneurysm would carry the risk of aneurysmal re-rupture. Coil embolization for the now angiographically occult thrombosed aneurysm had the potential of incomplete obliteration or distal migration of the intra-aneurysmal thrombus. The protruded thrombus from the aneurysm to the M1–M2 bifurcation was a cause of reduced inferior trunk blood flow with severe vasospasm; we removed the thrombus and reconstructed the tissue with aneurysmal clipping. Fortunately, the inferior trunk territory did not evolve into complete infarction due to a 6 hour
period between the ischemic event and surgical restoration of blood flow. This report highlights the importance of careful evaluation of ischemic patterns and aneurysm characteristics before choosing treatment modalities. Various therapeutic options for the ruptured aneurysm during vasospasm should be considered. References 1. Brisman JL, Roonprapunt C, Song JK, et al. Intentional partial coil occlusion followed by delayed clip application to wide-necked middle cerebral artery aneurysms in patients presenting with severe vasospasm. Report of two cases. J Neurosurg 2004;101:154–8. 2. Murayama Y, Song JK, Uda K, et al. Combined endovascular treatment for both intracranial aneurysm and symptomatic vasospasm. AJNR Am J Neuroradiol 2003;24:133–9. 3. Sugiu K, Katsumata A, Ono Y, et al. Angioplasty and coiling of ruptured aneurysm with symptomatic vasospasm: technical case report. Surg Neurol 2003;59:413–7.
doi:10.1016/j.jocn.2009.06.040
Giant cranionasal schwannoma Kai Zheng, Shu Jiang, Jian-guo Xu, Bo-yong Mao * Department of Neurosurgery, West China Hospital, Sichuan University, 37 Guoxuexiang Street, Chengdu, Sichuan, 610041, China
a r t i c l e
i n f o
Article history: Received 4 May 2009 Accepted 21 June 2009
Keywords: Cranionasal Schwannoma Skull base techniques Heterotopic
a b s t r a c t Giant schwannomas concomitantly involving the intracranial and nasal cavities are rare. We report a 22year-old male patient with a giant schwannoma involving the frontal skull base and extending into the nasal cavity. The patient had a 2-year history of nasal obstruction that was originally misdiagnosed as nasal polyps. A CT scan and an MRI revealed a large cranionasal tumor. Surgery was performed using a coronal incision with a bilateral frontal skull base extradural approach. The tumor originated from the anterior skull base and the dura and nasal mucosa were intact. Histopathology was consistent with a schwannoma. Schwannoma should be listed as part of the differential diagnosis of a cranionasal tumor and the surgical approach should be carefully selected to achieve total resection. Ó 2009 Elsevier Ltd. All rights reserved.
1. Introduction Intracranial schwannoma is a benign neoplasm arising from Schwann cells of the peripheral nerve sheath. It is most often associated with the eighth cranial nerve1,2 and next most commonly associated with the fifth and ninth cranial nerves. Schwannomas concomitantly involving the intracranial and nasal cavities are rare and present a surgical challenge. 2. Case report A 22-year-old healthy male without a history of neurofibromatosis presented with a nasal obstruction, which had slowly progressed over 2 years. The patient had previously been diagnosed with nasal polyps at a local otolaryngology clinic and underwent septoplasty for severe rhinocleisis without improvement. A CT scan * Corresponding author. Tel.: +86 028 85423622; fax: +86 028 85422411. E-mail address: [email protected] (B.-y. Mao).
showed a 6.6 7.3 10.5 cm space-occupying lesion in the nasal cavity with inferior extension into the ethmoid sinus and anterior extension into the frontal lobe (Fig. 1a,b). An MRI scan of the brain revealed a well-defined, encapsulated tumor with slightly decreased signal intensity on T1-weighted images and slightly increased signal intensity on T2-weighted images, which enhanced significantly after the administration of intravenous contrast media (Fig. 1c–e). Surgery was performed using a coronal incision and a bilateral frontal skull base extradural approach. Complete excision of the tumor was performed in collaboration with several otolaryngologists. During surgery, we found the tumor originated from the anterior skull base rather than the olfactory nerve. The tumor appeared vascular and was adherent to the dura; however, the nasal mucosa was intact. After careful removal of the tumor, the remaining 6 7 cm cranial defect was repaired using artificial dura. A postoperative contrast CT scan demonstrated complete resection of the tumor and satisfactory surgical reconstruction of the anterior skull
Case Reports / Journal of Clinical Neuroscience 17 (2010) 520–522
521
Fig. 1. (a) Pre-operative non-enhanced axial CT scan showing a large space-occupying lesion in the nasal cavity, ethmoid sinus and frontal lobe. (b) Pre-operative enhanced axial CT scan showing the tumor extending intracranially and compressing the frontal lobe. (c) Pre-operative T2-weighted axial MRI showing the tumor compressing the intracranial structures. (d) Pre-operative T1-weighted coronal MRI showing the tumor with a well-defined capsule with slightly lower signal intensity (insert: sagittal view). (e) Pre-operative sagittal MRI enhanced by gadolinium injection. (f) Non-enhanced axial CT scan performed postoperatively demonstrating total removal of the tumor.
base defect (Fig. 1f). At the 6-month follow-up no complications have occurred. Histologically, the tumor consisted of compact spindle cells with palisading neoplastic proliferation. Cellular areas with the appearance of Antoni A tissue, and loosely textured areas of rounded cells resembling Antoni B tissue, were noted focally on hematoxylin and eosin (H&E) staining (Supplementary Fig. 1a). Immunophenotypically, the neoplastic cells showed positive staining for S-100 antiserum (Supplementary Fig. 1b,c), negative staining for epithelial membrane antigen (Supplementary Fig. 1d) and glial fibrillary acidic protein (Supplementary Fig. 1e), and there were approximately 3–5% Ki-67 positive tumor cells (Supplementary Fig. 1f). 3. Discussion A typical intracranial schwannoma is a benign tumor arising from Schwann cells of the peripheral nerve sheath.2 As Schwann cells are present in the meningeal branch of the trigeminal nerve,3 and the glossopharyngeal nerve at the skull base and olfactory groove,4 this is the most likely origin of the tumor in our patient. Unusual sources for intracranial schwannoma include dysplasia of Schwann cells in a perivascular plexus,1 origin from a spinal dorsal root,5 transformation of mesoderm cells from the pia mater into Schwann cells,2 and ectopic Schwann cells due to abnormal embryonic development.6 Immunohistochemical studies showed positive staining for S-100, which is one of the characteristic markers of nerve sheath tumors. The schwannoma was also easily distinguished from meningioma and glioma by the negative staining for both EMA and GFAP.
Although schwannomas are primarily benign, one malignant intracerebral schwannoma has been reported.7 The growth fraction in malignant schwannomas, as determined by Ki-67/MIB-1 immunoreactivity, ranges from 5–65%. The 3–5% growth fraction in our patient’s tumor is consistent with borderline malignant potential. However, the additional features such as a complete capsule and slow growth of the tumor are more consistent with benign etiology. Close long-term clinical follow-up will be necessary to monitor for recurrence. CT scans and MRI are critical when choosing the appropriate surgical approach for resection of the tumor. Our surgical approach is commonly used for other tumors in a similar anatomical location, including olfactory neuroblastoma, adenocarcinoma, and squamous cell carcinoma. However, for tumors that invade the nasal cavity and paranasal sinuses, a combined craniofacial approach may be preferred. The addition of a lateral rhinotomy or mid-facial degloving approach could also extend the exposure of such a tumor.8 During surgery, collaboration with otolaryngologists is recommended to ensure successful total tumor resection.
Appendix A. Supplementary material Supplementary material associated with this article can be found, in the online version, at doi:10.1016/j.jocn.2009.06.039.
References 1. Adelman LS, Aronson SM. I nerve fiber and Schwann cell proliferation within the spinal cord (schwannosis). Neurology 1972;22:726–31. 2. Russel DS, Rubenstein LJ. Pathology of tumors of the nervous system. 5th ed. London, England: Edward Arnold; 1989.
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Case Reports / Journal of Clinical Neuroscience 17 (2010) 522–523
3. Guthikonda B, Theodosopoulos PV, van Loveren H, et al. Evolution in the assessment and management of trigeminal schwannoma. Laryngoscope 2008;118:195–203. 4. Erongun U, Ozkal E, Acar O, et al. Intracerebral schwannoma: case report and review. Neurosurg Rev 1996;19:269–74. 5. McCormick WF. Intramedullary spinal cord schwannoma. A unique case. Arch Pathol 1964;77:378–82.
6. Pardatselier K, Iraci G, Cappellotto P, et al. Multiple intramedullary neurinomas of the spinal cord. Case report. J Neurosurg 1979;50:817–22. 7. Gonzalez LF, Lekovic GP, Eschbacher J, et al. A true malignant schwannoma of the eighth cranial nerve: case report. Neurosurgery 2007;61:E421–2. 8. Zhao JC, Liu G, Zhang YT, et al. Microsurgery of skull base [In Chinese]. 1st ed. Tianjin, China: Science and Technology Translation Publishing Company; 2005.
doi:10.1016/j.jocn.2009.06.039
Intramedullary cysticercosis S. Kumar a, A. Handa a,*, S. Chavda a, R. Tiwari a, P. Abbey b a b
Department of Neurosurgery, St Stephen’s Hospital, Marg Tis Hazari, Delhi 110 054, India Department of Radiology, All India Institute of Medical Sciences, New Delhi, India
a r t i c l e
i n f o
Article history: Received 7 August 2008 Accepted 24 April 2009
Keywords: Intramedullary cysticercosis Magnetic resonance imaging Spinal cord compression Taenia solium
a b s t r a c t Intramedullary cysticercosis is a rare form of cysticercosis in the central nervous system. A 55-year-old woman was admitted with low back pain, urinary incontinence, paraparesis and sensory deficit. Her MRI showed a well-defined intramedullary cystic lesion at T7 vertebral level with peripheral enhancement on contrast. She underwent a T7–T8 laminectomy and excision of the lesion. Histopathology revealed the lesion to be a cysticercus granuloma. A postoperative course of albendazole was given. The patient showed significant neurological improvement at follow-up. Ó 2009 Elsevier Ltd. All rights reserved.
1. Introduction Cysticercosis, caused by Taenia solium, is one of the commonest parasitic diseases that involve the central nervous system. However, an intramedullary cysticercus granuloma is rare. Mohanty et al. reported a series of eight patients;1 otherwise there have been sporadic reports.2–5 In our patient the cyst came out easily from the surrounding well-defined granuloma, which is unusual. 2. Case report A 55-year-old female was admitted with low back pain of 1month duration, followed 2 days later by numbness and weakness in both lower limbs. She developed urinary incontinence 1 week prior to admission, for which she underwent catheterisation of the bladder. On examination she was bedridden with grade 1/5 spastic paraparesis in the right lower limb and grade 2/5 on the left side. All sensations were impaired by 50% to 60% below L1. Routine blood and urine investigations were normal. A plain radiograph of the thoracolumbar spine was within normal limits. MRI of the thoracolumbar spine, however, revealed a well-defined intramedullary cystic lesion at the T7 level with signal intensities resembling cerebrospinal fluid (CSF) and peripheral enhancement on gadolinium administration (Fig. 1). Intraoperatively the cord was found to be bulging and pale. Purulent fluid was drained via a midline myelotomy, following which a well-defined greyish white cyst was removed intact. A well-defined granuloma surrounding the cyst was dissected completely with a good plane from the cord tissue. There was no growth of any organism on microbiological culture of the fluid. Her postoperative course was uneventful. Histopathology confirmed the diagnosis of cysticercosis (Supplementary Fig. 1). She * Corresponding author. Tel.: +91 11 239 77 930; fax: +91 11 239 32 412. E-mail address: [email protected] (A. Handa).
was given a course of albendazole 400 mg twice daily for 3 weeks. At the 6-month follow-up, power in her lower limbs had improved to grade 4/5 with reduced spasticity and she could walk with support. Her bladder function had also returned to normal. 3. Discussion Cysticercosis is endemic to Brazil, Mexico, Korea and South Asian countries.1–3,6 Cysticercal involvement of the spinal cord is rare even in endemic areas and accounts for 0.7% to 5.85% of all patients.1,3–5,7 Among patients with spinal cysticercosis, intramedullary involvement is relatively uncommon, the commonest being in the intradural–extramedullary region. Extradural and vertebral locations have also been reported.1,4,5,8 About half the reported cases have concomitant extra spinal involvement.9 Singh and Sahai remarked that only 45 patients with intramedullary cysticercosis had been documented worldwide until 2004.4 The thoracic cord, which receives the maximum blood supply, is most commonly affected, followed by cervical, lumbar and sacral regions.10,11 Neurological symptoms in intramedullary cysticercosis can be attributed to direct mechanical compression, inflammatory changes, cord edema, gliosis, pachymeningitis or syrinx formation. The patient usually presents with progressive neurological worsening with spastic quadriparesis or paraparesis depending on the site of the lesion; sphincter involvement, impotence and sensory deficit may also be present. Neurological involvement may be acute. MRI normally shows a cystic lesion with signal intensities similar to CSF on both T1-weighted and T2-weighted MRI. The scolex when present, appears to be isointense or hyperintense on T1-weighted MRI. The cyst wall usually enhances on contrast administration. An associated syrinx is generally secondary to arachnoiditis, circulatory insufficiency or cord atrophy. In the absence of any extraspinal evidence of cysticercosis and in a setting of neurological deficits attributable to the lesion,
Case Reports / Journal of Clinical Neuroscience 17 (2010) 520–522
tissue plasminogen activator or anti-platelets agents may have suppressed further thrombosis during an endovascular maneuver, but this procedure carries a risk of re-bleeding following recanalization of the aneurysm, and hemorrhagic transformation in the superior trunk territory, which had already resulted in a complete infarction prior to the inferior trunk territory was affected. Balloon angioplasty at the M1–M2 bifurcation, close to the neck of the aneurysm would carry the risk of aneurysmal re-rupture. Coil embolization for the now angiographically occult thrombosed aneurysm had the potential of incomplete obliteration or distal migration of the intra-aneurysmal thrombus. The protruded thrombus from the aneurysm to the M1–M2 bifurcation was a cause of reduced inferior trunk blood flow with severe vasospasm; we removed the thrombus and reconstructed the tissue with aneurysmal clipping. Fortunately, the inferior trunk territory did not evolve into complete infarction due to a 6 hour
period between the ischemic event and surgical restoration of blood flow. This report highlights the importance of careful evaluation of ischemic patterns and aneurysm characteristics before choosing treatment modalities. Various therapeutic options for the ruptured aneurysm during vasospasm should be considered. References 1. Brisman JL, Roonprapunt C, Song JK, et al. Intentional partial coil occlusion followed by delayed clip application to wide-necked middle cerebral artery aneurysms in patients presenting with severe vasospasm. Report of two cases. J Neurosurg 2004;101:154–8. 2. Murayama Y, Song JK, Uda K, et al. Combined endovascular treatment for both intracranial aneurysm and symptomatic vasospasm. AJNR Am J Neuroradiol 2003;24:133–9. 3. Sugiu K, Katsumata A, Ono Y, et al. Angioplasty and coiling of ruptured aneurysm with symptomatic vasospasm: technical case report. Surg Neurol 2003;59:413–7.
doi:10.1016/j.jocn.2009.06.040
Giant cranionasal schwannoma Kai Zheng, Shu Jiang, Jian-guo Xu, Bo-yong Mao * Department of Neurosurgery, West China Hospital, Sichuan University, 37 Guoxuexiang Street, Chengdu, Sichuan, 610041, China
a r t i c l e
i n f o
Article history: Received 4 May 2009 Accepted 21 June 2009
Keywords: Cranionasal Schwannoma Skull base techniques Heterotopic
a b s t r a c t Giant schwannomas concomitantly involving the intracranial and nasal cavities are rare. We report a 22year-old male patient with a giant schwannoma involving the frontal skull base and extending into the nasal cavity. The patient had a 2-year history of nasal obstruction that was originally misdiagnosed as nasal polyps. A CT scan and an MRI revealed a large cranionasal tumor. Surgery was performed using a coronal incision with a bilateral frontal skull base extradural approach. The tumor originated from the anterior skull base and the dura and nasal mucosa were intact. Histopathology was consistent with a schwannoma. Schwannoma should be listed as part of the differential diagnosis of a cranionasal tumor and the surgical approach should be carefully selected to achieve total resection. Ó 2009 Elsevier Ltd. All rights reserved.
1. Introduction Intracranial schwannoma is a benign neoplasm arising from Schwann cells of the peripheral nerve sheath. It is most often associated with the eighth cranial nerve1,2 and next most commonly associated with the fifth and ninth cranial nerves. Schwannomas concomitantly involving the intracranial and nasal cavities are rare and present a surgical challenge. 2. Case report A 22-year-old healthy male without a history of neurofibromatosis presented with a nasal obstruction, which had slowly progressed over 2 years. The patient had previously been diagnosed with nasal polyps at a local otolaryngology clinic and underwent septoplasty for severe rhinocleisis without improvement. A CT scan * Corresponding author. Tel.: +86 028 85423622; fax: +86 028 85422411. E-mail address: [email protected] (B.-y. Mao).
showed a 6.6 7.3 10.5 cm space-occupying lesion in the nasal cavity with inferior extension into the ethmoid sinus and anterior extension into the frontal lobe (Fig. 1a,b). An MRI scan of the brain revealed a well-defined, encapsulated tumor with slightly decreased signal intensity on T1-weighted images and slightly increased signal intensity on T2-weighted images, which enhanced significantly after the administration of intravenous contrast media (Fig. 1c–e). Surgery was performed using a coronal incision and a bilateral frontal skull base extradural approach. Complete excision of the tumor was performed in collaboration with several otolaryngologists. During surgery, we found the tumor originated from the anterior skull base rather than the olfactory nerve. The tumor appeared vascular and was adherent to the dura; however, the nasal mucosa was intact. After careful removal of the tumor, the remaining 6 7 cm cranial defect was repaired using artificial dura. A postoperative contrast CT scan demonstrated complete resection of the tumor and satisfactory surgical reconstruction of the anterior skull
Case Reports / Journal of Clinical Neuroscience 17 (2010) 520–522
521
Fig. 1. (a) Pre-operative non-enhanced axial CT scan showing a large space-occupying lesion in the nasal cavity, ethmoid sinus and frontal lobe. (b) Pre-operative enhanced axial CT scan showing the tumor extending intracranially and compressing the frontal lobe. (c) Pre-operative T2-weighted axial MRI showing the tumor compressing the intracranial structures. (d) Pre-operative T1-weighted coronal MRI showing the tumor with a well-defined capsule with slightly lower signal intensity (insert: sagittal view). (e) Pre-operative sagittal MRI enhanced by gadolinium injection. (f) Non-enhanced axial CT scan performed postoperatively demonstrating total removal of the tumor.
base defect (Fig. 1f). At the 6-month follow-up no complications have occurred. Histologically, the tumor consisted of compact spindle cells with palisading neoplastic proliferation. Cellular areas with the appearance of Antoni A tissue, and loosely textured areas of rounded cells resembling Antoni B tissue, were noted focally on hematoxylin and eosin (H&E) staining (Supplementary Fig. 1a). Immunophenotypically, the neoplastic cells showed positive staining for S-100 antiserum (Supplementary Fig. 1b,c), negative staining for epithelial membrane antigen (Supplementary Fig. 1d) and glial fibrillary acidic protein (Supplementary Fig. 1e), and there were approximately 3–5% Ki-67 positive tumor cells (Supplementary Fig. 1f). 3. Discussion A typical intracranial schwannoma is a benign tumor arising from Schwann cells of the peripheral nerve sheath.2 As Schwann cells are present in the meningeal branch of the trigeminal nerve,3 and the glossopharyngeal nerve at the skull base and olfactory groove,4 this is the most likely origin of the tumor in our patient. Unusual sources for intracranial schwannoma include dysplasia of Schwann cells in a perivascular plexus,1 origin from a spinal dorsal root,5 transformation of mesoderm cells from the pia mater into Schwann cells,2 and ectopic Schwann cells due to abnormal embryonic development.6 Immunohistochemical studies showed positive staining for S-100, which is one of the characteristic markers of nerve sheath tumors. The schwannoma was also easily distinguished from meningioma and glioma by the negative staining for both EMA and GFAP.
Although schwannomas are primarily benign, one malignant intracerebral schwannoma has been reported.7 The growth fraction in malignant schwannomas, as determined by Ki-67/MIB-1 immunoreactivity, ranges from 5–65%. The 3–5% growth fraction in our patient’s tumor is consistent with borderline malignant potential. However, the additional features such as a complete capsule and slow growth of the tumor are more consistent with benign etiology. Close long-term clinical follow-up will be necessary to monitor for recurrence. CT scans and MRI are critical when choosing the appropriate surgical approach for resection of the tumor. Our surgical approach is commonly used for other tumors in a similar anatomical location, including olfactory neuroblastoma, adenocarcinoma, and squamous cell carcinoma. However, for tumors that invade the nasal cavity and paranasal sinuses, a combined craniofacial approach may be preferred. The addition of a lateral rhinotomy or mid-facial degloving approach could also extend the exposure of such a tumor.8 During surgery, collaboration with otolaryngologists is recommended to ensure successful total tumor resection.
Appendix A. Supplementary material Supplementary material associated with this article can be found, in the online version, at doi:10.1016/j.jocn.2009.06.039.
References 1. Adelman LS, Aronson SM. I nerve fiber and Schwann cell proliferation within the spinal cord (schwannosis). Neurology 1972;22:726–31. 2. Russel DS, Rubenstein LJ. Pathology of tumors of the nervous system. 5th ed. London, England: Edward Arnold; 1989.
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Case Reports / Journal of Clinical Neuroscience 17 (2010) 522–523
3. Guthikonda B, Theodosopoulos PV, van Loveren H, et al. Evolution in the assessment and management of trigeminal schwannoma. Laryngoscope 2008;118:195–203. 4. Erongun U, Ozkal E, Acar O, et al. Intracerebral schwannoma: case report and review. Neurosurg Rev 1996;19:269–74. 5. McCormick WF. Intramedullary spinal cord schwannoma. A unique case. Arch Pathol 1964;77:378–82.
6. Pardatselier K, Iraci G, Cappellotto P, et al. Multiple intramedullary neurinomas of the spinal cord. Case report. J Neurosurg 1979;50:817–22. 7. Gonzalez LF, Lekovic GP, Eschbacher J, et al. A true malignant schwannoma of the eighth cranial nerve: case report. Neurosurgery 2007;61:E421–2. 8. Zhao JC, Liu G, Zhang YT, et al. Microsurgery of skull base [In Chinese]. 1st ed. Tianjin, China: Science and Technology Translation Publishing Company; 2005.
doi:10.1016/j.jocn.2009.06.039
Intramedullary cysticercosis S. Kumar a, A. Handa a,*, S. Chavda a, R. Tiwari a, P. Abbey b a b
Department of Neurosurgery, St Stephen’s Hospital, Marg Tis Hazari, Delhi 110 054, India Department of Radiology, All India Institute of Medical Sciences, New Delhi, India
a r t i c l e
i n f o
Article history: Received 7 August 2008 Accepted 24 April 2009
Keywords: Intramedullary cysticercosis Magnetic resonance imaging Spinal cord compression Taenia solium
a b s t r a c t Intramedullary cysticercosis is a rare form of cysticercosis in the central nervous system. A 55-year-old woman was admitted with low back pain, urinary incontinence, paraparesis and sensory deficit. Her MRI showed a well-defined intramedullary cystic lesion at T7 vertebral level with peripheral enhancement on contrast. She underwent a T7–T8 laminectomy and excision of the lesion. Histopathology revealed the lesion to be a cysticercus granuloma. A postoperative course of albendazole was given. The patient showed significant neurological improvement at follow-up. Ó 2009 Elsevier Ltd. All rights reserved.
1. Introduction Cysticercosis, caused by Taenia solium, is one of the commonest parasitic diseases that involve the central nervous system. However, an intramedullary cysticercus granuloma is rare. Mohanty et al. reported a series of eight patients;1 otherwise there have been sporadic reports.2–5 In our patient the cyst came out easily from the surrounding well-defined granuloma, which is unusual. 2. Case report A 55-year-old female was admitted with low back pain of 1month duration, followed 2 days later by numbness and weakness in both lower limbs. She developed urinary incontinence 1 week prior to admission, for which she underwent catheterisation of the bladder. On examination she was bedridden with grade 1/5 spastic paraparesis in the right lower limb and grade 2/5 on the left side. All sensations were impaired by 50% to 60% below L1. Routine blood and urine investigations were normal. A plain radiograph of the thoracolumbar spine was within normal limits. MRI of the thoracolumbar spine, however, revealed a well-defined intramedullary cystic lesion at the T7 level with signal intensities resembling cerebrospinal fluid (CSF) and peripheral enhancement on gadolinium administration (Fig. 1). Intraoperatively the cord was found to be bulging and pale. Purulent fluid was drained via a midline myelotomy, following which a well-defined greyish white cyst was removed intact. A well-defined granuloma surrounding the cyst was dissected completely with a good plane from the cord tissue. There was no growth of any organism on microbiological culture of the fluid. Her postoperative course was uneventful. Histopathology confirmed the diagnosis of cysticercosis (Supplementary Fig. 1). She * Corresponding author. Tel.: +91 11 239 77 930; fax: +91 11 239 32 412. E-mail address: [email protected] (A. Handa).
was given a course of albendazole 400 mg twice daily for 3 weeks. At the 6-month follow-up, power in her lower limbs had improved to grade 4/5 with reduced spasticity and she could walk with support. Her bladder function had also returned to normal. 3. Discussion Cysticercosis is endemic to Brazil, Mexico, Korea and South Asian countries.1–3,6 Cysticercal involvement of the spinal cord is rare even in endemic areas and accounts for 0.7% to 5.85% of all patients.1,3–5,7 Among patients with spinal cysticercosis, intramedullary involvement is relatively uncommon, the commonest being in the intradural–extramedullary region. Extradural and vertebral locations have also been reported.1,4,5,8 About half the reported cases have concomitant extra spinal involvement.9 Singh and Sahai remarked that only 45 patients with intramedullary cysticercosis had been documented worldwide until 2004.4 The thoracic cord, which receives the maximum blood supply, is most commonly affected, followed by cervical, lumbar and sacral regions.10,11 Neurological symptoms in intramedullary cysticercosis can be attributed to direct mechanical compression, inflammatory changes, cord edema, gliosis, pachymeningitis or syrinx formation. The patient usually presents with progressive neurological worsening with spastic quadriparesis or paraparesis depending on the site of the lesion; sphincter involvement, impotence and sensory deficit may also be present. Neurological involvement may be acute. MRI normally shows a cystic lesion with signal intensities similar to CSF on both T1-weighted and T2-weighted MRI. The scolex when present, appears to be isointense or hyperintense on T1-weighted MRI. The cyst wall usually enhances on contrast administration. An associated syrinx is generally secondary to arachnoiditis, circulatory insufficiency or cord atrophy. In the absence of any extraspinal evidence of cysticercosis and in a setting of neurological deficits attributable to the lesion,