Letters to Editor
2.
Alkan A, Parlak M, Baysal T, Sigirci A, Kutlu R, Altinok T. En‑plaque tuberculomas of tentorium in a pregnant woman: Follow‑up with MRI (2003:2b). Eur Radiol 2003;13:1190‑3. 3. Bauer J, Johnson RF, Levy JM, Pojman DV, Ruge JR. Tuberculoma presenting as an en plaque meningioma. Case report. J Neurosurg 1996;85:685‑8. 4. Ng SH, Tang LM, Lui TN, Ko SF, Wong HF, Wai YY, et al. Tuberculoma en plaque: CT. Neuroradiology 1996;38:453‑5. 5. Singh KK, Nair MD, Radhakrishnan K, Tyagi JS. Utility of PCR assay in diagnosis of en‑plaque tuberculoma of the brain. J Clin Microbiol 1999;37:467‑70. 6. Khanna PC, Godinho S, Patkar DP, Pungavkar SA, Lawande MA. MR spectroscopy‑aided differentiation: “giant” extra‑axial tuberculoma masquerading as meningioma. AJNR Am J Neuroradiol 2006;27:1438‑40. 7. Wasay M, Kheleani BA, Moolani MK, Zaheer J, Pui M, Hasan S, et al. Brain CT and MRI findings in 100 consecutive patients with intracranial tuberculoma. J Neuroimaging 2003;13:240‑7. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.149417
Received: 10-11-2014 Review completed: 14-11-2014 Accepted: 05-12-2014
Craniocervical pneumatization presenting as cranial nerve IX–XII palsies: Case report and review of the literature Sir, A previously healthy 68‑year‑old male presented with dysphagia and slurred speech after a fall. Two weeks prior to presentation, patient suffered a mechanical fall and landed on his tail bone and right elbow. He denied loss of consciousness and hitting his head directly, but heard a “pop” on the right side. Afterwards, he had neck pain and trouble with eating food, specifically closing his mouth. His neurological exam was significant for dysarthria, a palate that deviated to the left, a tongue that deviated to the right [Figure 1a] and subtle right trapezius weakness. The rest of the exam was normal. Computed tomography (CT) and magnetic resonance imaging (MRI) of head and cervical spine revealed extensive craniocervical pneumatization, but no other abnormalities [Figure 1b‑d]. The patient was treated with intravenous solumedrol, followed by an extended steroid taper. He required total enteral nutrition and at 1‑month, a percutaneous endoscopic gastrostomy tube was placed. At 4 months, there was minimal improvement of the patient’s cranial nerve palsies. Neurology India | Nov-Dec 2014 | Vol 62 | Issue 6
a
b
c
d
Figure 1: Neurological exam was signification for deviation of the tongue to the right (a) Computed tomography (CT) (b, c, axial views) and magnetic resonance imaging (MRI) (d, coronal views) show occipital‑temporal and craniocervical pneumatization (arrows)
Pneumatization of both the skull base and cervical vertebrae is extremely rare. We identified four cases in the English literature that presented with cranial nerve palsies: XII and IX,[1] XII and V,[2] XII alone,[3] and IX alone.[4] All were treated conservatively. Of the two that reported outcome, one resolved completely at 10 days,[3] while the other one had persistent deficits at 4 years after presentation. [1] Pathogenesis of craniocervical pneumatization remains unclear. Theories involve either a developmental anomaly or a relationship to an elevated pressure in the middle and inner ear.[1,4‑6] This happens in patients with Eustachian tube dysfunction or those who habitually perform the Valsalva maneuver or engage in repetitive pressure activities such as scuba diving.[5,6] In our case, we were unable to demonstrate any skull base or vertebral fractures, but it was felt that the etiology of his cranial nerve dysfunctions was likely his traumatic fall that had resulted in small pathologic fracture of his skull base with a resultant pneumocele. The treatment was conservative because there was no instability involving the craniocervical junction and benefits from aggressive interventions were not considered to outweigh risks. Although the explanations of the findings in the present case remains unclear, this report illustrates that extensive craniocervical pneumatization can cause cranial nerve deficits that may not improve with conservative management alone.
Acknowledgement This work was supported by the Natural Science Foundation of China (No. 81301988 to L.Y.) and China Ministry of Education Doctoral Program Spot Foundation (20130162120061 to L.Y.).
Yanqiao Xiao, Harrison X. Bai1, Xin Zhao, Yi Shu2, Yingjie Zou, Li Yang 699
Letters to Editor
Department of Neurology, The Second Xiangya Hospital of Central South University, Changsha, 2Department of Neurology, The First Xiangya Hospital of Central South University, Changsha, Hunan, China, 1Department of Radiology Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania, United States E‑mail: [email protected]
References 1. 2. 3. 4. 5. 6.
Kaiser R, Mehdian H. Permanent twelfth nerve palsy secondary to C0 and C1 fracture in patient with craniocervical pneumatisation. Eur Spine J 2014. Sadler DJ, Doyle GJ, Hall K, Crawford PJ. Craniocervical bone pneumatisation. Neuroradiology 1996;38:330‑2. Renard D, Freitag C, Castelnovo G. Mystery case: Hypoglossal nerve palsy in occipito‑temporal pneumatization. Neurology 2012;79:e109‑10. Quigley AJ, Shannon H. Craniocervical pneumatization. J Radiol Case Rep 2013;7:27‑33. Moreira B, Som PM. Unexplained extensive skull base and atlas pneumatization: Computed tomographic findings. Arch Otolaryngol Head Neck Surg 2010;136:731‑3. Littrell LA, Leutmer PH, Lane JI, Driscoll CL. Progressive calvarial and upper cervical pneumatization associated with habitual valsalva maneuver in a 70‑year‑old man. AJNR Am J Neuroradiol 2004;25:491‑3. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.149433
Received: 15‑08‑2014 Review completed: 15‑09‑2014 Accepted: 05‑12‑2014
Spontaneous indirect CSF rhinorrhea following excision of a giant choroid plexus papilloma of lateral ventricle Sir, Indirect cerebrospinal fluid (CSF) rhinorrhea following excision of large remote intracranial lesions is uncommon. [1] This report describes a case of CSF rhinorrhea following total excision of a giant choroid plexus papilloma (CPP). A 26‑year‑old male presented with headache and decreased visual acuity in both eyes of 6‑month duration. There was no history of CSF rhinorrhea. On examination, perception of light was absent in both the eyes. Fundus examination was suggestive of secondary optic atrophy. 700
Magnetic resonance imaging (MRI) revealed a large intensely enhancing intraventricular tumor involving the body of the right lateral ventricle with extension to trigone and temporal horn associated with perilesional edema and hydrocephalus [Figure 1a‑c]. There was significant mass effect and midline shift. Herniation of brain through multiple defects in anterior cranial fossa (ACF) base was noted [Figure 1d and e]. Right temporal craniotomy and gross total excision of the tumor was done. Bone flap was not replaced as the brain was full at the end of surgery. Initial postoperative period was uneventful. Histopathology was suggestive of CPP. On the 2nd postoperative day, patient developed profuse CSF rhinorrhea which was resistant to conservative measures. Computed tomography (CT) revealed increase in the perilesional edema and normal‑sized ventricles. CT cisternogram revealed ACF base defects near the cribriform plate [Figure 1f]. In view of large and multiple bony defects in the ACF base, craniotomy and repair of the defects was considered to be a better option than endoscopic repair. Bifrontal craniotomy and repair of the ACF base was done with a vascularized pericranial graft and fibrin glue. Large bilateral dural and bony defects in ACF base were noted during surgery. CSF rhinorrhea completely stopped after ACF base repair. CT brain done 2 weeks later showed gross hydrocephalus and complete excision of the tumor [Figure 1g and h]. Left ventriculoperitoneal shunt was done. At the time of discharge, patient had no new neurological deficits and there was no improvement in his vision. CSF rhinorrhea can be caused by neoplasms in two ways. In the direct type of CSF rhinorrhea, the neoplastic lesions like pituitary adenomas directly cause erosion of meninges and bone. In indirect type of CSF rhinorrhea, raised intracranial pressure (ICP) results in progressive erosion of skull base.[1] Thin bone near the cribriform plate is the most common site of bony erosion in indirect type of CSF rhinorrhea. Indirect type of CSF rhinorrhea commonly present before the excision of the tumor (pretreatment type) and rarely after tumor excision (posttreatment type).[1‑5] The bony defects in pretreatment type tend to be smaller as the raised ICP is reduced by CSF leak and such small defects usually heal spontaneously after tumor excision.[1,4,5] The bony defects in posttreatment type are usually larger.[1] Herniating brain due to raised ICP plugs the bony and dural defects before tumor excision. Persistent raised ICP results in further herniation of brain tissue and progressive enlargement of the bony defects.[1] Decrease in ICP following tumor excision results in unplugging of these defects and profuse CSF rhinorrhea.[1] As the mechanism of posttreatment CSF rhinorrhea is due to unplugging of the large defect, there are seldom chances of settling of CSF rhinorrhea with any conservative measures. Neurology India | Nov-Dec 2014 | Vol 62 | Issue 6
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.
2.
Alkan A, Parlak M, Baysal T, Sigirci A, Kutlu R, Altinok T. En‑plaque tuberculomas of tentorium in a pregnant woman: Follow‑up with MRI (2003:2b). Eur Radiol 2003;13:1190‑3. 3. Bauer J, Johnson RF, Levy JM, Pojman DV, Ruge JR. Tuberculoma presenting as an en plaque meningioma. Case report. J Neurosurg 1996;85:685‑8. 4. Ng SH, Tang LM, Lui TN, Ko SF, Wong HF, Wai YY, et al. Tuberculoma en plaque: CT. Neuroradiology 1996;38:453‑5. 5. Singh KK, Nair MD, Radhakrishnan K, Tyagi JS. Utility of PCR assay in diagnosis of en‑plaque tuberculoma of the brain. J Clin Microbiol 1999;37:467‑70. 6. Khanna PC, Godinho S, Patkar DP, Pungavkar SA, Lawande MA. MR spectroscopy‑aided differentiation: “giant” extra‑axial tuberculoma masquerading as meningioma. AJNR Am J Neuroradiol 2006;27:1438‑40. 7. Wasay M, Kheleani BA, Moolani MK, Zaheer J, Pui M, Hasan S, et al. Brain CT and MRI findings in 100 consecutive patients with intracranial tuberculoma. J Neuroimaging 2003;13:240‑7. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.149417
Received: 10-11-2014 Review completed: 14-11-2014 Accepted: 05-12-2014
Craniocervical pneumatization presenting as cranial nerve IX–XII palsies: Case report and review of the literature Sir, A previously healthy 68‑year‑old male presented with dysphagia and slurred speech after a fall. Two weeks prior to presentation, patient suffered a mechanical fall and landed on his tail bone and right elbow. He denied loss of consciousness and hitting his head directly, but heard a “pop” on the right side. Afterwards, he had neck pain and trouble with eating food, specifically closing his mouth. His neurological exam was significant for dysarthria, a palate that deviated to the left, a tongue that deviated to the right [Figure 1a] and subtle right trapezius weakness. The rest of the exam was normal. Computed tomography (CT) and magnetic resonance imaging (MRI) of head and cervical spine revealed extensive craniocervical pneumatization, but no other abnormalities [Figure 1b‑d]. The patient was treated with intravenous solumedrol, followed by an extended steroid taper. He required total enteral nutrition and at 1‑month, a percutaneous endoscopic gastrostomy tube was placed. At 4 months, there was minimal improvement of the patient’s cranial nerve palsies. Neurology India | Nov-Dec 2014 | Vol 62 | Issue 6
a
b
c
d
Figure 1: Neurological exam was signification for deviation of the tongue to the right (a) Computed tomography (CT) (b, c, axial views) and magnetic resonance imaging (MRI) (d, coronal views) show occipital‑temporal and craniocervical pneumatization (arrows)
Pneumatization of both the skull base and cervical vertebrae is extremely rare. We identified four cases in the English literature that presented with cranial nerve palsies: XII and IX,[1] XII and V,[2] XII alone,[3] and IX alone.[4] All were treated conservatively. Of the two that reported outcome, one resolved completely at 10 days,[3] while the other one had persistent deficits at 4 years after presentation. [1] Pathogenesis of craniocervical pneumatization remains unclear. Theories involve either a developmental anomaly or a relationship to an elevated pressure in the middle and inner ear.[1,4‑6] This happens in patients with Eustachian tube dysfunction or those who habitually perform the Valsalva maneuver or engage in repetitive pressure activities such as scuba diving.[5,6] In our case, we were unable to demonstrate any skull base or vertebral fractures, but it was felt that the etiology of his cranial nerve dysfunctions was likely his traumatic fall that had resulted in small pathologic fracture of his skull base with a resultant pneumocele. The treatment was conservative because there was no instability involving the craniocervical junction and benefits from aggressive interventions were not considered to outweigh risks. Although the explanations of the findings in the present case remains unclear, this report illustrates that extensive craniocervical pneumatization can cause cranial nerve deficits that may not improve with conservative management alone.
Acknowledgement This work was supported by the Natural Science Foundation of China (No. 81301988 to L.Y.) and China Ministry of Education Doctoral Program Spot Foundation (20130162120061 to L.Y.).
Yanqiao Xiao, Harrison X. Bai1, Xin Zhao, Yi Shu2, Yingjie Zou, Li Yang 699
Letters to Editor
Department of Neurology, The Second Xiangya Hospital of Central South University, Changsha, 2Department of Neurology, The First Xiangya Hospital of Central South University, Changsha, Hunan, China, 1Department of Radiology Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania, United States E‑mail: [email protected]
References 1. 2. 3. 4. 5. 6.
Kaiser R, Mehdian H. Permanent twelfth nerve palsy secondary to C0 and C1 fracture in patient with craniocervical pneumatisation. Eur Spine J 2014. Sadler DJ, Doyle GJ, Hall K, Crawford PJ. Craniocervical bone pneumatisation. Neuroradiology 1996;38:330‑2. Renard D, Freitag C, Castelnovo G. Mystery case: Hypoglossal nerve palsy in occipito‑temporal pneumatization. Neurology 2012;79:e109‑10. Quigley AJ, Shannon H. Craniocervical pneumatization. J Radiol Case Rep 2013;7:27‑33. Moreira B, Som PM. Unexplained extensive skull base and atlas pneumatization: Computed tomographic findings. Arch Otolaryngol Head Neck Surg 2010;136:731‑3. Littrell LA, Leutmer PH, Lane JI, Driscoll CL. Progressive calvarial and upper cervical pneumatization associated with habitual valsalva maneuver in a 70‑year‑old man. AJNR Am J Neuroradiol 2004;25:491‑3. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.149433
Received: 15‑08‑2014 Review completed: 15‑09‑2014 Accepted: 05‑12‑2014
Spontaneous indirect CSF rhinorrhea following excision of a giant choroid plexus papilloma of lateral ventricle Sir, Indirect cerebrospinal fluid (CSF) rhinorrhea following excision of large remote intracranial lesions is uncommon. [1] This report describes a case of CSF rhinorrhea following total excision of a giant choroid plexus papilloma (CPP). A 26‑year‑old male presented with headache and decreased visual acuity in both eyes of 6‑month duration. There was no history of CSF rhinorrhea. On examination, perception of light was absent in both the eyes. Fundus examination was suggestive of secondary optic atrophy. 700
Magnetic resonance imaging (MRI) revealed a large intensely enhancing intraventricular tumor involving the body of the right lateral ventricle with extension to trigone and temporal horn associated with perilesional edema and hydrocephalus [Figure 1a‑c]. There was significant mass effect and midline shift. Herniation of brain through multiple defects in anterior cranial fossa (ACF) base was noted [Figure 1d and e]. Right temporal craniotomy and gross total excision of the tumor was done. Bone flap was not replaced as the brain was full at the end of surgery. Initial postoperative period was uneventful. Histopathology was suggestive of CPP. On the 2nd postoperative day, patient developed profuse CSF rhinorrhea which was resistant to conservative measures. Computed tomography (CT) revealed increase in the perilesional edema and normal‑sized ventricles. CT cisternogram revealed ACF base defects near the cribriform plate [Figure 1f]. In view of large and multiple bony defects in the ACF base, craniotomy and repair of the defects was considered to be a better option than endoscopic repair. Bifrontal craniotomy and repair of the ACF base was done with a vascularized pericranial graft and fibrin glue. Large bilateral dural and bony defects in ACF base were noted during surgery. CSF rhinorrhea completely stopped after ACF base repair. CT brain done 2 weeks later showed gross hydrocephalus and complete excision of the tumor [Figure 1g and h]. Left ventriculoperitoneal shunt was done. At the time of discharge, patient had no new neurological deficits and there was no improvement in his vision. CSF rhinorrhea can be caused by neoplasms in two ways. In the direct type of CSF rhinorrhea, the neoplastic lesions like pituitary adenomas directly cause erosion of meninges and bone. In indirect type of CSF rhinorrhea, raised intracranial pressure (ICP) results in progressive erosion of skull base.[1] Thin bone near the cribriform plate is the most common site of bony erosion in indirect type of CSF rhinorrhea. Indirect type of CSF rhinorrhea commonly present before the excision of the tumor (pretreatment type) and rarely after tumor excision (posttreatment type).[1‑5] The bony defects in pretreatment type tend to be smaller as the raised ICP is reduced by CSF leak and such small defects usually heal spontaneously after tumor excision.[1,4,5] The bony defects in posttreatment type are usually larger.[1] Herniating brain due to raised ICP plugs the bony and dural defects before tumor excision. Persistent raised ICP results in further herniation of brain tissue and progressive enlargement of the bony defects.[1] Decrease in ICP following tumor excision results in unplugging of these defects and profuse CSF rhinorrhea.[1] As the mechanism of posttreatment CSF rhinorrhea is due to unplugging of the large defect, there are seldom chances of settling of CSF rhinorrhea with any conservative measures. Neurology India | Nov-Dec 2014 | Vol 62 | Issue 6
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.
