- Email: [email protected]
Inverting papilloma of the sphenoid sinus: report of two cases
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Inverting papilloma of the sphenoid sinus: report of two cases S. Nishio, K. Samoto, I. Takeshita, K. Matsumoto, T. Matsushima, M. Fukui Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University
Fig. 1 Case 1. A plain skull X-ray reveals an opacification of the sphenoid and ethomoid sinuses (A). The tumour, which is isointense with the cerebral cortex on both T1- (B) and T2- (C) weighted images, is located in the sphenoid sinus and posterior part of the ethomoid sinus.
Summary Two patients with sphenoid sinus inverting papilloma who were treated either by transcranial or sublabial trans-septal approach are reported. Inverting papillomas arising from the sphenoid sinus are exceedingly rare. The clinical and neuro-imaging features, as well as surgical treatment, for sphenoid sinus tumours are also briefly discussed. © 2001 Harcourt Publishers Ltd Journal of Clinical Neuroscience (2001) 8(2), 168–170 © 2001 Harcourt Publishers Ltd DOI: 10.1054/jocn.2000.0727, available online at http://www.idealibrary.com on
Keywords: rhinoseptal approach, skull base surgery, sphenoid sinus tumour, transcranial approach Received 22 December 1999 Accepted 15 February 2000 Correspondence to: S. Nishio MD, Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan; Fax: ;81-92-642-5526; E-mail: [email protected]
INTRODUCTION Isolated sphenoid sinus disease is uncommon, and symptoms are generally non-specific. However, with the use of routine computed tomography (CT) and magnetic resonance imaging (MRI) in otolaryngology, ophthalmology and neurology, it is now being reported with increasing frequency.1 While inverting papilloma (IP) is an uncommon but well described neoplasm and usually involves the mucosa of the nasal cavity,2,3 this tumour has also been reported to occur within the paranasal sinuses. We present herein two cases of IP arising in the sphenoid sinus which were treated surgically either by transcranial approach or sublabial transseptal microscopic sphenoidotomy. CASE REPORTS Case 1 A 54-year-old woman developed dysarthria, gait disturbance and neck tremor in 1988. She was without nasal discharge, epistaxis, nasal obstruction, headache, or diplopia. A CT scan demonstrated cerebellar atrophy and an enhancing mass in the sphenoid sinus with bony thinning of the sinus walls. She was admitted to an ENT clinic, and a diagnostic endonasal endoscopic biopsy for the sphenoid sinus tumour was performed showing IP. After administration of alpha-interferon for 6 months, the tumour slightly reduced in size. A follow up CT scan in 1990, however, revealed enlargement of the tumour, which occupied the sphenoid sinus and the posterior part of the ethmoid sinus. An MRI revealed the tumour to be isointense on both T1- and T2-weighted images (Fig. 1). Neither rhinological nor neurological symptoms related to this sphenoid sinus tumour were apparent. The patient was then transferred to us for surgery. Journal of Clinical Neuroscience (2001) 8(2)
Fig. 2 Case 1. Preoperative CT scan shows a mass in the sphenoid sinus as well as in the posterior part of the ethomoid sinus (A). Postoperative CT scan reveals total removal of the tumour (B).
At surgery, a bifrontal free bone flap craniotomy was performed, and the base of the frontal fossa was exposed. The planum sphenoidale was then drilled, and the sphenoid sinus opened. Through this opening, which was 2.5 cm in sagittal length and 1.5 cm in width, a grayish multinodular tumour filling the sphenoid and the posterior ethmoid sinuses was exposed. During the removal of the tumour, the right olfactory tract was damaged. Using the anterior interhemispheric trans-planum sphenoidale approach, the tumour was totally removed (Fig. 2). The opening of the sphenoid sinus was closed with several pieces of temporalis muscle and temporalis fascia graft, and was covered with a pedunculated pericranial flap. The patient’s postoperative course was uneventful and the patient was free of disease when last seen in September 1998. Case 2 This 50-year-old man was discovered to have a sphenoid sinus tumour as an incidental finding on MRI scans. He denied symptoms of mucopurulent nasal discharge, epistaxis, anosmia or obstruction, and physical examination revealed a normal nasal cavity. A neurologic work up, including ophthalmologic examinations, was negative. On MRI the tumour was isointense to the cerebral cortex on both T1- and T2-weighted images (Fig. 3), and was homogeneously enhanced after the administration of contrast medium (Fig. 4A). A CT scan revealed a well defined, slightly hyperdense soft tissue mass within the sphenoid sinus, not associated with bony destruction. The patient underwent a sublabial trans-septal microscopic sphenoidotomy that revealed a friable and vascular soft tissue mass filling the sphenoid sinus. While intraoperative histological examination of biopsy specimens revealed the tumour to be a papilloma, it was only partly excised as the tumour visualisation was limited for total removal and there was massive haemorrhage from the tumour (Fig. 4B,C,D). After surgery the patient has done well with the residual tumour. Histological analysis on paraffin embedded specimens revealed the tumour to be an IP (Fig. 5). © 2001 Harcourt Publishers Ltd
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Fig. 3 Case 2. An MRI shows the tumour filling the sphenoid sinus, which is isointense both on T1-weighted (A) and T2-weighted (B) images. Note the intact pituitary gland.
Fig. 4 Case 2. The sphenoid sinus tumour is enhanced after the administration of Gd-DTPA (A: Preoperative coronal T1-weighted image). Postoperative T1-weighted coronal images with Gd-DTPA show the tumour to be partially removed.
pathologic entities of the lesion. Bony expansion or erosion on CT scan usually suggests a benign or a slow growing nature of the lesion, as was demonstrated in our cases, and bone destruction indicates a malignant nature.13 While IP and sinusitis may appear similar on a CT scan, the MRI scan may help in differentiating these two lesions. Sinusitis is usually isointense on T1-weighted images and hyperintense on T2-weighted images, and IP is isointense both on T1- and T2-weighted images,12 as demonstrated here. For many years surgical access to the sphenoid sinus, which is located deep in the geometric centre of the head and surrounded by vital structures, was problematic.14 However, the development of the nasal endoscope has made the approach to the sphenoid sinus both easier and safer.15 This procedure provides excellent visualisation within the nasal cavity and direct access to the natural ostium of the sphenoid sinus, and is an effective modality for diagnosis and treatment of most sphenoid sinus disorders.1,12 While this procedure was initially chosen for biopsy of the tumour in case 1, the nasal endoscopic approach potentially provides adequate tumour removal – even in large tumours – if they can be visualised. Among other approaches for the sphenoid sinus, including the transethmoidal and transpalatal routes, the most popular one has been the sublabial trans-septal sphenoidotomy.1 Through this approach, however, we could only partially resect the sphenoid sinus tumour of case 2 because of insufficient visualisation of the tumour and severe haemorrhage from the tumour. Any IP that is extensive and is outside the visualisation of the operating microscope may be treated by other routes.12 The tumour of case 1, which involved both the sphenoid and the posterior ethmoid sinuses, was totally removed through a midline transcranial approach. Regardless of the surgical approach, the key to successful treatment of IP remains wide surgical excision under optimal visualisation,2,3,12 since IP is characterised by its tendency to recur and association with malignant evolution. In addition, conventional radiotherapy for IP has been shown to be of little success.2,3,16–20 For a radical removal of the sphenoid sinus IP, an image-guided navigation technique may be of help. REFERENCES 1. 2.
Fig. 5 Photomicrographs of the tumour. A low power micrograph shows the hyperplastic epithelium inverting into the underlying stroma (A: case 1, H&E,70). A high power micrograph shows pseudostratified ciliated columnar lining (B: case 2, H&E,120).
3.
4.
DISCUSSION IP is an uncommon but well described neoplasm, with the overall incidence of this tumour varying from 0.5 to 4% of all primary nasal tumours.4 IP most commonly arises from the lateral nasal wall3 and rarely develops primarily within a sinus cavity.5–7 While we have presented two cases of IP arising in the sphenoid sinus, only a few such cases have previously been described.2,3,8 The most common complaints in patients with IP are nasal obstruction, followed by mucopurulent nasal discharge, epistaxis, anosmia, and facial pain.6,9 However, symptoms of patients with disease localised to the sphenoid sinus are initially quite subtle and vague.2 In our two cases, sphenoid sinus tumours were found incidentally on neuro-imaging studies, and no clinical signs or symptoms related to the tumour were apparent. Although there are only a limited number of reports on the CT and MRI features of IP, neuro-imaging studies play an important role in locating and defining the specific site and extent of this tumour.1–3,10–12 In addition, it may help to characterise histo© 2001 Harcourt Publishers Ltd
5. 6.
7. 8. 9. 10. 11. 12.
13. 14.
Hadar T, Yaniv E, Shvero J. Isolated sphenoid sinus changes – history, CT and endoscopic finding. J Laryngol Otol 1996; 110: 850–853. McElveen JT Jr, Fee WE Jr. Inverting papilloma of the sphenoid sinus. Otolaryngol Head Neck Surg 1981; 89: 710–712. Peters BW, O’Reilly RC, Willcox TO Jr, Rao VM, Lowry LD, Keane WM. Inverted papilloma isolated to the sphenoid sinus. Otolaryngol Head Neck Surg 1995; 113: 771–777. Myers E, Fernau J, Johnson J, Tabet J, Barnes L. Management of inverted papilloma. Laryngoscope 1990; 100: 481–490. Lawson W, Biller H, Jacobson A, Som P. The role of conservative surgery in the management of inverted papilloma. Laryngoscope 1983; 93: 148–155. Phillips P, Gustafson R, Facer G. The clinical behavior of inverting papilloma of the nose and paranasal sinuses: report of 112 cases and review of the literature. Laryngoscope 1990; 100: 463–469. Suh KW, Facer GW, Devine KD, Weiland LH, Zujko RD. Inverting papilloma of the nose and paranasal sinuses. Laryngoscope 1977; 87: 35–46. Edison BD. Primary inverting papilloma of the sphenoid sinus. Trans Am Acad Ophthalmol Otolaryngol 1975; 80: 434–437. Kamal S. Inverted papilloma of the nose. J Laryngol Otol 1981; 95: 1069–1079. Myers EN, Schramm V Jr, Barnes E Jr. Management of inverted papilloma of the nose and paranasal sinuses. Laryngoscope 1981; 91: 2071–2084. Sethi DS, Lau DP, Chee LW, Chong V. Isolated sphenoethmoid recess polyps. J Laryngol Otol 1998; 112: 660–663. Stankiewicz JA, Girgis SJ. Endoscopic surgical treatment of nasal and paranasal sinus inverted papilloma. Otolaryngol Head Neck Surg 1993; 109: 988–995. Dubois PJ, Schultz JC, Perrin RL Dastur KJ. Tomography in expansile lesions of the nasal and paranasal sinuses. Radiology 1977; 125: 149–158. Donald PJ, Boggan JE. Sphenoid sinus malignancies. J Craniofacial Surg 1995; 6: 15–23.
Journal of Clinical Neuroscience (2001) 8(2)
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170 Vinas et al. 15. 16.
17. 18. 19. 20.
spinal neuraxis, typically at the craniocervical junction. Patients with this condition may require conservative or surgical treatment, depending on the severity and progression of the symptoms. In recent years, new neuroimaging techniques have revived the interest in this entity, with respect to indications and results of treatment. With the widespread use of magnetic resonance scanning, lesions in the spinal cord such as syrinx or hydromyelia can be diagnosed earlier, often before producing clinical symptoms. The natural history of syrinx, however, is not well understood. The authors present a case of syringomyelia with spontaneous remission and clinical improvement. Similar cases reported in the literature were collected and analysed.
Wigand ME. Transnasal ethmoidectomy under endoscopic control. Rhinology 1981; 19: 7–15. Dolgin S, Zaveri V, Casiano R, Maniglia A. Different options for treatment of inverting papilloma of the nose and paranasal sinuses: a report of 41 cases. Laryngoscope 1992; 102: 231–236. Fechner RE, Alford DO. Inverted papilloma and squamous carcinoma: An unusual case. Arch Otolaryngol 1968; 88: 507–512. Vrabec DP. The inverted Schneiderian papilloma: a clinical and pathological study. Laryngoscope 1975; 85: 186–220. Cummings CW, Goodman ML. Inverted papillomas of the nose and paranasal sinuses. Arch Otolaryngol 1970; 92: 445–449. Norris HJ. Papillary lesions of the nasal cavity and paranasal sinuses. II. Inverting papillomas: A study of 29 cases. Laryngoscope 1973; 73: 1–17.
METHODS A Medline search in the English, French and Spanish literature performed for the years 1966 to August 1999, and review of bibliographic citations for additional cases prior to 1966, yielded seven patients with syrinx which spontaneously resolved without surgical intervention (Table 1). The analysis included demographic, clinical and radiological features, non-surgical therapeutic interventions, and time of follow up.
Spontaneous resolution of a syrinx Federico C. Vinas MD, Julie Pilitsis MD, Harvey Wilner MD Department of Neurological Surgery and Radiology, Wayne State University, School of Medicine, Detroit, MI
CASE REPORT Summary Syrinx are often related to trauma, tumours or abnormalities of the craniocervical junction. Only a few cases of spontaneous resolution have been reported in the literature. The authors present a case of spontaneous resolution of a cervical syrinx, and analyse the possible physiopathological mechanisms leading to resolution. Other similar cases reported in the literature were collected and analysed. © 2001 Harcourt Publishers Ltd Journal of Clinical Neuroscience (2001) 8(2), 170–172 © 2001 Harcourt Publishers Ltd DOI: 10.1054/jocn.2000.0768, available online at http://www.idealibrary.com on
Keywords: hydromyelia, syrinx, natural history, treatment Received 2 February 2000 Accepted 16 March 2000 Correspondence to: F. C. Vinas, Tel.: 904 253 7644; Fax: 904 252 1776
A 58-year-old male presented with a 3-month history of neck pain and pain and paraesthesias in his right upper extremity. Past medical history was significant for cataract on his left eye. He did not have a history of previous trauma. On physical examination, he had normal muscle tone and strength, and no myelopathic signs. There were some patchy areas of decreased sensation to light touch and pin prick in both upper extremities which did not follow any dermatome. A magnetic resonance image (MRI) of the cervical spine showed protrusion of the C4–C5 disc, and a central syrinx extending from C-6 to T-1 (Fig. 1). There was no enhancement after the administration of gadolinium DTPA. The patient was subsequently lost to follow up. His pain gradually improved over time. A follow up MRI was obtained 5 years later and showed complete disappearance of the syrinx (Fig. 2). There was no abnormal sign intensity in the spinal cord. The C4–C5 disc bulge appeared unchanged.
INTRODUCTION DISCUSSION The term syrinx includes a group of conditions associated with the presence of a cavity in the spinal cord. This process is usually associated with trauma or congenital abnormalities along the
Table 1
Syringomyelia was first recognized as a disease process approximately 400 years ago but the term syringomyelia or cystic
Clinical and demographic characteristics of patients with spontaneous resolution of syringomyelia reported in the literature
Author
Age/sex
Presentation
Diagnosis
Location
Assoc Chiari
Clinical course
Follow-up Ref
Sudo et al. (1990)
16 M
MRI
Cervicothoracic
Yes
Improvement
2 years
15
Jack et al. (1991)
30 F
MRI
Cervicothoracic
Yes
Little improvement
2 years
14
Olivero et al. (1992)
28 F
MRI
Cervical
Yes
Improvement
2 months
16
Yeager and Lusser (1992) Santoro et al. (1993)
19m F 42 M
MRI MRI
Cervical Cervicothoracic
No Yes
No symptoms Unchanged
2 years 3 years
17 13
Santoro et al. (1993) Pierallini et al. (1997) Present case
31 F 25 F 58 M
Left side hypalgesia, hypoaesthesia Paresthesias, decrease sensation on chest wall and left upper extremity Post-traumatic bilateral arm weakness Seizures, normal examination Paraesthesias, gait disturbance, atrophy of hand muscles Incidental finding Headaches, dizziness Pain on right upper extremity
MRI MRI MRI
Cervicothoracic Cervical Cervicothoracic
Yes Yes No
Asymptomatic Improvement Improvement
3 years 4 years 5 years
13 11
F, female; M, male; m, months.
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Inverting papilloma of the sphenoid sinus: report of two cases S. Nishio, K. Samoto, I. Takeshita, K. Matsumoto, T. Matsushima, M. Fukui Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University
Fig. 1 Case 1. A plain skull X-ray reveals an opacification of the sphenoid and ethomoid sinuses (A). The tumour, which is isointense with the cerebral cortex on both T1- (B) and T2- (C) weighted images, is located in the sphenoid sinus and posterior part of the ethomoid sinus.
Summary Two patients with sphenoid sinus inverting papilloma who were treated either by transcranial or sublabial trans-septal approach are reported. Inverting papillomas arising from the sphenoid sinus are exceedingly rare. The clinical and neuro-imaging features, as well as surgical treatment, for sphenoid sinus tumours are also briefly discussed. © 2001 Harcourt Publishers Ltd Journal of Clinical Neuroscience (2001) 8(2), 168–170 © 2001 Harcourt Publishers Ltd DOI: 10.1054/jocn.2000.0727, available online at http://www.idealibrary.com on
Keywords: rhinoseptal approach, skull base surgery, sphenoid sinus tumour, transcranial approach Received 22 December 1999 Accepted 15 February 2000 Correspondence to: S. Nishio MD, Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan; Fax: ;81-92-642-5526; E-mail: [email protected]
INTRODUCTION Isolated sphenoid sinus disease is uncommon, and symptoms are generally non-specific. However, with the use of routine computed tomography (CT) and magnetic resonance imaging (MRI) in otolaryngology, ophthalmology and neurology, it is now being reported with increasing frequency.1 While inverting papilloma (IP) is an uncommon but well described neoplasm and usually involves the mucosa of the nasal cavity,2,3 this tumour has also been reported to occur within the paranasal sinuses. We present herein two cases of IP arising in the sphenoid sinus which were treated surgically either by transcranial approach or sublabial transseptal microscopic sphenoidotomy. CASE REPORTS Case 1 A 54-year-old woman developed dysarthria, gait disturbance and neck tremor in 1988. She was without nasal discharge, epistaxis, nasal obstruction, headache, or diplopia. A CT scan demonstrated cerebellar atrophy and an enhancing mass in the sphenoid sinus with bony thinning of the sinus walls. She was admitted to an ENT clinic, and a diagnostic endonasal endoscopic biopsy for the sphenoid sinus tumour was performed showing IP. After administration of alpha-interferon for 6 months, the tumour slightly reduced in size. A follow up CT scan in 1990, however, revealed enlargement of the tumour, which occupied the sphenoid sinus and the posterior part of the ethmoid sinus. An MRI revealed the tumour to be isointense on both T1- and T2-weighted images (Fig. 1). Neither rhinological nor neurological symptoms related to this sphenoid sinus tumour were apparent. The patient was then transferred to us for surgery. Journal of Clinical Neuroscience (2001) 8(2)
Fig. 2 Case 1. Preoperative CT scan shows a mass in the sphenoid sinus as well as in the posterior part of the ethomoid sinus (A). Postoperative CT scan reveals total removal of the tumour (B).
At surgery, a bifrontal free bone flap craniotomy was performed, and the base of the frontal fossa was exposed. The planum sphenoidale was then drilled, and the sphenoid sinus opened. Through this opening, which was 2.5 cm in sagittal length and 1.5 cm in width, a grayish multinodular tumour filling the sphenoid and the posterior ethmoid sinuses was exposed. During the removal of the tumour, the right olfactory tract was damaged. Using the anterior interhemispheric trans-planum sphenoidale approach, the tumour was totally removed (Fig. 2). The opening of the sphenoid sinus was closed with several pieces of temporalis muscle and temporalis fascia graft, and was covered with a pedunculated pericranial flap. The patient’s postoperative course was uneventful and the patient was free of disease when last seen in September 1998. Case 2 This 50-year-old man was discovered to have a sphenoid sinus tumour as an incidental finding on MRI scans. He denied symptoms of mucopurulent nasal discharge, epistaxis, anosmia or obstruction, and physical examination revealed a normal nasal cavity. A neurologic work up, including ophthalmologic examinations, was negative. On MRI the tumour was isointense to the cerebral cortex on both T1- and T2-weighted images (Fig. 3), and was homogeneously enhanced after the administration of contrast medium (Fig. 4A). A CT scan revealed a well defined, slightly hyperdense soft tissue mass within the sphenoid sinus, not associated with bony destruction. The patient underwent a sublabial trans-septal microscopic sphenoidotomy that revealed a friable and vascular soft tissue mass filling the sphenoid sinus. While intraoperative histological examination of biopsy specimens revealed the tumour to be a papilloma, it was only partly excised as the tumour visualisation was limited for total removal and there was massive haemorrhage from the tumour (Fig. 4B,C,D). After surgery the patient has done well with the residual tumour. Histological analysis on paraffin embedded specimens revealed the tumour to be an IP (Fig. 5). © 2001 Harcourt Publishers Ltd
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Fig. 3 Case 2. An MRI shows the tumour filling the sphenoid sinus, which is isointense both on T1-weighted (A) and T2-weighted (B) images. Note the intact pituitary gland.
Fig. 4 Case 2. The sphenoid sinus tumour is enhanced after the administration of Gd-DTPA (A: Preoperative coronal T1-weighted image). Postoperative T1-weighted coronal images with Gd-DTPA show the tumour to be partially removed.
pathologic entities of the lesion. Bony expansion or erosion on CT scan usually suggests a benign or a slow growing nature of the lesion, as was demonstrated in our cases, and bone destruction indicates a malignant nature.13 While IP and sinusitis may appear similar on a CT scan, the MRI scan may help in differentiating these two lesions. Sinusitis is usually isointense on T1-weighted images and hyperintense on T2-weighted images, and IP is isointense both on T1- and T2-weighted images,12 as demonstrated here. For many years surgical access to the sphenoid sinus, which is located deep in the geometric centre of the head and surrounded by vital structures, was problematic.14 However, the development of the nasal endoscope has made the approach to the sphenoid sinus both easier and safer.15 This procedure provides excellent visualisation within the nasal cavity and direct access to the natural ostium of the sphenoid sinus, and is an effective modality for diagnosis and treatment of most sphenoid sinus disorders.1,12 While this procedure was initially chosen for biopsy of the tumour in case 1, the nasal endoscopic approach potentially provides adequate tumour removal – even in large tumours – if they can be visualised. Among other approaches for the sphenoid sinus, including the transethmoidal and transpalatal routes, the most popular one has been the sublabial trans-septal sphenoidotomy.1 Through this approach, however, we could only partially resect the sphenoid sinus tumour of case 2 because of insufficient visualisation of the tumour and severe haemorrhage from the tumour. Any IP that is extensive and is outside the visualisation of the operating microscope may be treated by other routes.12 The tumour of case 1, which involved both the sphenoid and the posterior ethmoid sinuses, was totally removed through a midline transcranial approach. Regardless of the surgical approach, the key to successful treatment of IP remains wide surgical excision under optimal visualisation,2,3,12 since IP is characterised by its tendency to recur and association with malignant evolution. In addition, conventional radiotherapy for IP has been shown to be of little success.2,3,16–20 For a radical removal of the sphenoid sinus IP, an image-guided navigation technique may be of help. REFERENCES 1. 2.
Fig. 5 Photomicrographs of the tumour. A low power micrograph shows the hyperplastic epithelium inverting into the underlying stroma (A: case 1, H&E,70). A high power micrograph shows pseudostratified ciliated columnar lining (B: case 2, H&E,120).
3.
4.
DISCUSSION IP is an uncommon but well described neoplasm, with the overall incidence of this tumour varying from 0.5 to 4% of all primary nasal tumours.4 IP most commonly arises from the lateral nasal wall3 and rarely develops primarily within a sinus cavity.5–7 While we have presented two cases of IP arising in the sphenoid sinus, only a few such cases have previously been described.2,3,8 The most common complaints in patients with IP are nasal obstruction, followed by mucopurulent nasal discharge, epistaxis, anosmia, and facial pain.6,9 However, symptoms of patients with disease localised to the sphenoid sinus are initially quite subtle and vague.2 In our two cases, sphenoid sinus tumours were found incidentally on neuro-imaging studies, and no clinical signs or symptoms related to the tumour were apparent. Although there are only a limited number of reports on the CT and MRI features of IP, neuro-imaging studies play an important role in locating and defining the specific site and extent of this tumour.1–3,10–12 In addition, it may help to characterise histo© 2001 Harcourt Publishers Ltd
5. 6.
7. 8. 9. 10. 11. 12.
13. 14.
Hadar T, Yaniv E, Shvero J. Isolated sphenoid sinus changes – history, CT and endoscopic finding. J Laryngol Otol 1996; 110: 850–853. McElveen JT Jr, Fee WE Jr. Inverting papilloma of the sphenoid sinus. Otolaryngol Head Neck Surg 1981; 89: 710–712. Peters BW, O’Reilly RC, Willcox TO Jr, Rao VM, Lowry LD, Keane WM. Inverted papilloma isolated to the sphenoid sinus. Otolaryngol Head Neck Surg 1995; 113: 771–777. Myers E, Fernau J, Johnson J, Tabet J, Barnes L. Management of inverted papilloma. Laryngoscope 1990; 100: 481–490. Lawson W, Biller H, Jacobson A, Som P. The role of conservative surgery in the management of inverted papilloma. Laryngoscope 1983; 93: 148–155. Phillips P, Gustafson R, Facer G. The clinical behavior of inverting papilloma of the nose and paranasal sinuses: report of 112 cases and review of the literature. Laryngoscope 1990; 100: 463–469. Suh KW, Facer GW, Devine KD, Weiland LH, Zujko RD. Inverting papilloma of the nose and paranasal sinuses. Laryngoscope 1977; 87: 35–46. Edison BD. Primary inverting papilloma of the sphenoid sinus. Trans Am Acad Ophthalmol Otolaryngol 1975; 80: 434–437. Kamal S. Inverted papilloma of the nose. J Laryngol Otol 1981; 95: 1069–1079. Myers EN, Schramm V Jr, Barnes E Jr. Management of inverted papilloma of the nose and paranasal sinuses. Laryngoscope 1981; 91: 2071–2084. Sethi DS, Lau DP, Chee LW, Chong V. Isolated sphenoethmoid recess polyps. J Laryngol Otol 1998; 112: 660–663. Stankiewicz JA, Girgis SJ. Endoscopic surgical treatment of nasal and paranasal sinus inverted papilloma. Otolaryngol Head Neck Surg 1993; 109: 988–995. Dubois PJ, Schultz JC, Perrin RL Dastur KJ. Tomography in expansile lesions of the nasal and paranasal sinuses. Radiology 1977; 125: 149–158. Donald PJ, Boggan JE. Sphenoid sinus malignancies. J Craniofacial Surg 1995; 6: 15–23.
Journal of Clinical Neuroscience (2001) 8(2)
JOCN-126.QXD
2/25/01 3:54 PM
Page 170
170 Vinas et al. 15. 16.
17. 18. 19. 20.
spinal neuraxis, typically at the craniocervical junction. Patients with this condition may require conservative or surgical treatment, depending on the severity and progression of the symptoms. In recent years, new neuroimaging techniques have revived the interest in this entity, with respect to indications and results of treatment. With the widespread use of magnetic resonance scanning, lesions in the spinal cord such as syrinx or hydromyelia can be diagnosed earlier, often before producing clinical symptoms. The natural history of syrinx, however, is not well understood. The authors present a case of syringomyelia with spontaneous remission and clinical improvement. Similar cases reported in the literature were collected and analysed.
Wigand ME. Transnasal ethmoidectomy under endoscopic control. Rhinology 1981; 19: 7–15. Dolgin S, Zaveri V, Casiano R, Maniglia A. Different options for treatment of inverting papilloma of the nose and paranasal sinuses: a report of 41 cases. Laryngoscope 1992; 102: 231–236. Fechner RE, Alford DO. Inverted papilloma and squamous carcinoma: An unusual case. Arch Otolaryngol 1968; 88: 507–512. Vrabec DP. The inverted Schneiderian papilloma: a clinical and pathological study. Laryngoscope 1975; 85: 186–220. Cummings CW, Goodman ML. Inverted papillomas of the nose and paranasal sinuses. Arch Otolaryngol 1970; 92: 445–449. Norris HJ. Papillary lesions of the nasal cavity and paranasal sinuses. II. Inverting papillomas: A study of 29 cases. Laryngoscope 1973; 73: 1–17.
METHODS A Medline search in the English, French and Spanish literature performed for the years 1966 to August 1999, and review of bibliographic citations for additional cases prior to 1966, yielded seven patients with syrinx which spontaneously resolved without surgical intervention (Table 1). The analysis included demographic, clinical and radiological features, non-surgical therapeutic interventions, and time of follow up.
Spontaneous resolution of a syrinx Federico C. Vinas MD, Julie Pilitsis MD, Harvey Wilner MD Department of Neurological Surgery and Radiology, Wayne State University, School of Medicine, Detroit, MI
CASE REPORT Summary Syrinx are often related to trauma, tumours or abnormalities of the craniocervical junction. Only a few cases of spontaneous resolution have been reported in the literature. The authors present a case of spontaneous resolution of a cervical syrinx, and analyse the possible physiopathological mechanisms leading to resolution. Other similar cases reported in the literature were collected and analysed. © 2001 Harcourt Publishers Ltd Journal of Clinical Neuroscience (2001) 8(2), 170–172 © 2001 Harcourt Publishers Ltd DOI: 10.1054/jocn.2000.0768, available online at http://www.idealibrary.com on
Keywords: hydromyelia, syrinx, natural history, treatment Received 2 February 2000 Accepted 16 March 2000 Correspondence to: F. C. Vinas, Tel.: 904 253 7644; Fax: 904 252 1776
A 58-year-old male presented with a 3-month history of neck pain and pain and paraesthesias in his right upper extremity. Past medical history was significant for cataract on his left eye. He did not have a history of previous trauma. On physical examination, he had normal muscle tone and strength, and no myelopathic signs. There were some patchy areas of decreased sensation to light touch and pin prick in both upper extremities which did not follow any dermatome. A magnetic resonance image (MRI) of the cervical spine showed protrusion of the C4–C5 disc, and a central syrinx extending from C-6 to T-1 (Fig. 1). There was no enhancement after the administration of gadolinium DTPA. The patient was subsequently lost to follow up. His pain gradually improved over time. A follow up MRI was obtained 5 years later and showed complete disappearance of the syrinx (Fig. 2). There was no abnormal sign intensity in the spinal cord. The C4–C5 disc bulge appeared unchanged.
INTRODUCTION DISCUSSION The term syrinx includes a group of conditions associated with the presence of a cavity in the spinal cord. This process is usually associated with trauma or congenital abnormalities along the
Table 1
Syringomyelia was first recognized as a disease process approximately 400 years ago but the term syringomyelia or cystic
Clinical and demographic characteristics of patients with spontaneous resolution of syringomyelia reported in the literature
Author
Age/sex
Presentation
Diagnosis
Location
Assoc Chiari
Clinical course
Follow-up Ref
Sudo et al. (1990)
16 M
MRI
Cervicothoracic
Yes
Improvement
2 years
15
Jack et al. (1991)
30 F
MRI
Cervicothoracic
Yes
Little improvement
2 years
14
Olivero et al. (1992)
28 F
MRI
Cervical
Yes
Improvement
2 months
16
Yeager and Lusser (1992) Santoro et al. (1993)
19m F 42 M
MRI MRI
Cervical Cervicothoracic
No Yes
No symptoms Unchanged
2 years 3 years
17 13
Santoro et al. (1993) Pierallini et al. (1997) Present case
31 F 25 F 58 M
Left side hypalgesia, hypoaesthesia Paresthesias, decrease sensation on chest wall and left upper extremity Post-traumatic bilateral arm weakness Seizures, normal examination Paraesthesias, gait disturbance, atrophy of hand muscles Incidental finding Headaches, dizziness Pain on right upper extremity
MRI MRI MRI
Cervicothoracic Cervical Cervicothoracic
Yes Yes No
Asymptomatic Improvement Improvement
3 years 4 years 5 years
13 11
F, female; M, male; m, months.
Journal of Clinical Neuroscience (2001) 8(2)
© 2001 Harcourt Publishers Ltd