- Department of Neurosurgery, Juntendo University Shizuoka Hospital, Izunokuni, Japan
- Department of Neurosurgery, Juntendo University Urayasu Hospital, Urayasu, Japan.
Correspondence Address:
Satoshi Adachi, Department of Neurosurgery, Juntendo University Shizuoka Hospital, Izunokuni, Japan.
DOI:10.25259/SNI_260_2023
Copyright: © 2023 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.How to cite this article: Satoshi Adachi1, Hideaki Ueno2, Shunsuke Magami1, Naohide Fujita1, Shintaro Nakajima1, Ryogo Ikemura1, Yasuhito Ueki1, Yuki Takaki1, Keisuke Murofushi1, Yasuaki Nakao1, Takuji Yamamoto1. Cerebrospinal fluid rhinorrhea with meningoencephalocele related to Sternberg’s canal: A report of two cases. 30-Jun-2023;14:228
How to cite this URL: Satoshi Adachi1, Hideaki Ueno2, Shunsuke Magami1, Naohide Fujita1, Shintaro Nakajima1, Ryogo Ikemura1, Yasuhito Ueki1, Yuki Takaki1, Keisuke Murofushi1, Yasuaki Nakao1, Takuji Yamamoto1. Cerebrospinal fluid rhinorrhea with meningoencephalocele related to Sternberg’s canal: A report of two cases. 30-Jun-2023;14:228. Available from: https://surgicalneurologyint.com/surgicalint-articles/12384/
Abstract
Background: Cerebrospinal fluid (CSF) rhinorrhea with meningoencephalocele (MEC) associated with Sternberg’s canal is rare. We treated two such cases.
Case Description: A 41-year-old man and a 35-year-old woman presented with CSF rhinorrhea and mild headache worsening with standing posture. Head computed tomography showed a defect close to the foramen rotundum in the lateral wall of the left sphenoid sinus in both cases. Head magnetic resonance (MR) imaging and MR cisternography revealed that brain parenchyma had herniated into the lateral sphenoid sinus through the defect of the middle cranial fossa. The intradural and extradural spaces and bone defect were sealed with fascia and fat through both intradural and extradural approaches. The MEC was cut away to prevent infection. CSF rhinorrhea completely stopped after the surgery.
Conclusion: Our cases were characterized by empty sella, thinning of the dorsum sellae, and large arteriovenous malformations that suggest chronic intracranial hypertension. The possibility of Sternberg’s canal in patients with CSF rhinorrhea with chronic intracranial hypertension should be considered. The cranial approach has the advantages of lower infection risk and the ability to close the defect with multilayer plasty under direct vision. The transcranial approach is still safe if performed by a skillful neurosurgeon.
Keywords: Arteriovenous malformation, Cerebrospinal fluid rhinorrhea, Lateral craniopharyngeal canal, Meningoencephalocele, Surgical repair
INTRODUCTION
Sternberg’s canal (lateral craniopharyngeal canal) was first described by Maximilian Sternberg in 1888,[
CASE REPORTS
Case 1
A 41-year-old man became aware of left-sided intermittent nasal discharge which gradually worsened and mild headache worsening with standing posture. He had no previous history of head trauma, medical disorders, or seizures. Clinical examination revealed left-sided CSF rhinorrhea, and biochemical tests showed protein 46 mg/ dL and glucose 75 mg/dL in the nasal discharge. The rhinorrhea worsened with cervical anteflexion. Head computed tomography (CT) showed a defect lateral to the foramen rotundum in the lateral wall of the left sphenoid sinus and erosion of the dorsum sellae [
Figure 1:
Case 1 - (a and b) Computed tomography scans showed a defect (white arrow, black arrow) in the lateral wall of the left sphenoid sinus lateral to the foramen rotundum (black arrowhead) and erosion of the dorsum sellae (asterisk). (c and d) Head T2-weighted magnetic resonance (MR) image (c) and MR cisternogram (d) revealed brain parenchyma had herniated into the lateral sphenoid sinus through the defect of the middle cranial fossa (white arrow). (e) Schema of meningoencephalocele and cerebrospinal fluid rhinorrhea associated with Sternberg’s canal.
Figure 2:
Case 1 - Intraoperative photographs showing the left frontotemporal craniotomy with temporal extension. (a) The intradural approach exposing middle cranial dura and brain tissue protruding into the defect in the middle skull base leading to the sphenoid sinus (arrow). (b) Herniated dura (arrow). (c) Dural defect was sutured through the extradural approach (arrow). (d) Defect in the lateral sphenoid sinus was 5 mm in size (arrow). (e-g) Intradural (e) and extradural spaces (f) and bone defect (g) were sealed with abdominal external oblique fascia containing fat, fibrin glue, and biological tissue reinforcement material.
Case 2
A 35-year-old woman suffered intermittent nasal discharge without seizures. She had a history of large arteriovenous malformations (AVMs) in the right frontal lobe. CT showed a defect in the floor of the sella turcica and AVMs in the right frontal lobe, with nidus of 5 cm [
Figure 3:
Case 2 - (a and b) Sagittal (a) and coronal computed tomography scans (b) showed a defect in the floor of the sella turcica (arrow). (c) Arteriovenous malformations in the right frontal lobe, with nidus of 5 cm (arrow). (d) Magnetic resonance cisternogram revealed abnormal fluid collection in the sphenoid sinus and the empty sella.
Figure 4:
Case 2 - (a and b) Computed tomography scans showed severe pneumocephalus and a defect in the lateral wall of the left sphenoid sinus between the foramen rotundum and foramen ovale (black arrow). (c and d) Axial (c) and coronal (d) magnetic resonance cisternogram showed that brain parenchyma had herniated into the lateral sphenoid sinus through the defect of the middle cranial fossa (white arrow).
Figure 5:
Case 2 - Intraoperative photographs of the second surgery. (a and b) Intradural (a) and extradural approaches (b) to the Stenberg’s canal. The cranial dura involved multiple holes and protruded with brain parenchyma (black arrow) into the skull base defect between the foramen rotundum and foramen ovale in the middle skull base leading to the sphenoid sinus. (c) Herniated dura and meningoencephalocele were cut away and the nasal mucosa (black arrow) exposed. (d) Bone defect was reconstructed with a titanium plate. (e-h) Intradural (e and f) and extradural spaces (g and h) including the bone defect were sealed with temporal fascia, abdominal fat, fibrin glue, and biological tissue reinforcement material.
DISCUSSION
Various classifications of CSF rhinorrhea have been reported. CSF rhinorrhea was divided into three categories as follows: (1) traumatic, (2) postsurgical, and (3) spontaneous.[
Sternberg’s canal (lateral craniopharyngeal canal) has been considered to be an underlying factor related to sphenoid sinus spontaneous CSF rhinorrhea.[
Rhinorrhea and MEC associated with Sternberg’s canal are rare.[
Treatment of MEC and CSF rhinorrhea consists of reconstruction through the transcranial or endoscopic approach. Herniated dura mater and brain parenchyma should be excised because these tissues are considered contaminated and functionless.[
CONCLUSION
Our cases were characterized by empty sella and thinning of the dorsum sellae which suggest chronic intracranial hypertension. Furthermore, our Case 2 had a long history of large AVMs and previous CSF rhinorrhea due to a defect in the floor of the sella turcica. The possibility of Sternberg’s canal in patients with spontaneous CSF rhinorrhea with chronic intracranial hypertension should be considered. The transcranial closure technique which requires multi-layered reconstruction might be safe and effective if performed by a skillful neurosurgeon. Intracranial pressure management and prolonged clinical follow-up are essential in patients with chronic intracranial hypertension.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Disclaimer
The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.
References
1. Bendersky DC, Landriel FA, Ajler PM, Hem SM, Carrizo AG. Sternberg’s canal as a cause of encephalocele within the lateral recess of the sphenoid sinus: A report of two cases. Surg Neurol Int. 2011. 2: 171
2. Cruz Silva V, Luís A, Féria RM, Marques L, Chorão M, Reizinho C. Transsphenoidal meningoencephalocele protruding into the nasal cavity. BJR Case Rep. 2017. 3: 20160082
3. Hanwate R, Thorawade V, Jagade M, Attakil A, Parelkar K, Pandare M. CSF rhinorrhea with encephalocele through Sternberg’s canal: Our experience. Int J Otolaryngol Head Neck Surg. 2015. 4: 50-4
4. Hanz SZ, Arko L, Schmidt F, Kacker A, Tsiouris AJ, Anand VK. Low incidence of true Sternberg’s canal defects among lateral sphenoid sinus encephaloceles. Acta Neurochir (Wien). 2020. 162: 2413-20
5. Khatri D, Singh S, Das KK, Jaiswal AK, Kumar R. Endoscopic repair of CSF rhinorrhea through persistent Sternberg’s canal in an elderly patient with ankylosing spondylitis. Glob Imaging Insights. 2016. 1: 5-6
6. Kirtane MV, Lall A, Chavan K, Satwalekar D. Endoscopic repair of lateral sphenoid recess cerebrospinal fluid leaks. Indian J Otolaryngol Head Neck Surg. 2012. 64: 188-92
7. Kwon JE, Kim E. Middle fossa approach to a temporosphenoidal encephalocele--technical note. Neurol Med Chir (Tokyo). 2010. 50: 434-8
8. Maselli G, Ricci A, Galzio RJ. Endoscope-assisted transsphenoidal approach for treatment of Sternberg’s canal. J Korean Neurosurg Soc. 2012. 52: 555-7
9. Ommaya AK, Di Chiro G, Baldwin M, Pennybacker JB. Non-traumatic cerebrospinal fluid rhinorrhoea. J Neurol Neurosurg Psychiatry. 1968. 31: 214-25
10. Rossi Izquierdo M, Martín CM, Caballer TL. Association between cerebrospinal fluid fistula and persistent Sternberg canal: coincidence or cause?. Acta Otolaryngol Esp. 2012. 63: 144-6
11. Samadian M, Moghaddasi H, Vazirnezami M, Haddadian K, Rezaee O, Armanfar M. Transcranial approach for spontaneous CSF rhinorrhea due to Sternberg’s canal intrasphenoidal meningoencephalocele: Case report and review of the literature. Turk Neurosurg. 2012. 22: 242-5
12. Sanjari R, Mortazavi SA, Amiri RS, Ardestani SH, Amirjamshidi A. Intrasphenoidal meningo-encephalocele: Report of two rare cases and review of literature. Surg Neurol Int. 2013. 4: 5
13. Schick B, Brors D, Prescher A. Sternberg’s canal--cause of congenital sphenoidal meningocele. Eur Arch Otorhinolaryngol. 2000. 257: 430-2
14. Sternberg M. A previously undescribed canal in the human sphenoid bone. Anat Anz. 1888. 3: 784-5
15. Tabaee A, Anand VK, Cappabianca P, Stamm A, Esposito F, Schwartz TH. Endoscopic management of spontaneous meningoencephalocele of the lateral sphenoid sinus. J Neurosurg. 2010. 112: 1070-7
16. Takabayashi K, Nagamine M, Fujita T. Encephalocele through Sternberg’s canal: A case report. Jpn J Rhinol. 2020. 59: 26-33
17. Tomaszewska M, Brożek-Mądry E, Krzeski A. Spontaneous sphenoid sinus cerebrospinal fluid leak and meningoencephalocele-are they due to patent Sternberg’s canal?. Wideochir Inne Tech Maloinwazyjne. 2015. 10: 347-58
18. Vrabec DP, Hallberg OE. Cerebrospinal fluid rhinorrhea. Intranasal approach, review of the literature, and report of a case. Arch Otolaryngol. 1964. 80: 218-29