Spontaneous sphenoid sinus meningocele with associated amenorrhea and headache: illustrative case

Deveney Franklin School of Medicine, University of North Carolina, Chapel Hill, North Carolina

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Ali S Yamani College of Medicine, University of Cincinnati, Cincinnati, Ohio

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Andre E Boyke Departments of Neurosurgery

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Simon A Menaker Departments of Neurosurgery

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Dennis Tang Otolaryngology, Cedars-Sinai Medical Center, Los Angeles, California

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Adam N Mamelak Departments of Neurosurgery

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BACKGROUND

Developmental meningoceles of the sphenoid sinus are uncommon. When encountered, they are often associated with cerebrospinal fluid (CSF) rhinorrhea.

OBSERVATIONS

The authors present the case of a 27-year-old female with a large meningocele eroding through the sella turcica and sphenoid sinus into the nasopharynx. The patient presented with intractable headaches and amenorrhea without CSF rhinorrhea.

LESSONS

The patient underwent an endoscopic endonasal transsphenoidal reduction of the meningocele with reelevation of the pituitary gland and skull base reconstruction with abdominal fat graft and nasoseptal flap.

ABBREVIATIONS

CSF = cerebrospinal fluid; MRI = magnetic resonance imaging

BACKGROUND

Developmental meningoceles of the sphenoid sinus are uncommon. When encountered, they are often associated with cerebrospinal fluid (CSF) rhinorrhea.

OBSERVATIONS

The authors present the case of a 27-year-old female with a large meningocele eroding through the sella turcica and sphenoid sinus into the nasopharynx. The patient presented with intractable headaches and amenorrhea without CSF rhinorrhea.

LESSONS

The patient underwent an endoscopic endonasal transsphenoidal reduction of the meningocele with reelevation of the pituitary gland and skull base reconstruction with abdominal fat graft and nasoseptal flap.

ABBREVIATIONS

CSF = cerebrospinal fluid; MRI = magnetic resonance imaging

Meningoceles in the sphenoid sinus usually occur secondary to trauma, skull base erosion from inflammation, or iatrogenic injury. Spontaneously occurring or developmental meningoceles are extremely rare, and their etiology is not well understood. Proposed theories include incomplete fusion of the embryological precursors to the greater sphenoid wing and increased intracranial pressure, among others.1,2 The most common presenting symptoms in adulthood include headache, meningitis, and cerebrospinal fluid (CSF) rhinorrhea. Diagnosis is confirmed with computed tomography and/or magnetic resonance imaging.

Surgical intervention is often recommended in patients with sphenoid sinus meningoceles with the goal of preventing complications such as meningitis, pneumocephalus, and herniation of critical intracranial structures.1,3 The endoscopic endonasal approach provides excellent visualization of this pathology, allowing for early identification and protection of adjacent anatomical structures with decreased morbidity compared to traditional open cranial approaches. In this report, we present a rare case of a spontaneous meningocele of the sphenoid sinus and nasopharynx presenting with amenorrhea and headache and undergoing endoscopic endonasal transsphenoidal repair.4

Illustrative Case

A 27-year-old female presented to our institution for further evaluation of a known sellar lesion. The lesion had been discovered on magnetic resonance imaging (MRI) when the patient was 16 years old, as part of a workup for secondary amenorrhea. At the time, radiographic reports described the lesion as cystic, and the differential diagnosis included but was not limited to a Rathke cleft cyst or herniation of the third ventricle through the sella (Fig. 1). Additionally, the patient presented with mild hyperprolactinemia of 68.8 µg/L. After pharmacological treatment with cabergoline, the patient had a return of menses.

FIG. 1
FIG. 1

Original coronal (left) and sagittal (right) MRI from 2013 showing a cystic lesion.

On initial presentation to our institution, she reported chronic intractable headaches with gradual worsening over the past 3 years. She was amenorrheic, because she had stopped taking cabergoline because of insurance issues. She reported prior syncopal episodes as well as constant fatigue. She did not exhibit visual deficits, and visual field testing was full of confrontation. Her neurological examination was without focal deficits, and she had no history of CSF rhinorrhea. Labs were drawn after restarting a half dose of cabergoline, as recommended by endocrinology, and showed a prolactin value of 48.4 ng/mL. MRI was performed and demonstrated a large cystic lesion measuring 13 × 38 × 16 mm, extending from the suprasellar cistern, and eroding through both the floor of the sella turcica and the sphenoid sinus with protrusion into the soft tissue just above the nasopharynx (Fig. 2A). Imaging also revealed inferior retraction of the optic chiasm as well as herniation and flattening of the pituitary gland inferiorly to the base of the sphenoid sinus (Fig. 2B). There was no increased signal intensity within the lesion to suggest the lesion was a Rathke cleft cyst and no obvious cortical tissue to suggest an encephalocele (Fig. 2C).

FIG. 2
FIG. 2

A: Cystic lesion with extension through the floor of the sella turcica and filling the sphenoid sinus. B: Downward traction of the cyst caused inferior retraction of the optic chiasm, stretching of the pituitary stalk, and flattening of the pituitary gland at the base of the sphenoid. C: Signal intensity within the cystic lesion was consistent with CSF.

Despite the long-standing and unchanged appearance of this lesion, a decision was made to operate given the long-term risk for CSF leakage over the patient’s lifetime, as well as the possibility of this contributing to her headaches. The patient underwent an endoscopic endonasal transsphenoidal reduction of the cyst with elevation of the pituitary gland into the sella. Fibrous bands between the gland and the surrounding tissues were freed up so the gland itself could be elevated, and the remaining defect was obliterated with an abdominal fat graft, covered with a nasoseptal flap (Fig. 3). A lumbar subarachnoid drain was placed to reduce pressure and was subsequently drained for 3 days postoperatively.

FIG. 3
FIG. 3

Intraoperative endoscopic images of the meningocele and final repair. A: Meningocele after the sphenoid roof was removed (prior to opening). B: Meningocele cavity after the pituitary gland was freed up and elevated. C: Fat graft placed to obliterate the meningocele. D: Nasoseptal flap covering the meningocele.

After surgery, the patient was clinically stable with unrevealing visual field and neurological examinations and no evidence of CSF leakage. She was discharged after 3 days. Postoperative MRI showed a significant reduction in the size of the meningocele with a small amount of pneumocephalus within the right lateral ventricle (Fig. 4). Headaches were reduced at the 1-month follow-up with persistent amenorrhea, and no other issues were noted. Serial prolactin labs at 1 month and 4 months postoperatively were 0.9 and 15.4 ng/mL, respectively, with cabergoline discontinued. Postoperative MRI at 3 months was stable.

FIG. 4
FIG. 4

Postoperative axial (A), coronal (B), and sagittal (C) T2-weighted MRI sequences showed a reduction of the meningocele with the fat graft in place obliterating much of the sphenoid sinus.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

Etiology and Demographics

A meningocele is a herniation of the meninges through a defect in the skull or spine. They are classified based on the location of the herniation and osseous defect such as basal, frontal, parietal, or occipital. Basal meningoceles involving the sphenoid sinus can be further classified into intrasphenoidal (contained within the sphenoid sinus) or transsphenoidal (protrusion through the sinus into the nasopharynx).3 The exact etiology of transsphenoidal meningoceles is unknown; spontaneous, iatrogenic, traumatic, and congenital cases have been reported.3

We conducted a comprehensive review of the literature using PubMed and MeSH terms “transsphenoidal meningocele” and “sphenoidal meningocele”; to our knowledge, there are only 19 reported cases of sphenoidal meningocele. Among the 18 prior documented cases, 72.2% of patients were female and 27.8% were male with an age range of 22 to 73 years. The most common presenting symptom was CSF rhinorrhea (83.3%). There was only one other case in addition to ours presenting with amenorrhea. All patients underwent surgical repair with 11% undergoing a frontotemporal approach and 89% undergoing endoscopic endonasal approach. There were no documented complications. A summary of cases including ours can be found in Table 1.

TABLE 1

Literature review and epidemiological data of documented sphenoid meningoceles

Authors & YearSample SizeSexAverage Age (yrs)Presenting SymptomsTreatmentComplications
Zoli et al., 201617 12M: 3 (37%), F: 9 (63%)53CSF rhinorrhea: 11/12 (91.6%), headaches: 5/12 (41.6%), meningitis: 2/12 (16.6%); subjects had overlapping symptomsEENone
Rozzi et al., 20203 1F38HeadacheEEUnknown
Yıldırım et al., 20141 1M51CSF rhinorrheaEENone
Chandonnet et al., 200815 1F22Amenorrhea & hypogonadismEEUnknown
Firat et al., 200418 1M53CSF rhinorrheaBilat frontotemporal transcranialNone
Schick et al., 20009 1F29CSF rhinorrheaFronto-temporalNone
Blaivie et al., 200619 1F73CSF rhinorrheaEENone
Current reported patient1F27AmenorrheaEENone

EE = endoscopic endonasal.

Differential Diagnosis

The differential diagnosis for a sellar cystic lesion like the one reported herein is broad, including Sternberg’s canal defects, empty sella syndrome, arachnoid cyst, and sphenoid sinus meningocele, among others.

The most common causes of CSF leakage are trauma and iatrogenic injury. Spontaneous CSF fistulas are less common and account for 6% to 23% of all CSF leaks.5–7 The cribriform plate and fovea ethmoidalis are the most common sites for CSF leaks, although spontaneous leaks are more often located in the sphenoid sinus.6,7 Sphenoid sinus CSF leaks have been attributed to defects in the upper roof of the lateral recess, the parasellar area, the roof of the sphenoid sinus and the presence of a patent lateral craniopharyngeal canal (Sternberg’s canal).7 The canal originates posteriorly between the body of the sphenoid bone and the lesser sphenoid wing and then passes along the median border of the sphenoid sinus medial to the foramen rotundum and vidian canal before terminating anteriorly in the vaginal process of the nasopharynx.6–8 During embryonic development, the anterior and posterior sphenoid bone, greater and lesser sphenoid wings, and lateral pterygoid processes initially form as cartilaginous precursors.9 These precursors subsequently undergo ossification and ultimately fuse to form the sphenoid bone.9 However, incomplete fusion of sphenoid bone components can result in the formation of bony defects such as a persistent Sternberg’s canal through which meninges and brain tissue can herniate.6,9,10 It should be noted that the literature is divided regarding the role of Sternberg’s canal in the development of sphenoid sinus encephaloceles and CSF leakage. Some studies identify the canal as a possible site of these pathologies6,9 whereas other studies find this less likely, because most sphenoid CSF leaks occur lateral to the foramen rotundum.7 Furthermore, some authors argue that altered CSF flow dynamics and increased intracranial pressure more likely influence the pathogenesis of sphenoid sinus CSF leaks, citing the presence of arachnoid pits, an empty sella, and extensive lateral pneumatization of the sphenoid in patients with spontaneous CSF leakage.5–7

Empty sella syndrome develops when CSF fills the sella turcica and compresses the pituitary gland. In adults, this condition is most often found in older, obese, multiparous women and is often asymptomatic. Symptoms can occur in adults secondary to intracranial hypertension. If an empty sella syndrome occurs in childhood, it is often associated with endocrinopathies such as growth hormone deficits and hypogonadism.11 Empty sella syndrome is commonly diagnosed on brain MRI or CT in the absence of an obvious pituitary gland. Therapeutic management of an empty sella syndrome includes the correction of endocrinopathies and the treatment of intracranial hypertension, with surgical intervention as only a last resort. Empty sella syndrome should be distinguished from other pituitary abnormalities that may require more acute neurosurgical intervention.12

An intrasellar arachnoid cyst is another rare, benign malformation that may be considered when evaluating a sellar cystic lesion. Visual disturbance is the most common presenting symptom, which can manifest as decreased visual acuity, optic atrophy, or bitemporal hemianopia. Skull radiography and CT can reveal thinning of the bone of the sella floor and cystic lesions occupying the intrasellar and suprasellar regions.13 On MRI, the cystic contents are isointense to CSF. Although intrasellar arachnoid cysts rarely require treatment, surgical management via a transsphenoidal approach has been documented.14

In our case, given the presence of the pituitary gland on MRI and protrusion of the lesion through the sella turcica, which is uncommon for an arachnoid cyst, the most likely diagnosis for the patient was a sphenoid sinus meningocele. Symptomatic presentation is not often associated with endocrinopathies such as amenorrhea and has only been reported in one other case in the literature.15 We suspect our patient’s endocrinopathy was related to a stalk effect rather than compression of the pituitary gland itself. She carried a presumptive diagnosis of polycystic ovarian syndrome as well for many years, which may have been a primary driver of her amenorrhea. Additionally, our patient reported headaches, which could have been attributed to intracranial hypertension, although there was no objective measurement or radiographic demonstration of this.

Lessons

Treatment and Prognosis

Current data in the literature support surgical intervention in symptomatic patients with the optimal approach being endoscopic endonasal transsphenoidal repair with lumbar drain placement for CSF diversion, because CSF leakage is a common complication.3 As seen in Table 1, only 11% of patients underwent a frontotemporal or pterional approach. Cranial approaches can cause anosmia, frontal lobe injury, and intracranial hemorrhage.1 No major complications were reported in the literature, and most patients endorsed improved or stable symptoms postoperatively, our patient included.16 Given the rarity of these lesions, recurrence rates are unknown.

Spontaneous transsphenoidal meningoceles are uncommon and can present with endocrinopathies such as amenorrhea, but they should be considered in the differential diagnosis when sellar lesions are encountered. Surgical intervention via a transsphenoidal approach is the most common treatment for symptomatic patients, with generally positive results.

Author Contributions

Conception and design: Yamani, Boyke, Menaker, Tang. Acquisition of data: Franklin, Yamani, Boyke, Tang, Mamelak. Analysis and interpretation of data: Franklin, Yamani, Boyke, Mamelak. Drafting the article: Franklin, Yamani, Boyke, Menaker. Critically revising the article: all authors. Reviewed submitted version of manuscript: Franklin, Yamani, Boyke, Menaker, Mamelak. Approved the final version of the manuscript on behalf of all authors: Franklin. Statistical analysis: Boyke. Administrative/technical/material support: Boyke. Study supervision: Boyke, Tang, Mamelak.

References

  • 1

    Yıldırım AE, Dıvanlıoglu D, Cetinalp NE, Belen AD Endoscopic endonasal repair of spontaneous sphenoid sinus lateral wall meningocele presenting with cerebrospinal fluid leak. J Neurosci Rural Pract. 2014;5(2):168170.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2

    Herman P, Sauvaget E, Guichard JP, Tran Ba Huy P Intrasphenoidal transsellar encephalocele repaired by endoscopic approach. Ann Otol Rhinol Laryngol. 2003;112(10):890893.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Rozzi R, Behringer J, Obajuluwa A, Wilczynski M Spontaneous unilateral intrasphenoidal meningocele. J Radiol Case Rep. 2020;14(8):17.

  • 4

    Hegazy HM, Carrau RL, Snyderman CH, Kassam A, Zweig J Transnasal endoscopic repair of cerebrospinal fluid rhinorrhea: a meta-analysis. Laryngoscope. 2000;110(7):11661172.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Settecase F, Harnsberger HR, Michel MA, Chapman P, Glastonbury CM Spontaneous lateral sphenoid cephaloceles: anatomic factors contributing to pathogenesis and proposed classification. AJNR Am J Neuroradiol. 2014;35(4):784789.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    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 Malo Inwazyjne. 2015;10(2):347358.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Hanz SZ, Arko L 4th, Schmidt F, et al. Low incidence of true Sternberg’s canal defects among lateral sphenoid sinus encephaloceles. Acta Neurochir (Wien). 2020;162(10):24132420.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Sternberg M A previously undescribed canal in the sphenoid bone of humans. Anat Anz. 1888;3:784785.

  • 9

    Schick B, Brors D, Prescher A Sternberg’s canal—cause of congenital sphenoidal meningocele. Eur Arch Otorhinolaryngol. 2000;257(8):430432.

  • 10

    Gisselsson L Intranasal forms of encephalomeningocele. Acta Otolaryngol. 1947;35:56, 519–531.

  • 11

    Lenz AM, Root AW Empty sella syndrome. Pediatr Endocrinol Rev. 2012;9(4):710715.

  • 12

    Chiloiro S, Giampietro A, Bianchi A, De Marinis L Empty sella syndrome: multiple endocrine disorders. Handb Clin Neurol. 2021;181:2940.

  • 13

    Fujiwara M, Bitoh S, Hasegawa H, Ohtsuki H [A case of intrasellar arachnoid cyst]. No Shinkei Geka. 1984;12(3 suppl):331337.

  • 14

    Sasaki N, Tani S, Funakoshi Y, Imamura H, Fukumitsu R, Sakai N Endoscopic management of an intrasellar arachnoid cyst through the tuber cinereum in an adult: a case report. Acta Neurochir (Wien). 2020;162(10):23972401.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15

    Chandonnet M, Fisch C, Schmutz GR Intrasphenoidal meningocele and osteopetrosis: a case report. Article in French. J Radiol. 2008;89(2):255257.

  • 16

    Bhaisora KS, Das KK, Jamdar J, et al. Trans-sellar trans-sphenoidal herniation of third ventricle with cleft palate and microophthalmia: report of a case and review of literature. Asian J Neurosurg. 2018;13(3):782785.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Zoli M, Farneti P, Ghirelli M, et al. Meningocele and meningoencephalocele of the lateral wall of sphenoidal sinus: the role of the endoscopic endonasal surgery. World Neurosurg. 2016;87:9197.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Firat AK, Firat Y Spontaneous bilateral intrasphenoidal lateral encephaloceles: CT and MRI findings. Ear Nose Throat J. 2004;83(12): 831833.

  • 19

    Blaivie C, Lequeux T, Kampouridis S, Louryan S, Saussez S Congenital transsphenoidal meningocele: case report and review of the literature. Am J Otolaryngol. 2006;27(6):422424.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • Collapse
  • Expand
  • FIG. 1

    Original coronal (left) and sagittal (right) MRI from 2013 showing a cystic lesion.

  • FIG. 2

    A: Cystic lesion with extension through the floor of the sella turcica and filling the sphenoid sinus. B: Downward traction of the cyst caused inferior retraction of the optic chiasm, stretching of the pituitary stalk, and flattening of the pituitary gland at the base of the sphenoid. C: Signal intensity within the cystic lesion was consistent with CSF.

  • FIG. 3

    Intraoperative endoscopic images of the meningocele and final repair. A: Meningocele after the sphenoid roof was removed (prior to opening). B: Meningocele cavity after the pituitary gland was freed up and elevated. C: Fat graft placed to obliterate the meningocele. D: Nasoseptal flap covering the meningocele.

  • FIG. 4

    Postoperative axial (A), coronal (B), and sagittal (C) T2-weighted MRI sequences showed a reduction of the meningocele with the fat graft in place obliterating much of the sphenoid sinus.

  • 1

    Yıldırım AE, Dıvanlıoglu D, Cetinalp NE, Belen AD Endoscopic endonasal repair of spontaneous sphenoid sinus lateral wall meningocele presenting with cerebrospinal fluid leak. J Neurosci Rural Pract. 2014;5(2):168170.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2

    Herman P, Sauvaget E, Guichard JP, Tran Ba Huy P Intrasphenoidal transsellar encephalocele repaired by endoscopic approach. Ann Otol Rhinol Laryngol. 2003;112(10):890893.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Rozzi R, Behringer J, Obajuluwa A, Wilczynski M Spontaneous unilateral intrasphenoidal meningocele. J Radiol Case Rep. 2020;14(8):17.

  • 4

    Hegazy HM, Carrau RL, Snyderman CH, Kassam A, Zweig J Transnasal endoscopic repair of cerebrospinal fluid rhinorrhea: a meta-analysis. Laryngoscope. 2000;110(7):11661172.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Settecase F, Harnsberger HR, Michel MA, Chapman P, Glastonbury CM Spontaneous lateral sphenoid cephaloceles: anatomic factors contributing to pathogenesis and proposed classification. AJNR Am J Neuroradiol. 2014;35(4):784789.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    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 Malo Inwazyjne. 2015;10(2):347358.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Hanz SZ, Arko L 4th, Schmidt F, et al. Low incidence of true Sternberg’s canal defects among lateral sphenoid sinus encephaloceles. Acta Neurochir (Wien). 2020;162(10):24132420.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Sternberg M A previously undescribed canal in the sphenoid bone of humans. Anat Anz. 1888;3:784785.

  • 9

    Schick B, Brors D, Prescher A Sternberg’s canal—cause of congenital sphenoidal meningocele. Eur Arch Otorhinolaryngol. 2000;257(8):430432.

  • 10

    Gisselsson L Intranasal forms of encephalomeningocele. Acta Otolaryngol. 1947;35:56, 519–531.

  • 11

    Lenz AM, Root AW Empty sella syndrome. Pediatr Endocrinol Rev. 2012;9(4):710715.

  • 12

    Chiloiro S, Giampietro A, Bianchi A, De Marinis L Empty sella syndrome: multiple endocrine disorders. Handb Clin Neurol. 2021;181:2940.

  • 13

    Fujiwara M, Bitoh S, Hasegawa H, Ohtsuki H [A case of intrasellar arachnoid cyst]. No Shinkei Geka. 1984;12(3 suppl):331337.

  • 14

    Sasaki N, Tani S, Funakoshi Y, Imamura H, Fukumitsu R, Sakai N Endoscopic management of an intrasellar arachnoid cyst through the tuber cinereum in an adult: a case report. Acta Neurochir (Wien). 2020;162(10):23972401.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15

    Chandonnet M, Fisch C, Schmutz GR Intrasphenoidal meningocele and osteopetrosis: a case report. Article in French. J Radiol. 2008;89(2):255257.

  • 16

    Bhaisora KS, Das KK, Jamdar J, et al. Trans-sellar trans-sphenoidal herniation of third ventricle with cleft palate and microophthalmia: report of a case and review of literature. Asian J Neurosurg. 2018;13(3):782785.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Zoli M, Farneti P, Ghirelli M, et al. Meningocele and meningoencephalocele of the lateral wall of sphenoidal sinus: the role of the endoscopic endonasal surgery. World Neurosurg. 2016;87:9197.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Firat AK, Firat Y Spontaneous bilateral intrasphenoidal lateral encephaloceles: CT and MRI findings. Ear Nose Throat J. 2004;83(12): 831833.

  • 19

    Blaivie C, Lequeux T, Kampouridis S, Louryan S, Saussez S Congenital transsphenoidal meningocele: case report and review of the literature. Am J Otolaryngol. 2006;27(6):422424.

    • PubMed
    • Search Google Scholar
    • Export Citation

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