European Annals of Otorhinolaryngology, Head and Neck diseases (2013) 130, 345—347

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CASE REPORT

Endoscopic endonasal management of congenitalintranasal meningocele in a 2-month-old infant

E. Gassaba,∗, N. Krifaa, S. Kedousa, A. Zrigb, N. Hattabc, K. Harrathia,J. Koubaaa, A. Gassaba

a Service d’ORL et de chirurgie cervico-faciale, CHU Fattouma-Bourguiba, rue 1er-Juin-1995, 5000 Monastir, Tunisiab Service de radiologie, CHU Fattouma-Bourguiba, rue 1er-Juin-1995, 5000 Monastir, Tunisiac Service de neurochirurgie, CHU Fattouma-Bourguiba, rue 1er-Juin-1995, 5000 Monastir, Tunisia

KEYWORDSMeningocele;Congenital;Skull base;Imaging;Endoscopic endonasalsurgery

SummaryIntroduction: Nasal meningocele, which is usually congenital, is a rare anomaly resulting frommeningeal herniation into the nasal cavities through a bone defect in the skull base.Case report: An 8-day-old boy was referred with respiratory disturbance and nasal obstruction.Examination showed a cyst-like grayish swelling filling the right nasal cavity. CT scan showedopacity, of fluid-like density, filling the right nasal fossa, in contact with a small bony defectin the right cribriform plate. MRI ruled out herniated brain parenchyma and enabled diagnosisof meningocele. The patient was operated on at the age of 2 months through a transnasalendoscopic approach. Immediate postoperative course was favorable. MRI control at 8 monthswas normal.Discussion: Modern imaging (CT scan and MRI) is of paramount importance in the preoperative

evaluation of nasal meningocele. Endoscopic endonasal (EE) surgery is currently the treatmentof choice.Conclusion: Steady progress in instrumentation, technique and skills will increase the feasibilityof skull-base surgery using an endonasal approach in the pediatric population.© 2013 Published by Elsevier Masson SAS.

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Introduction

Nasal meningocele, which is usually congenital, is a rareanomaly resulting from meningeal herniation into the nasalcavities through a bone defect in the skull base, often at an

anterior location [1].

Progress in endoscopic endonasal (EE) surgery has revo-lutionized the management of skull-base defects [2].

∗ Corresponding author. Tel.: +216 96 33 90 94.E-mail address: eliegas@voila.fr (E. Gassab).

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1879-7296/$ – see front matter © 2013 Published by Elsevier Masson SAShttp://dx.doi.org/10.1016/j.anorl.2011.10.013

The present study concerns the youngest patient as yeteported in the literature, successfully operated on forasal meningocele via an EE approach. The case illustrateshe clinical and therapeutic aspects of congenital nasaleningocele and highlights the interest of the EE technique.

ase report

n 8-day-old male neonate was referred with respiratory dis-urbance and nasal obstruction. Physical examination foundn enlarged nasal pyramid with cyst-like grayish formation

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346 E. Gassab et al.

Figure 1 Facial CT without contrast enhancement: sagittalreconstruction. Liquid-like mass almost entirely filling the rightna

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Figure 3 Postoperative T2-weighted MRI: sagittal slice. Skull-b(

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asal cavity (star) and communicating with the endocranium via bone defect of the ethmoid cribiform plate (arrow).

lling the entire right nasal cavity. The patient was free ofever.

The differential diagnoses considered were: lacrimaluct cyst, nasal glioma, and nasal meningocele.

Facial and cerebral CT and MRI established the diagnosiss transethmoidal meningocele (Figs. 1 and 2).

The patient was operated on at the age of 2 monthsollowing an anti-pneumococcal vaccination. Surgery usedn EE approach with a 2.7 mm diameter 0◦ rigid endo-cope and ear microsurgery instruments (EE instrumentationdapted to this age not being available). The meningoceleac was resected after puncture aspiration of its contentsnd partial middle turbinectomy. Ethmoid cribriform plateefect reconstruction required an assembly comprising two

ucosal flaps taken from the resected middle turbinate

one in extradural underlay and the other in overlay), andiologic glue (Glubran®) then Surgicel®. Lumbar drainageas not associated. Antibioprophylaxis and antihistamine

vied

igure 2 Preoperative T2-weighted MRI (a: sagittal slice; b: corontranasal hernia of a pure liquid sac limited by the meningeal struc

ase bone defect filled by a material in linear hyposignalarrow).

herapy were initiated. Postoperative course was favorablewithout cerebrospinal fluid [CSF] leakage or meningitis).he patient was discharged at 4 days post-surgery. ControlRI findings at 8 months were normal (Fig. 3). Over 2 years

egular follow-up, there was no CSF leakage or meningitis.

iscussion

he contents of nasal meningocele comprise meninges andSF; meningoencephalocele additionally contains cerebralarenchyma [1].

Clinical presentation is variable. Nasal meningoceleay present as meningitis, nasal obstruction, difficulty in

eeding, craniofacial deformity or cerebrospinal rhinorrhea2—4] or else be discovered serendipitously on radiology [2].

Preoperative imaging determines whether cerebral blood

essels are present in the herniated tissue and assessesntracranial connections [2,5]. The skull base should bexamined in its entirety, to screen for possible multipleefects [3].

nal slice). Ethmoid cribiform plate defect (6 mm × 4 mm) withtures, corresponding to a meningocele.

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Endoscopic endonasal management of congenital intranasal

CT provides detailed exploration of skull-base bonestructures, whereas MRI provides better soft-tissue andCSF exploration, distinguishing between meningocele andinflammatory nasal polyp, nasal glioma and nasal dermoidcyst [3].

EE surgery has been increasingly employed during the lastdecade in the various forms of skull-base lesion. In compar-ison to frontal craniotomy, it improves patient comfort andreduces morbidity and hospital stay [1].

The ideal age for surgery in children is controver-sial. In the absence of symptoms requiring early surgery(meningitis, cerebrospinal rhinorrhea, facial deformity, orrespiratory or feeding difficulties), most authors recommendwaiting until the age of 2 or 3 years, when facial growth willfacilitate an endonasal procedure [1—5].

Others, however, argue that, while the risk of ascendingmeningitis associated with congenital skull base defect with-out active cerebrospinal rhinorrhea is slight, intact mucosaand dura do not represent an impermeable barrier to prop-agation of infection, making early surgery advisable [3,4].

Moreover, endoscopic sinus surgery does not compromisefacial development, so that early treatment is not contra-indicated on that score [6].

The youngest patients reported to have successfullyundergone EE surgery for nasal encephalocele were 23 [7],6 [5] and 5 months old [1]. The present patient was success-fully operated on at the age of only 2 months, making himthe youngest case of an endonasal approach so far reported.Taken together, these observations illustrate the interestof endoscopic approaches in congenital skull-base defectreconstruction, providing lower morbidity than frontal cran-iotomy.

Several reconstruction techniques have been reported:underlay and overlay, using cartilage, bone, mucosa, fat,fascia (temporalis or lata) or acellular skin graft (AlloDerm®)[8—10].

Castelnuovo recommended reconstruction using severallayers of vascularized flaps, such as nasoseptal or posteriorpedicle inferior turbinate flap [3].

El-Banhawy argued that large defects should be recon-structed using bone taken from the septum or mastoidcortex, applied in underlay, with a soft-tissue graft in over-lay [10].

Locating the skull-base defect may in some cases requiremiddle turbinectomy, ethmoidectomy or sphenoidotomy

[3,7].

Postoperative lumbar drainage is controversial [3].Follow-up should be prolonged, as growth continues,

modifying skull-base geometry [3].

[

ingocele 347

onclusion

hildhood nasal meningocele can be successfully treated byn EE approach, avoiding the complications associated withxternal approaches [3].

Constant progress in instrumentation, technique andkills will increase the feasibility of endonasal approachesn pediatric skull-base surgery [1,7].

isclosure of interest

he authors declare that they have no conflicts of interestoncerning this article.

eferences

[1] El-Banhawy OA, Halaka AN. Endoscopic endonasal exci-sion of congenital midline meningoencephalocele in a5-month infant. Int J Pediatr Otorhinolaryngol Extra 2009;4:66—71.

[2] Nogueira Jr JF, Stamm AC, Vellutini E, et al. Endo-scopic management of congenital meningo-encephalocelewith nasal flaps. Int J Pediatr Otorhinolaryngol 2009;73:133—7.

[3] Castelnuovo P, Bignami M, Pistochini A, et al. Endo-scopic endonasal management of encephaloceles in children:an eight-year experience. Int J Pediatr Otorhinolaryngol2009;73:1132—6.

[4] Woodworth BA, Schlosser RJ, Faust RA, et al. Evolutions in themanagement of congenital intranasal skull base defects. ArchOtolaryngol Head Neck Surg 2004;130:1283—8.

[5] Hamlin K, Kubba H. Endoscopic excision of a nasal meningoen-cephalocoele in an infant. Int J Pediatr Otorhinolaryngol Extra2007;2:92—4.

[6] Bothwell MR, Piccirillo JF, Lusk RP, et al. Long-term outcomeof facial growth after functional endoscopic sinus surgery.Otolaryngol Head Neck Surg 2002;126:628—34.

[7] Woodworth B, Schlosser RJ. Endoscopic repair of a congenitalintranasal encephalocele in a 23-month-old infant. Int J PediatrOtorhinolaryngol 2005;69:1007—9.

[8] Schlosser RJ, Bolger WE. Nasal cerebrospinal fluid leaks:critical review and surgical considerations. Laryngoscope2004;114:255—65.

[9] Lorenz RR, Dean RL, Hurley DB, et al. Endoscopic recon-struction of anterior and middle cranial fossa defectsusing acellular dermal allograft. Laryngoscope 2003;113:496—501.

10] El-Banhawy OA, Halaka AN, El-Dien AHS, et al. Subcranial trans-nasal repair of cerebrospinal fluid rhinorrhea by the combinedoverlay and underlay techniques. Minim Invasive Neurosurg J2004;47:1—6.