Endoscopic repair of cerebrospinal fluid leak in paediatric patients

International Journal of Pediatric Otorhinolaryngology 78 (2014) 1898–1902

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Endoscopic repair of cerebrospinal fluid leak in paediatric patients E. Emanuelli *, P. Bossolesi 1, D. Borsetto, E. D’Avella 2 ENT and Otosurgery Unit, Department of Neurosciences, University Hospital of Padua, Padua, Italy

A R T I C L E I N F O

A B S T R A C T

Article history: Received 20 April 2014 Received in revised form 14 August 2014 Accepted 15 August 2014 Available online 26 August 2014

Background: In scientific literature exist fewer case series regarding endoscopic treatment of paediatric cerebrospinal fluid fistulas. Compared to craniotomy endoscopic repair does not reach wider consent even if craniotomy carries higher complications rate. Objective: The aim of the present study was to report our institutional experience on paediatric cerebrospinal fluid leak to demonstrate the safety and efficacy of the endoscopic approach in a variety of cases. Methods: Clinical records of all paediatric patients who underwent endoscopic repair of anterior and middle fossa skull base defects are reviewed for several parameters. Results: 10 patients were enrolled, 6 males and 4 females with a mean age of 10 years. The aetiology of the leak was congenital in 2 cases, trauma-induced in 6 cases and iatrogenic in 2 cases. In all the cases the defect was localized by computed tomography. Remedial was obtained using multiple grafts technique with autologous materials during a single procedure in all but one case, this one case requiring a second operation for delayed recurrence. The mean follow-up duration is 36 months. Conclusion: CSF fistulas can be approached in paediatrics of any age with high success and low complications rate using the endonasal technique. ß 2014 Elsevier Ireland Ltd. All rights reserved.

Keywords: Paediatric cerebrospinal fluid leak Rhinorrhea Endoscopic repair Meningoencephalocele Endonasal approach Paediatric neurosurgery

1. Introduction Endonasal approach is the primary surgical strategy to repair cerebrospinal fluid leaks (CSFL) of the anterior skull base in adult patients [1–6]. Nevertheless the anatomical and physiological peculiarities of paediatric population, such as the narrow nasal passage and the different degree of sinus pneumatization, can be challenging [7–9]. The advantage of this approach depends on the limit of conventional intra- and extra-cranial skull base surgery. The extreme fragility of intradural CNS structures with reference to the cranial nerves and the pituitary stalk in infants is a critical factor with great risk of permanent panhypopytuitarism and other life-threatening complications. Far less important is the potential disruption of the growth centres in the craniofacial skeleton that may result in cranial asymmetries and impairment of nose and sinuses normal function [10–13]. The literature

* Corresponding author at: Dipartimento di Neuroscienze, Otochirurgia Universita’ degli Studi di Padova, via Giustiniani 2, 35128 Padova, Italy. Tel.: +39 049 8211993; fax: +39 0498211994; mobile: +39 347 0464904. E-mail address: [email protected] (E. Emanuelli). 1 ENT Department, Hospital St. Valentine, Montebelluna, Italy. 2 Department of Neurosurgery, University Hospital of Padua, Padua, Italy. http://dx.doi.org/10.1016/j.ijporl.2014.08.020 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.

displays separating views about several topics and many observations and considerations regarding endoscopic management of CSFL in paediatric population remain to be discussed [14]. We report our experience of endoscopic approach in paediatric patients for the treatment of anterior and middle fossa skull base defects. 2. Materials and methods (1) Case demographics: From October 2001 to November 2012, 10 paediatric patients with anterior or middle fossa basal defects were operated via transnasal endoscopic approach in our institution. Patient’s age ranged from 2 months to seventeen years (mean 10.5 years). The study included 6 males and 4 females. (2) Clinical features: In 2 cases the leak was associated with developmental anomalies of the CNS: one case was a 2 month old infant presenting with a meningoencephalocele of the planum sphenoidale (Fig. 1) and one case was a 15 year old female with Arnold–Chiari II Syndrome and persistence of the craniopharyngeal canal ‘‘canalis craniopharyngeus medius’’ (Fig. 2). 6 cases were post-traumatic patients: 5 as result of road traffic accident and one of gunshot wound to the head. 2

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Fig. 1. Infant with meningoencephalocele of the planum sphenoidale: (A) Preoperative MRI, the white arrow points the meningeal herniation into the nasopharynx. (B) Postoperative MRI fourteen months after the first operation shows successful repair of the defect.

Fig. 2. Patient with Arnold–Chiari II Syndrome and persistence of the craniopharyngeal medial canal: (A) Preoperative MRI, the red arrow indicates the orifice of the craniopharyngeal canal bordering the nasopharynx. (B) Intraoperative detail of the skeletonized bony canal, the white arrows show as it crosses the sphenoid sinus. (C) The canal has been sealed with bone fragments, fascia and fibrin glue. (D) MRI one year after the operation showing complete recovery. (For interpretation of the references to color in figure legend, the reader is referred to the web version of the article.)

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cases were iatrogenic following surgery for craniosynostosis (Table 1). (3) Preoperative work-up: All patients underwent physical examination throughout 2.7 mm Ø  18 cm rigid endoscope with 308 lens, CT scan of the brain and sinuses with a 2 mm slice bony window. MR images were obtained where a congenital malformation was suspected. In case other means failed to localize the leakage site, the fluorescein dye test should have been reserved to patients older than 15 years of age in order to minimize the risk of neurological complications [15,16], anyway in this cohort of patients there was no need for intrathecal fluorescein administration. (4) Operation: Surgery was performed under general anaesthesia, using 4 mm 1  18 cm rigid endoscopes, with 08 and 458 lenses always connected to a high definition monitor. Patients were kept supine in an anti-Trendelenburg position with the head slightly extended. In this way the plane of the ethmoidal roof and planum sphenoidale remained perpendicular to the longitudinal axis of the telescope offering the best field visualization. Mucosal shrinking with less bleeding was induced by cotton pledges soaked in xylometazoline, mepivacaine with or without 1:100,000 epinephrine solution. A purely endoscopic approach was performed in all patients. The homolateral middle turbinate to the side of the fistula was removed to obtain a better exposition of the defect’s region and to harvest autologous repair material. Ethmoidectomy and sphenoidectomy were performed for better exposure of the ethmoidal roof and planum. Once the skull base defect was visualized the mucosa around the defect’s area was removed with the aim of defining precisely the dimension of the bony leak and to improve the engraftment of the repairing material. Ordinarily the site and shape of the defect influence the type of graft to be used: if the bony defect is small and located in the olfactory groove, smoothing of surrounding bone, coagulation of underling meningeal line and covering with overlay mucoperoisteal flap can be enough to ensure healing. If the lesion involves the planum sphenoidale or sphenoid posterior wall, the bony edge of the defect has to be cleared out and dura elevated few millimetres around to introduce a multilayer graft [9]. The grafts made up of bone or conchal cartilage, were positioned with an underlay technique between the dura and the bony skull base; fascia lata underlay and overlay, mucoperiosteum just overlay. Skull base defects localized in the posterior wall of the sphenoid sinus were repaired with the

‘‘gasket seal’’ technique [17–19]. At the completion of the operation grafting was insured with haemostatic oxidized cellulose (fibrillary Tabotamp1) and nasal fossa packed with sponges medicated with gentamycin ointment. In patients younger than ten years nasal packing was done with oxidized cellulose to avoid troublesome removal. (5) Postoperative care: In all patients packing was removed on day 2 or 3 after surgery. Patients were required to rest in bed for 3–4 days. All patients received intravenous antibiotic prophylaxis before surgery and oral antibiotics for at least one week. (6) Follow-up: Clinical examination with nasal endoscopy was performed at almost monthly intervals for the first 6 months, every 3 months over the following 12 months, then once a year. MRI scan was scheduled twelve months after surgery to monitor the development of connective tissue and later on just in case of unclear sinus view at endoscopy. The follow-up duration ranged from 14 months to eleven and a half years, with a mean follow-up, skipping the extreme values, of 36 months (Table 2). The patients’ clinical charts were retrospectively analyzed for several parameters: aetiology of the defect, presence of active CSF leak, meningitis or other revealing symptoms, location of the defect, type of surgical approach, grafting materials, complications and long-term success of the repair. 3. Results The use of nasal endoscopy enabled direct visualization of the source of leakage in all but one case. In the case of meningoencephalocele a pulsating mass appeared projecting into the nasopharynx. In the case with persistent craniopharyngeal canal no CSFL was evident under direct view, nor active leakage was detected by beta2 transferrin test. A fluorescein test was not performed because of positive diagnostic imaging and the enhanced risk of epilepsy attack. The history of repeated meningitis in presence of pneumoencephalo was considered relevant enough to plan surgery. In all the cases CT scans ascertained the site of the leak that was subsequently confirmed during surgery. MRI was used to understand the amount of ectopic brain and pituitary tissue to preserve in the 2 cases of congenital malformation. Removal of the mucoperiosteal layer around the defect allowed a clear understanding of the site and shape of the leak. Repair was

Table 1 Details on symptoms, clinical features, associated conditions and diagnosis. Age/sex

Symptoms

Clinical features

Associated conditions

Diagnosis

15 yr/F 10 yr/M 2 mo/M

Acute rhinorrhea Acute rhinorrhea Nasal blockage, snoring, recurrent signs of meningitis like neck stiffness, crying and temperature over 398. CSF culture: negative

Craniofacial injuries – Hypertelorism, meningoencephalocele.

CT CT Endoscopy/ MR/CT

4 yr/F 15 yr/F

Acute rhinorrhea Recurrent meningitis (Pneumococcus +), pneumoencephalo into the craniopharyngeal canal (computed tomography)

Head injury/motor vehicle crash Head injury/motor vehicle vs pedestrian endoscopy revealed a polyp-like mass occupying the rhinopharynx and partially obstructing choanal apertures, MR scan ascertained the cleft Rhinorrhea following Cranioplasty MR displayed anomalies of CNS including the craniopharyngeal canal, CT scan showed pneumoencephalous

CT MR/CT

17 yr/M

Meningitis (delayed 1 mo later)

Head injury/motor vehicle crash

17 yr/F 9 mo/M

Acute rhinorrhea Acute rhinorrhea

Head injury/motor vehicle crash Rhinorrhea following Cranioplasty

11 yr/M

Acute rhinorrhea following Gunshot wound to the head

16 yr/M

Acute rhinorrhea

CT showed the missile entered the pterion to be found into the sphenoid sinus; neither fatal nor optic nerve injury amazingly occurred Head injury/motor vehicle crash

Craniosynostosis Arnold–Chiari II Sd; panhypopituitaris,mental retardation, epilepsy following meningitis Extradural haemorrhage, craniofacial injuries – Craniosynostosis, Saethre Chotzen Sd Subaracnoid haemorrhage

–

CT

CT CT CT CT

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Table 2 Details on aetiology, site of leakage, type of graft repair and follow-up. Age/sex

Aetiology

Site of leakage

Type of grafts for repair

Outcome/follow-up

15 yr/F 10 yr/M

Traumatic Traumatic

Planum sphenoidale Posterior sphenoidal wall

Bone fragment replaced, fascia lata Bone fragment replaced, fascia lata

Good/24 mo Good/25 mo

2 mo/M

Malformative: meningoencephalocele

Planum sphenoidale

First attempt: nasal septum bone, temporalis fascia Second attempt: conchal cartilage, temporalis fascia

Recurrence 12 mo later Good/14 mo

4 yr/F 15 yr/F

Iatrogenic Malformative: persistence of the craniopharyngeal canal Traumatic Traumatic Iatrogenic Traumatic Traumatic

Ethmoidal roof Pharyngeal orifice of the craniopharyngeal canal

Fascia lata, middle turbinate bone and mucoperiostium Fascia lata, middle turbinate bone and mucoperiostium

Good/32 mo Good/36 mo

Ethmoidal roof Ethmoidal roof Ethmoidal roof Posterior sphenoidal wall Ethmoidal roof

Fascia lata, middle turbinate bone and mucoperiostium Fascia lata, middle turbinate bone and mucoperiostium Middle turbinate bone and mucoperiostium Fascia lata, middle turbinate bone and mucoperiostium Fascia lata, middle turbinate bone and mucoperiostium

Good/38 mo Good/38 mo Good/39 mo Good/46 mo Good/11 yr/6 mo

17 yr/M 17 yr/F 9 mo/M 11 yr/M 16 yr/M

achieved with various free grafts: mucoperiosteum and bone from middle turbinate in 9 patients, fascia lata in 8 patients and temporalis muscle fascia with conchal cartilage in one patient (Table 2). Neither of the cases had any major complication relating to the procedure. We did not use allogenic material for grafting and we did not have to position a lumbar drain or shunts to deal with postoperative persistent CSFL. The sole patient who presented a delayed failure with rhinorrhea after twelve months was the infant with meningoencephalocele; this patient was re-operated with success using the same technique. The first time grafting included temporalis fascia and vomerian bone; the second time temporalis fascia and conchal cartilage harvested from the opposite side. The success rate was 90% after the first closure attempt. None of the patients experienced further episodes of meningitis or rhinorrhea. No patient required a craniotomy to close the defect. Postoperative MRI scan showed in all cases a complete repair. No disturbance on facial growth different from the preoperative condition was observed with a follow-up ranging from 14 months to eleven and a half years, fibreoptic endoscopy always confirmed good healing and normal nasal breathing. 4. Discussion The spontaneous CSFL in children presents peculiarity compared to adult population because of its association with developmental congenital syndromic or non-syndromic anomalies. An inconspicuous CSFL may be misdiagnosed and even if the leak is active it is not easy to collect a sample for B2-trasferrin or Btrace test. Infants with a basal mass are more likely to apply for nasal related symptoms so office 2.9 mm Ø flexible endoscopy is cost-effective and useful to rule out suspect of upper airway malformation. Basal encephaloceles protrude through a defect in the cribriform plate or body of the sphenoid and appear as intranasal masses. We can broadly identify two main categories the anterior (or ethmoidal) and the transphenoidal type. The first one is the most common and appears as a translucent sac along the cribriform plate adjacent to the middle turbinate vertical attachment, usually its content does not involve vital structures [20]. The transphenoidal encephalocele is a midline mass that may associate with hypertelorism and broadening of the bony nasal vault, it extends inferiorly from a defect in the floor of the sella turcica and may include the pituitary gland or the hypothalamus. Different cases of these two categories are described in the literature [7–9,21–24]. Defects in the skull base without meningeal herniations or clefts may accompany congenital malformations; we report a case of persistence of the craniopharyngeal canal

associated with Arnold–Chiari II Syndrome. Localization of the CSF leak is important, the use of thin slice coronal CT provides fine details helpful in both confirming the diagnosis and defining the optimum approach [20]. MRI is necessary to display the soft tissue anatomy in particular the functional brain or pituitary gland displaced downward [21]. The dimension of the cleft and the consistency of CNS structures involved are critical in surgical decision-making. Endoscopic approach is indicated where the defect involves the cribriform plate and each time an aperture is evident through the bony vault in presence of a relatively small amount of non functional herniated tissue, on the other hand great malformations with displacement of brain structures are the most challenging cases. Attention has to be paid not to damage ectopic CNS vital structures like hypophysis, pituitary stalk, infundibulum. At the moment the surgical treatment of CSFL associated with congenital malformations is still neither simple nor straightforward. Nevertheless between the congenital anomalies, anterior and middle cranial fossa meningoencephaloceles are supposed to be amenable for endonasal repair each time a graft large enough to close the defect can be placed through the nose. At least this route should always be attempted within a setting able to switch to an open procedure if necessary. Uneven information can be found in the literature regarding the endoscopic treatment of skull base defects in children, general principles that rule endoscopic surgery to adults cannot be directly extrapolated to treat children so this approach remains to be evaluated [3,7–9,14,22–25]. The small size of the nasal cavity obstacles the movement of the endoscope and surgical instruments and could make it difficult to expose adequately the area of the skull base defect. In young children when nares appeared to offer not enough room to place the endoscope and instruments, a sub labial incision has been advocated to enter the nasal cavity [25]. The incomplete pneumatization of the nasal sinuses, in particular the sphenoid sinus may disorientate the surgeon. For this reason paediatric transnasal endoscopic surgery has been considered challenging pre 2–3 year old, however we did not experienced the severe complications that accompany craniotomy such as intracranial haemorrhages, hypopytuitarism, epilepsy or brain oedema. In our study, we report on the closure of a CSF leak in a 2 month old. The available literature indicates that he is the second youngest patient treated with this technique following a one and a half month old infant [26]. Theoretically children nasal growth could be compromised with removal of bone or cartilage during transnasal surgery but compared to other alternatives, it appears far less risky and disruptive [27,28] in our chart we have not got any record of nose and sinus dysfunction or cosmetic disorder. Most of the traumatic

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CSF-leaks close spontaneously within 7–10 days, but they may subside over the following weeks or months and a 10% risk of developing meningitis per year exists [29]. This is confirmed by one of our cases admitted with meningitis one month after trauma. To avoid the chance of ascending meningitis we performed surgery as soon as the diagnosis was ascertained. With regard to the use of autologus grafts we did not find a difference between adult and paediatric patients with the exception that donor sites like iliotibial tract in infant are immature, therefore the 2 month old infant was treated the first time with temporalis fascia plus bone from nasal septum and the second time with conchal cartilage and temporalis fascia of the other side. For the other cases we mainly used ethmoid mucoperiosteum and bone or fascia lata because the healing process is rapidly induced by fascia [30,31]. The debate regarding the usefulness of lumbar drainage to facilitate the healing of skull base defect is still open [32]. In our study lumbar drainage was never inserted during and/nor after surgery. 5. Conclusions Although the number of patients in our study is small, the results indicate that endoscopic approach is safe and can be effective in paediatric patients of any age. It should be considered the treatment of choice, as endoscopic approach decreases the chance of complications associated with intra- or extra-cranial operation and permits a rapid recovery. The accurate location of the leak is mandatory before surgery and a thin slice computed tomography seems to be the best method. Meningoencephaloceles and skull base congenital malformations are the most challenging cases, MR study is recommended to detect functional ectopic pituitary gland. Even though grafting material can be various, we consider fascia the best material to ensure success in larger fistulas with correct placement of the graft closely in contact with the freshened defect. We recommend this technique to surgeons thoroughly experienced in nasal endoscopy prepared to switch to an open procedure if necessary. References [1] V.J. Lund, Endoscopic management of cerebrospinal fluid leaks, Am. J. Rhinol. 16 (1) (2002) 17–23. [2] H. Stammberger, Surgical occlusion of cerebrospinal fistulas of anterior skull base using intrathecal sodium fluorescein, Laryngorhinootologie 76 (1997) 595–607. [3] D.E. Mattox, D.W. Kennedy, Endoscopic management of cerebrospinal fluid leaks and cephaloceles, Laryngoscope 100 (1990) 857–862. [4] B. Schick, R. Ibing, D. Brors, W. Draf, Long-term study in endonasal duraplasty and review of the literature, Ann. Otol. Rhinol. Laryngol. 110 (2001) 142–147. [5] E.E. Dodson, C.W. Gross, J.L. Swerdloff, L.M. Gustafson, Transnasal endoscopic repair of cerebrospinal fluid rhinorrhea and skull base defects: a review of twentynine cases, Otolaryngol. Head Neck Surg. 111 (1994) 600–605. [6] D.C. Lanza, D.A. O’Brien, D.W. Kennedy, Endoscopic repair of cerebrospinal fluid fistulae and encephaloceles, Laryngoscope 106 (1996) 1119–1125. [7] R.F. Wetmore, A.C. Duhaime, R.D. Klausner, Endoscopic repair of traumatic CSF rhinorrhea in a pediatric patient, Int. J. Pediatr. Otorhinolaryngol. 36 (July (2)) (1996) 109–115.

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