Tension pneumocephalus as a result of endonasal surgery: an uncommon intracranial complication

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1 Eur Arch Otorhinolaryngol (2014) 271: DOI /s RHINOLOGY Tension pneumocephalus as a result of endonasal surgery: an uncommon intracranial complication Gabriel Martínez-Capoccioni Ramón Serramito-García Eduardo Cabanas-Rodríguez Alfredo García-Allut Carlos Martín-Martín Received: 15 May 2013 / Accepted: 23 July 2013 / Published online: 3 August 2013 Ó Springer-Verlag Berlin Heidelberg 2013 Abstract Tension pneumocephalus (TP) is a clinical entity characterized by continued build-up of air within the cranial cavity, leading to abnormal pressure exerted upon the brain and subsequent neurologic deterioration, due to development of a mass effect and potentially a herniation syndrome. Intracranial complications of endoscopic sinus surgery (ESS) and other endonasal procedures are fortunately very rare, occurring in less than 3 % of cases. We report 4 cases of small bone defects (\3 mm) in the anterior cranial base accompanied by TP, caused by ESS and other endonasal procedures. The pathophysiology and management of this clinical entity is discussed with a pertinent literature. Four patients with small (\3 mm) skull base defects were identified. All patients presented with active cerebrospinal fluid leaks. CT scans showed All authors contributed equally to this work. G. Martínez-Capoccioni (&) E. Cabanas-Rodríguez C. Martín-Martín Servizo Galego de Saúde, Service of ENT Head and Neck Surgery, University Hospital Complex of Santiago de Compostela (CHUS), Santiago de Compostela, Spain Gabriel.adolfo.martinez.capoccioni@sergas.es; gabrielmartinez82@hotmail.com E. Cabanas-Rodríguez Eduardo.cabanas.rodriguez@sergas.es C. Martín-Martín cs.martin@usc.es R. Serramito-García A. García-Allut Servizo Galego de Saúde, Service of Neurosurgery, University Hospital Complex of Santiago de Compostela (CHUS), Santiago de Compostela, Spain ramon.serramito.garcia@sergas.es A. García-Allut Alfredo.Garcia.Allut@sergas.es intracranial tension pneumocephalus. Using image-guided endoscopic techniques, all defects were addressed with multi-layer repair. Closure was achieved in all patients on the first attempt, with an average follow-up of 36 months. Tension pneumocephalus is a rare event that can occur as a result of traumatic or iatrogenic violation of the dura and should be considered in all patients presenting with altered mental status after endoscopic sinus surgery or other surgical and diagnostic procedures that violate either the cranial or spinal dura. Because of the potential for rapid clinical deterioration and death, prompt brain imaging is warranted to rule out the diagnosis, and urgent neurosurgical consultation is indicated for definitive management. Keywords Tension pneumocephalus Pneumocephalus Endoscopic sinus surgery Endonasal procedures Cerebrospinal fluid leaks Introduction Pneumocephalus is defined as the presence of air in the cranial cavity, usually associated with cranial surgery, cranio-facial trauma (especially injuries involving basilar skull/sinus fractures or iatrogenic violation of the dura), nasopharyngeal tumor invasion, and meningitis [1 5]. Tension pneumocephalus (TP) is a clinical entity characterized by continued build-up of air within the cranial cavity, leading to abnormal pressure exerted upon the brain and subsequent neurologic deterioration, due to development of a mass effect and potentially a herniation syndrome. Knowledge of risk factors, radiographic findings, and clinical signs/ symptoms associated with TP is crucial to its prompt identification and treatment [6, 7]. Clinical diagnostic and treatment delays may result in poor neurologic outcome and mortality.

2 1044 Eur Arch Otorhinolaryngol (2014) 271: Intracranial complications of endoscopic sinus surgery (ESS) and other endonasal procedures are fortunately very rare, occurring in less than 3 % of cases [8]. The most common intracranial complication is cerebrospinal fluid (CSF) leak [3]. The presence of CSF rhinorrhea is potential risk factor for ascending meningitis and pneumocephalus, which may occur in up to one-third of patients [9]. We report 4 cases of small bone defects (\3 mm) in the anterior cranial base accompanied by TP, caused by ESS and other endonasal procedures, which were repaired successfully by endoscopic endonasal approach. The pathophysiology and management of this clinical entity are discussed with pertinent literature. Materials and methods This retrospective study was based on a review of the hospital clinical records of a consecutive series of patients who underwent ESS and other endonasal procedures between December 1999 and December 2012, presenting to the Endonasal Endoscopic Skull Base Unit (EESBU), University Hospital Complex of Santiago de Compostela (CHUS). Complete data that were sufficient for subsequent collection/analysis of the following variables: patient age; patient gender; mechanism of injury; primary injury associated with TP; signs and symptoms of TP; and therapeutic approach to TP. The diagnosis of tension pneumocephalus required all three of the following criteria: (1) clear radiographic changes on postoperative CT scan demonstrating intracranial air under pressure; (2) documented changes in neurologic function including decreased alertness, altered mental status, obtundation, or focal neurologic deficits; and (3) specific documentation of the diagnosis of tension pneumocephalus. The study was approved by the University Hospital Complex of Santiago de Compostela (CHUS) medical ethics board. Results Case 1 A 49-year-old man with a history of 2 previous sinus surgeries underwent revision ESS without apparent complication. During the post phase the patient was sneezing repeatedly. The day after surgery, the patient had a severe headache and neurological deterioration. The nasal packs were removed and profuse bleeding and fluid drainage were noted from the left side. Packing was replaced and a CT scan was obtained that revealed a small anterior ethmoid roof defect with massive left-sided TP (Fig. 1). The Fig. 1 CT scan showing a small anterior ethmoid roof defect with massive left-sided TP following day the patient underwent endoscopic repair. Intraoperative localization was achieved with image guidance. Endoscopic visualization revealed a small bone defect with exposed dura in the ethmoid roof. An active CSF leak was visualized. After debridement of bony fragments and removal of mucosa from around the defect, multi-layer closure was accomplished with a slice of cartilage and free middle turbinate mucosal graft above the smaller defects, fibrin glue to improve adherence of the graft, and supported in place with layers of Surgicel Ò,to separate the graft from the packing material and to prevent avulsion during its removal. Subsequent to sealing of the skull base defect, the patient recovered without further sequelae. No lumbar drainage of CSF was used. The patient continues to do well 7 years after surgery. Case 2 A 26-year-old man underwent primary ESS for indication of chronic sinus with massive polyposis disease unresponsive to medical therapy. Postoperatively, the patient noted progressively worsening headache and progressive neurological deterioration. The nasal packing was removed, and had left-side rhinorrhea. CT scan revealed pneumocephalus and a small left-sided ethmoid roof defect (Fig. 2). Immediately, the patient underwent image-guided endoscopic repair. Endoscopic visualization revealed an injury posterior to the left-side anterior ethmoid artery with active CSF drainage. Multi-layer closure was accomplished with a slice of septal cartilage, fibrin glue to improve adherence of the graft and supported in place with layers of Surgicel Ò. Subsequent to sealing of the skull base defect, the patient recovered without further sequelae. No lumbar

3 Eur Arch Otorhinolaryngol (2014) 271: Therefore, the patient underwent image-guided endoscopic repair of the defect and neurosurgery team performed an urgent cranial burr hole. Endoscopic visualization revealed a small skull base defect at the right-side ethmoid roof and pulsatile CSF drainage. The defect was repaired using a multi-layered technique including a slice of septal cartilage, fibrin glue and Surgicel Ò. Nasal packs were placed beneath to support the repair. The patient was kept at bed rest and on IV antibiotics for 3 days. No lumbar drainage was required. The patient has not experienced any further leak or other complications at 38-month follow-up. Certainly these patients are at risk for meningitis, so repair should be performed as soon as possible. Case 4 Fig. 2 CT scan demonstrating pneumocephalus and a small leftsided ethmoid roof defect drainage of CSF was used. The patient continues to do well 48 months after surgery. Case 3 A 55-year-old woman underwent primary ESS for symptoms of chronic sinusitis with polyposis. 7 days postoperatively, the patient reported severe headache and pressure behind her eyes. Because of these complaints, the patient was admitted and a CT scan was obtained. The CT scan revealed TP above the right ethmoid sinus and a small bone defect in the right cribriform plate (Fig. 3). Fig. 3 CT scan demonstrating TP above the right ethmoid sinus and a small bone defect in the right cribriform plate A 22-year-old woman underwent septoplasty with inferior turbinate radiofrequency. The application of radiofrequency electrode was incorrect performed by an inexpert surgeon. The electrode crossed the right inferior turbinate and perforated the back wall of posterior ethmoid sinus, penetrating the cranial cavity through the posterior ethmoidal sinus. Radiofrequency was applied intracranially, near the optic nerve. In the early postoperative period the patient referred right-blindness, but had no bleeding from the right nostril. Ophthalmologic examination demonstrated right amaurosis. CT scan revealed the trajectory of the electrode, bypassing the posterior wall of posterior ethmoid sinus and pneumocephalus (Fig. 4a c). The following day, the patient had severe headache and febrile episodes. Therefore, wide spectrum antibiotics thinking meningitis was initiated. Day 5 after the surgery, severe headache persisted and the patient had minor neurological deterioration. The patient underwent an urgent MRI, demonstrating an extensive pneumocephalus causing a mass effect and gross compression of the brain (Fig. 5a, b). Urgently, the patient underwent endonasal endoscopic repair. Exploration of the defects was performed according to our standard protocol for repairing skull base defects of our EESBU. The bone defect was identified on preoperative HRCT and an image-guided system was used intraoperatively for identification of the bone defect. The skull base penetration defect was recognized and the repair was undertaken by the otolaryngologist with neurosurgical assistance. The bone defect was completely exposed, and mucosa was removed around the nasal side of the defect. Smaller defects were repaired with free nasal septal perichondrium and cartilage grafts only. Fibrin glue was also used to secure the graft, along with multiple layers of absorbable packing. The patient was then discharged on the postoperative day 7 after a new uneventful hospital stay. Postoperative CT scan demonstrated resolution of the

4 1046 Eur Arch Otorhinolaryngol (2014) 271: Discussion Fig. 4 a Axial scan of computer tomography scan the brain demonstrating acute pneumocephalus after iatrogenic inferior turbinate radiofrequency treatment. b Coronal scan of computer tomography scan the brain demonstrating acute pneumocephalus after iatrogenic inferior turbinate radiofrequency treatment. c Sagittal scan of computer tomography scan the brain demonstrating acute pneumocephalus after iatrogenic inferior turbinate radiofrequency treatment preoperative pathologic findings. The patient gradually recovered over a period of 3 weeks. Two months later the patient returned to clinic doing well. Serial CT demonstrated resolving pneumocephalus. The patient has not experienced any further leak or other complications at 48-month follow-up. Reports of TP are sporadic in the literature. TP is a rare complication of ESS and other endonasal procedures [10]. It is important to note that violation of the cranial dura is the most common source of this problem [11 13]. There are two proposed mechanisms for air movement into the intracranial compartment and development of pneumocephalus: the inverted bottle and the ball-valve mechanisms [14 18]. The first mechanism involves a ball-valve effect exerted by intracranial tissue in the area of the dural defect, whereby air enters through the dural defect after increases in nasopharyngeal pressure, such as sneezing, coughing, and vomiting, force air through the cranial defect, but cannot escape through that area. The second mechanism, referred to as the inverted bottle mechanism, proposes that CSF leakage through the dural defect creates negative pressure in the intracranial space that allows air to move along a pressure gradient from the atmosphere to the cranial cavity [14 18]. This complication can occur rapidly and without warning, typically within 24 h of surgery or symptoms can occur within the first postoperative week, after enough air has reached the intracranial cavity to create pressure on the dura. Patients may present postoperatively with headache, clear rhinorrhea, meningismus, seizures, and altered mental status [17]. Immediate onset of symptoms may occur after a positive pressure event such as coughing, sneezing, vomiting, or straining. Altered mental status and headache are the most common presenting symptoms; the latter can often be severe and can occur with as little as 2 ml air in the cranial cavity [19]. TP can cause a wide variety of other symptoms, including visual disturbances, dizziness, confusion, and personality changes, but can also be present Fig. 5 a MRI (axial) demonstrating a frontal lobe tension pneumocephalus after iatrogenic inferior turbinate radiofrequency treatment. b MRI (Coronal) demonstrating a frontal lobe tension pneumocephalus after iatrogenic inferior turbinate radiofrequency treatment

5 Eur Arch Otorhinolaryngol (2014) 271: without symptoms [20]. In rare cases, patients can present with bruit hydro-aerique, a splashing sound heard by the patient on movement of the head caused by a succussion splash within the cranium [21, 22]. The most common presenting symptoms/signs in our patients were severe headache (4 patients), CSF rhinorrhea (4 patients) and neurological deterioration symptoms (4 patients). Blindness (1 patient) was reported less frequently. More insidious onset can occur without a positive pressure event. Significant headache or mental status change after ESS or other endonasal procedures should alert the physician of a serious problem. A CT scan should be performed immediately, and in most instances, a site of bony dehiscence and pneumocephalus may be identified [23]. Plain imaging can diagnose pneumocephalus, but CT scan is the diagnostic modality of choice, with an ability to detect as little as 0.5 cm 3 of air [19, 24]. Computed tomography (CT) was utilized in all patients, with MRI used as the primary neuroimaging modality in one case (case 4), although initially a CT scan was performed. The most common injuries to be directly associated with TP were at the ethmoid roof in all cases. Beyond the CT findings noted here, worsening headache, vomiting, focal neurologic deficits, and progressive obtundation are all signs concerning of tension physiology. Patients with TP should be placed in a supine position to help decrease additional air accumulation. Antiemetics should be given, if needed, to prevent vomiting. As bacterial infections have been reported as causes of pneumocephalus, broad-spectrum antibiotics should be considered if no other precipitating factor has been identified. Ascending meningitis has been clearly documented in the literature as a serious complication of unrepaired skull base defects in patients with CSF leaks. Bernal-Sprekelsen [9] reported that patients with traumatic CSF leak who were treated with conservative management alone had a 29 % risk of subsequent meningitis. In another series, Eljamel and Foy [25] reported an overall risk of meningitis of 30.6 % in their report of 160 patients with traumatic CSF leaks. They found a cumulative risk of meningitis of 85 % for 10 years, in addition to a 30 % risk of recurrent meningitis. Regardless of the size of the defect, conservative management was not effective because air continued to enter the intracranial cavity through the persistent skull base defect. Pneumocephalus may have spontaneous resolution with conservative management [26]. This is likely due to the gradual absorption of the intracranial air after the defect has temporarily sealed by blood clot or granulation. Since pneumocephalus is associated with a skull base defect, we believe that it should be treated in a similar fashion to CSF leaks resulting from intranasal surgery. Conservative management was not effective because air continued to enter into the intracranial cavity through the persistent skull base defect. Patients with TP required surgical closure of the bone defect and dural defect. If the dural defect does not resolve, air will continue to accumulate and the patient can progress to tension pneumocephalus, in which the steadily accumulating air collection exerts mass effect and possibly leads to cerebral herniation. TP is considered a surgical emergency [27]. Such findings should prompt urgent neurosurgical consultation and intervention. In some cases, emergency trephination should be performed to relieve the tension physiology until patients can undergo diagnostic workup and surgical repair of the dural defect. This is one of the few indications for urgent cranial burr hole or trephination. A less common but reported technique is to use a needle to aspirate air in patients who have a previous skull defect [27]. Case 3, it was necessary to perform emergency trephination due to the large volume of TP made in a short time. In each case, a multi-layer repair was performed, septal cartilage, with or without septal perichondrium and subsequently tissue glue. The strength of the cartilage was felt to be necessary for longer stability of the reconstruction site. Each of the repairs was supported with packing for 4 days to achieve stability during this initial period. Similar layered techniques have been previously described to repair skull base defects. [28 31]. In our case, we did not have the need for an emergency trepanation. Any penetration of dura requires watertight closure either primarily if possible or with suitable graft material as well as a flap for secondary reinforcement. Lumbar puncture should not be considered as a treatment modality for TP [10]. Similar to the risks associated with lumbar puncture in patients with elevated intracranial pressure due to a tumor or other sources of mass effect, lumbar puncture could provoke a brain herniation syndrome by creating a significant pressure gradient between the cranial and lumbar spaces and removing CSF that is otherwise exerting a buoying effect on the brainstem and other cranial structures. We do not routinely place a lumbar drain, relying instead on other techniques such as steroids, and mannitol to achieve brain relaxation without sacrificing adequate exposure of the anterior cranial fossa. The relatively large size of these defects did not predict a poor outcome, as all were successfully repaired on the first attempt. With an average follow-up of 27 months, none of the patients had recurrence of CSF rhinorrhea. Patients were observed with routine endoscopy in the office. There has been no endoscopic or CT evidence of encephalocele development in these patients to date. In addition, despite the intracranial injuries, there have been no permanent neurologic deficits following these injuries and repairs.

6 1048 Eur Arch Otorhinolaryngol (2014) 271: The paper describes a unique severe complication after inferior turbinate radiofrequency treatment. Complications of this technique may be related to several other factors. These include the learning curve associated with the use of this technology and especially understanding of the electrophysiology of lesion generation for the various devices and treatment sites, the number and location of lesions created during each treatment session. Unfortunately, the application of inferior turbinate radiofrequency technology appears easy because the electrode is placed deep to the mucosa where there is essentially no visual evidence of the treatment effect below the surface without visual control. However, it is critical to have a basic understanding of the electrophysiologic principles of delivered radiofrequency energy to limit undesired tissue damage and accomplish a positive treatment effect. Conclusion Tension pneumocephalus is a rare disease that can occur as a result of traumatic or iatrogenic violation of the dura and should be considered in all patients presenting with altered mental status after endoscopic sinus surgery or other surgical and diagnostic procedures with potential to violate either the cranial or spinal dura. Because of the potential for rapid clinical deterioration and death, prompt brain imaging is warranted to rule out the diagnosis, and urgent neurosurgical consultation is indicated for definitive management. All endonasal procedures should be controlled under direct vision with endoscopes. Acknowledgments We would like to thank the Service of ENT Head and Neck Surgery and Service of Neurosurgery, University Hospital Complex of Santiago de Compostela. The authors declare that they have no com- Conflict of interest peting interests. References 1. 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