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5. Hedner T, Samuelsson O, Lunde H, et al: Angio-oedema inrelation to treatment with angiotensin converting enzyme in-hibitors. BMJ 304:941, 1992

6. Sadeghi N, Panje WR: Life-threatening perioperative angio-edema related to angiotensin-converting enzyme inhibitor ther-apy. J Otolaryngol 28:354, 1999

7. Kyrmizakis DE, Papadakis CE, Fountoulakis EJ, et al: Tongueangioedema after long-term use of ACE inhibitors. Am J Oto-laryngol 19:394, 1998

8. Mchaourab A, Sarantopoulos C, Stowe DF: Airway obstructiondue to late-onset angioneurotic edema from angiotensin-con-

verting enzyme inhibition. Can J Anaesth 46:975, 1999

Scott Tate, DDS, MS,* Richard A. Rak, MD,

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9. Guo X, Dick L: Late onset angiotensin-converting enzyme in-duced angioedema: Case report and review of the literature. JOkla State Med Assoc 92:71, 1999

0. Carr AA, Prisant LM: Losartan: First of a new class of angio-tensin antagonists for the management of hypertension.J Clin Pharmacol 36:3, 1996

1. Chiu AG, Krowiak EJ, Deeb ZE: Angioedema associated withangiotensin II receptor antagonists: Challenging our knowledge ofangioedema and its etiology. Laryngoscope 111:1729, 2001

2. Abdi R, Dong VM, Lee CJ, et al: Angiotensin II receptorblocker-associated angioedema: On the heels of ACE inhib-

itor angioedema. Pharmacotherapy 22:1173, 2002

Oral Maxillofac Surg3:556–559, 2005

Unusual Presentation of a CervicalPseudomeningocele: A Case Report

and Review of the Literature

† and Jonathan S. Bailey, DMD, MD‡

ral and maxillofacial surgeons are commonlyalled on to manage patients with cerebrospinaluid (CSF) leaks, which present as otorrhea orhinorrhea, secondary to craniomaxillofacial trau-a.1,2 We present an unusual case of a CSF leakresenting as an anterior cervical pseudomeningo-ele. Review of the literature revealed only 1 pre-ious case report of an anterior cervical pseudome-ingocele.3 Eight additional cases with leaksresenting in the retropharyngeal space, posteriorervical subgaleal space, and spinal canal weredentified. Previously reported treatment optionsor cervical pseudomeningocele include surgicalxcision or repair, lumboperitoneal or ventriculo-eritoneal shunting, and lumbar drainage. Identifi-ation and treatment of this problem are imperativeo prevent associated neurologic decline.

eceived from the Carle Foundation Hospital and Clinic Associa-

ion, Urbana, IL.

*Formerly, Chief Resident, Division of Oral and Maxillofacial

urgery.

†Assistant Clinical Professor, Division of Neurosurgery.

‡Assistant Clinical Professor, Division of Oral and Maxillofacial

urgery.

Address correspondence and reprint requests to Dr Bailey: De-

artment of Oral and Maxillofacial Surgery, Carle Foundation Hos-

ital and Clinic Association, 602 W University Ave, Urbana, IL

1801; e-mail: jonathan.bailey@carle.com

2005 American Association of Oral and Maxillofacial Surgeons

278-2391/05/6304-0021$30.00/0

eport of a Case

The patient was a 26-year-old morbidly obese womanho was the unrestrained driver involved in a motor vehi-

le accident on April 3, 2002. She had been stabilized at anutside hospital and was transferred to Carle Foundationospital (CFH) for further evaluation and treatment. Her

njuries included C6-7 fracture dislocation with quadriplegiand bilateral pulmonary contusions. Her past medical his-ory was significant for morbid obesity and asthma. Theatient was admitted to the intensive care unit and initiallyaintained on biphasic positive airway pressure but hyper-

arbia and hypoxia developed. She was intubated for respi-atory failure. On April 5, 2002, she underwent C6-7 ante-ior cervical discectomy and anterior arthrodesis with iliacrest allograft. On exposure of the anterior vertebral col-mn, a CSF collection and leak was noted.Postoperatively, the patient was weaned from the venti-

ator and extubated on April 15, 2002. The patient subse-uently became bradycardic and progressed to asystole. Sheas emergently reintubated. The resuscitative efforts wereifficult secondary to her morbid obesity and the presencef the cervical collar. After stabilization, a firm swelling wasoted in the right neck underlying the cervical fusion site.he neurosurgery service was notified, and the clinicaliagnosis of a postoperative hematoma was made.The Division of Oral and Maxillofacial Surgery was con-

ulted for tracheostomy due to respiratory failure. On phys-cal examination, the patient’s neck was short and obese.he previous surgical site was firm to palpation but hemo-tatic. In consultation with the neurosurgery service, theecision was made to proceed with tracheostomy and evac-ation of the hematoma.In the operating room, a horizontal suprasternal incisionas marked out 2 fingerbreadths above the sternal notch,

nferior to the previous cervical fusion incision. The inci-ion was made through the skin and subcutaneous tissue. Ashe platysma was incised, a significant leak of clear fluid,

resumably CSF, was expressed through the wound. The

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ound was packed. The neurosurgeon was notified andnspected the wound. The decision was made to close the

ound and plan to observe for reaccumulation and possibleecompression if indicated.Magnetic resonance imaging shows a 4.5 � 3.0 � 3.0-cm

uid collection, consistent with a pseudomeningocele, lo-ated anterior to the lower cervical spine C6-7 region (Figs-3). Despite several attempts at percutaneous aspiration,erformed under sterile conditions, the CSF reaccumulated.n attempt to place a lumbar drain was unsuccessful due toifficulty positioning the patient secondary to her morbidbesity, cervical spine injury, and the presence of the oralndotracheal tube. A second attempt at lumbar punctureas performed but was again unsuccessful. On the third

ttempt, with fluoroscopic guidance, a lumbar drain waslaced. In addition, 20 mL of CSF accumulation was aspi-ated from her anterior cervical pseudomeningocele. Therain output was 25 mL on day 1, 12 mL on day 2, and 5 mLn day 3. The lumbar drain was removed 3 days afterlacement. Five days after lumbar drain placement, theatient underwent tracheostomy by the oral and maxillofa-ial surgery service without complication. One week afterhe tracheostomy, the tracheostomy tube was changed to aenestrated style and no CSF leakage was noted. Repeatomputed tomography scan of the cervical spine showedesolution of the pseudomeningocele. During the next 3eeks, the tracheostomy tube was downsized and the pa-

ient was transferred to an out-of-house rehabilitation facil-ty for the remainder of her care and recovery.

iscussion

Oral and maxillofacial surgeons are commonly

IGURE 1. Sagittal magnetic resonance image of the cervical spinefter cervical spine fusion. Numbers indicate cervical spine vertebrae.arge white arrow shows pseudomeningocele, with small white arrowndicating upper aerodigestive tract. Spinal cord edema is also notedosterior to traumatized segments.

ate, Rak, and Bailey. Unusual Cervical Pseudomeningocele Pre-entation. J Oral Maxillofac Surg 2005.

alled on to manage patients with CSF otorrhea orTs

hinorrhea as a result of craniomaxillofacial trauma.1,2

SF otorrhea and rhinorrhea are most commonlyreated with a multidisciplinary approach. Treatmentptions include observation and conservative mea-ures, lumbar drain placement, and reduction of crani-maxillofacial fractures including extracranial or in-racranial dural repair when indicated.1-9

We present an unusual case of a CSF leak present-ng as an anterior cervical pseudomeningocele. Re-iew of the literature revealed only 1 previous caseeport of an anterior cervical pseudomeningocele.3

ight additional cases presenting in the retropharyn-eal space, posterior cervical subgaleal space, andpinal canal were identified4,10-16 (Table 1). All previ-us cases were reported in the otolaryngology, neu-osurgery, or anesthesia literature.

As in the more typical presentation of a CSF leak,evelopment of cervical pseudomeningocele resultsrom a disruption in the integrity of the dura allowingSF to leak into low-pressure regions. The most com-on etiology is atlanto-occipital dislocation second-

ry to trauma.10,12,13,15 This injury results in a tear ofhe dura at the craniocervical junction.15,16 Thesenjuries have been described after motor vehicle ac-idents and may occur in combination with a closedead injury, hydrocephalus, and elevated intracranialressure.10,13,15 The presence of hydrocephalus andlevated intracranial pressure may lead to diversion ofSF through the traumatic defect.15

IGURE 2. Sagittal magnetic resonance image of cervical spine afterervical spine fusion. Large white arrow shows pseudomeningoceleommunicating with spinal canal. Small white arrow indicates thepper aerodigestive tract.

ate, Rak, and Bailey. Unusual Cervical Pseudomeningocele Pre-entation. J Oral Maxillofac Surg 2005.

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558 UNUSUAL CERVICAL PSEUDOMENINGOCELE PRESENTATION

Cervical pseudomeningocele has also been re-orted after cervical spine surgery.3,4,14 Nair et al3

eported the case of a 51-year-old woman who pre-ented 2 months after anterior cervical fusion per-ormed for cervical myelopathy and radiculopathy. Ater initial surgery, a small tear in the dura was iden-ified and managed with bovine pericardium andcrylic glue. One day after discharge, the patient re-resented with pain and swelling at the operative sitend dysphagia. A clinical diagnosis of pseudomenin-ocele was made and managed with a lumbar drain.he swelling promptly resolved and the drain wasemoved. Two months later, she again presented with

IGURE 3. Axial magnetic resonance image of cervical spine afterervical spine fusion. Largest arrow indicates pseudomeningoceleommunicating with spinal canal. Small thin arrow indicates airway,nd short white arrow points to the right lung apex. “R” indicatesatient’s right side.

ate, Rak, and Bailey. Unusual Cervical Pseudomeningocele Pre-entation. J Oral Maxillofac Surg 2005.

Table 1. PREVIOUS CERVICAL PSEUDOMENINGOCELE C

Author (year)Age (yr)/Gender Site

ollalto et al (1986)10 3/Male Spine C1-T3klar et al (1990)11 17/Male Spine C6-T4ouw (1991)12 40/Female Postcervicalilliams et al (1995)13 3/Male Retropharyngealosono et al (1995)14 45/Male Cervical (postaracaso et al (1997)15 26/Male Retropharyngeal

11/Male Retropharyngealair et al (1998)3 51/Female Anterior cervicaloel et al (2001)16 8/Male Occipitocervical

Abbreviations: AOD, atlanto-occipital dislocation; AOI, atlanto-oc

ate, Rak, and Bailey. Unusual Cervical Pseudomeningocele Presentati

ntermittent swelling in the anterior neck and dyspha-ia. A computed tomography scan showed a 3.2 �.4-cm right prevertebral and paralaryngeal fluid den-ity mass. At surgical exploration, an empty scarredseudomeningocele cavity was entered. The regionas reinforced with fibrin sealant and a drain waslaced. Her recovery was uneventful.Our case of an anterior cervical pseudomeningo-

ele is the result of her C6-7 fracture dislocation. Athe time of her anterior cervical fusion, the neurosur-eon noted CSF located anterior to the cervical spinalolumn with an active leak arising from the disc spacef C6-7. The CSF leak was identified on exposure ofhe spinal column before instrumentation and re-oval of the cervical disc. Primary repair of the dural

isruption was not possible. The dural tear was mostikely the result of the shearing forces created by theervical spine fracture dislocation. This mechanism isimilar to that which occurs with atlanto-occipitalislocation.Previously reported treatment options for cervical

seudomeningocele include surgical excision or re-air, lumboperitoneal or ventriculoperitoneal shunt-

ng, and lumbar drainage.3,4,10-16 In one of the firsteports of surgical repair of cervical pseudomeningo-ele, Louw et al12 identified the lesion incidentallyhile performing an atlanto-occipital arthrodesis. Inosono et al’s14 case report, the clinical situation

equired surgical exploration and repair due to theresence of spinal cord herniation into the pseudo-eningocele cavity. Alternatively, Goel et al16 cite the

elative ease of lumboperitoneal shunt surgery as aeason to avoid direct surgical repair of the duralisruption. However, the use of a lumboperitoneal orentriculoperitoneal catheter does require placementf a foreign body and repeated surgery for shuntevision.17

The use of a lumbar drain to prevent or treat CSFstula formation has been reported by Shapiro andcully.4 Their series included 107 patients undergoing

EPORTS

Etiology Treatment

Trauma, AOD VP shuntTrauma Surgical repairAOI Surgical repairTrauma, AOD VP shuntTumor extirpation Surgical repairTrauma, AOD VP shuntTrauma, AOD No treatment, patient diedAnterior cervical fusion Surgical repairTrauma LP shunt

instability; VP, ventriculoperitoneal; LP, lumboperitoneal.

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TATE, RAK, AND BAILEY 559

ranial surgery or spinal surgery or who experiencedraumatic CSF rhinorrhea or otorrhea. One hundredne patients (94%) were successfully managed withhis technique. Specifically, the authors make refer-nce to 4 patients with cervical pseudomeningoceleshat were treated successfully in this manner. Com-lications included transient nerve root irritation14%), overdrainage (5%), and spinal headache (1%).

The surgeon who treats patients with cranial andpinal injuries should be familiar with this unusuallinical problem. Prompt identification and treatmentf a cervical pseudomeningocele are mandatory.hese lesions often present in a delayed manner anday be associated with significant neurologic decline.aso et al15 reported the case of a 26-year-old manith atlanto-occipital dislocation following a motor

ehicle accident. At 3.5 months into the patient’secovery, he developed significant neurologic deteri-ration that included headache, nausea, emesis, cog-itive decline, and progressive dysphagia requiringercutaneous endoscopic gastrostomy tube place-ent and tracheostomy. Magnetic resonance imaging

evealed the retropharyngeal pseudomeningocele,hich was treated by ventriculoperitoneal shunting.he patient’s neurologic function improved immedi-tely. The patient was decannulated and the gastros-omy tube was removed. At the time of discharge, theatient was able to perform the activities of daily

iving and was able to ambulate with a walker.Hosono et al14 reported a case of a cervical pseudo-eningocele occurring 14 years after removal of a

enign spinal cord tumor. In this instance, the patienteveloped gait disturbance and spasticity of his rightpper extremity. Magnetic resonance imaging re-ealed a pseudomeningocele posterior to the sub-rachnoid space with herniation of the spinal cordnto the cyst cavity. Following surgical exploration,xcision of the pseudomeningocele, and reduction ofhe herniated spinal cord, the patient’s neurologicmpairments resolved.

In our case, the patient was morbidly obese anduadriplegic and had bilateral pulmonary contusions.racheostomy was required to improve pulmonary

oilet and allow for a slow ventilatory wean. Pro-onged oral intubation would not allow for ventilatory

eaning due to her body habitus and high airwayressures required to ventilate the patient. However,ith the cervical pseudomeningocele located anterior

o the trachea, tracheostomy carried the risk of seed-ng a sterile CSF collection with respiratory flora. Inddition, surgical repair of the dural leak was notossible due to the inaccessible location of the duralisruption. Therefore, the need to decompress theseudomeningocele and allow for healing of the dural

isruption was imperative.

1

It was the opinion of the treating services to pro-eed with lumbar drain placement. This was techni-ally difficult due to the patient’s body habitus, cer-ical spine injury, and the presence of thendotracheal tube. The lumbar drain was placed onhe third attempt using fluoroscopic guidance. Theervical pseudomeningocele was decompressed andracheostomy was performed without complication.

No series exists that prospectively or retrospec-ively compares treatment options for the manage-ent of a cervical pseudomeningocele. We offer our

xperience to increase the awareness of this uniquelinical problem. Treatment guidelines cannot be of-ered based on our limited experience. The etiologynd surgical accessibility of the dural disruption, theresence or absence of hydrocephalus, and the neu-ologic status of the patient should be consideredhen deciding on a treatment course.

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