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Show Orbital Cerebrospinal Fluid Accumulation After Complicated Pterional-Orbitozygomatic Craniotomy Michael K. Yoon, MD, Wachirapon Jordan Piluek, MD, Jason P. Ruggiero, MD, Michael W. McDermott, MD, Timothy J. McCulley, MD Abstract: We describe 2 patients who developed postopera-tive orbital cerebrospinal fluid (CSF) collection after orbitozygo-matic pterional craniotomy. An 18-year-old woman underwent exploratory pterional-orbitozygomatic craniotomy. Five days postoperatively, after removal of a lumbar drain, proptosis and a compressive optic neuropathy developed. Computed tomography demonstrated a CSF collection contiguous with the craniotomy site. Resolution followed percutaneous aspira-tion and replacement of the lumbar drain. A 57-year-old woman underwent a pterional-orbitozygomatic craniotomy for removal of a left anterior clinoid meningioma, complicated by a large left hemorrhagic stroke requiring decompressive hemicraniectomy. Extracranial CSF collections accumulated in both the orbit and subgaleal spaces. Resolution followed placement of an exter-nal ventricular drain. Based on these cases, the mechanism seems to be the combination of iatrogenic formation of a com-munication with the subarachnoid space and elevated intracra-nial pressure. Resolution was achieved by normalizing intracranial pressure. Journal of Neuro-Ophthalmology 2014;34:346-349 doi: 10.1097/WNO.0000000000000125 © 2014 by North American Neuro-Ophthalmology Society Cerebrospinal fluid (CSF) leaks are most commonly encountered after trauma. With facial trauma, the inci-dence has been reported to range from 0.5% to 25% of cases (1,2). In the setting of trauma, few reports of orbital CSF accumulation have been published (3-5). CSF leaks can also follow cranial surgery (6,7). This may result in CSF commu-nicating with the nasal or paranasal sinus cavities presenting as rhinorrhea or the auditory canal resulting in otorrhea. When craniotomies involve disruption of the bony orbit, such as with a pterional-orbitozygomatic craniotomy, a communication between the orbital and CSF spaces is created. We describe 2 patients who developed orbital CSF collections presenting with proptosis after craniotomy and discuss pathophysiology and management of this complication. CASE REPORT Case 1 An 18-year-old woman underwent pterional-orbitozygo-matic craniotomy for attempted removal of a bony fragment in the hypothalamus after complicated endoscopic nasal surgery. The specific mechanism of injury during the nasal surgery was not documented. After the pterional-orbitozy-gomatic craniotomy, an abdominal fat graft was placed into the sphenoid sinus to occlude the sellar defect. A small piece of pericranium was then laid over the fat. Dura was closed with suture and pericranium reflected over the orbitozygo-matic osteotomy. The supraorbital bone was fixated with miniplates and screws. The pterion was reconstructed with titanium mesh. The temporalis muscle was closed over this. A lumber drain was placed. On the first postoperative day, the ophthalmology service was consulted for the assessment of reduced vision in the left eye. Visual acuity could not be precisely measured because of decreased mental status. However, letters on a near card were identified with the right but not the left eye. There was a left relative afferent pupillary defect (RAPD). Extraocular motility evaluation was limited, but no gross abnormalities or asymmetry were present. Department of Ophthalmology (MKY), Harvard Medical School, Boston, Massachusetts; Ophthalmic Plastic Surgery, Massachusetts Eye and Ear Infirmary (MKY), Boston, Massachusetts; Wilmer Eye Institute (WJP, TJM), Johns Hopkins University School of Medicine, Baltimore, Maryland; Department of Ophthalmology (JPR), University of Penn-sylvania, Philadelphia, Pennsylvania; and Department of Neurological Surgery (MWM), University of California, San Francisco, California. Supported in part by an unrestricted grant to the Wilmer Ophthal-mological Institute by Research to Prevent Blindness, Inc. Presented at the North American Neuro-Ophthalmology Annual Meeting, Vancouver, BC, Canada, February 8, 2011. The authors report no conflicts of interest. Address correspondence to Timothy J. McCulley, MD, Wilmer Eye Institute, Johns Hopkins University School of Medicine, 600 North Wolfe Street, Wilmer 110, Baltimore, MD 21287; E-mail: tmccull5@ jhmi.edu 346 Yoon et al: J Neuro-Ophthalmol 2014; 34: 346-349 Original Contribution Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Intraocular pressure was 16 mm Hg in the right eye, and 14 mm Hg in the left eye. The remainder of the anterior segment and dilated fundus examinations was normal. Five days postoperatively, the lumbar drain was removed. Within 3 hours, the patient reported marked left periocular pain with diplopia and further reduction of vision in her left eye. She was able to count fingers with her left eye. The left RAPD was more pronounced, and left eye motility was reduced in all directions. There was 6 mm of left proptosis with lagophthalmos and corneal exposure. Computed tomography (CT) revealed a homogeneous lenticular-shaped mass in the superolateral aspect of the orbit, adjacent to the osteotomy, with inferomedial dis-placement of the orbital contents (Fig. 1). Percutaneous aspiration of serosanguinous fluid from the left superolateral orbit was performed, with immediate resolution of the proptosis. Beta-2 transferrin assessment confirmed the fluid to be CSF. The lumbar drain was replaced with the removal of 15 mL of CSF per hour. Within 24 hours, the patient reported improvement of vision in her left eye. Five days later, the lumbar drain was removed. CSF did not reaccumulate in the orbit. Six weeks later, visual acuity was 20/20 in the right eye and hand motions in the left eye. There was no proptosis, and extraocular movements of the left eye were full. Case 2 A 57-year-old woman underwent resection of a large left anterior clinoid meningioma using a left pterional- orbitozygomatic craniotomy. On preoperative ophthalmic evaluation, visual acuity was 20/30 in the right eye and 20/800 in the left eye with a left RAPD, bilateral optic disc pallor, no proptosis, and normal ocular motility. Dura was closed directly and partially grafted with pericranium. A second piece of pericranium was placed extradurally and covered with Tisseel glue (Baxter Healthcare Corporation, Deerfield, IL). Previously removed orbital bone flaps were replaced and fixated with titanium miniplates. The pterion was reconstructed using titanium mesh. Gaps were filled with hydroxyapatite. The temporalis muscle was closed over this. A lumbar drain was placed at the end of surgery and was removed on the fifth postoperative day. Her recovery after the discharge was complicated by a left hemorrhagic stroke with edema on postoperative Day 14 requiring de-compressive left hemicraniectomy. An ophthalmology consult was requested 3 days later to evaluate new onset left proptosis. The patient was intubated and so visual acuity could not be measured. There was a left RAPD and 7 mm of left proptosis (Fig. 2A, B). On CT, there was a large left superotemporal orbital fluid collection contiguous with the subgaleal space overlying the hemicra-niectomy site (Fig. 3). CT-guided percutaneous drainage of the subgaleal fluid was performed with the removal of 78 mL of serosangui-nous fluid. Beta-2 transferrin assessment confirmed the fluid to be CSF. Despite this, 4 days later CSF in the orbit and subgaleal space persisted. A right frontal external ventricular drain was placed, and within 3 days, the left proptosis had resolved. The ventricular drain was removed 11 days later without recurrence. Visual acuity was not measured because of the patient's aphasia, but she had a per-sistent left RAPD. Her ophthalmic evaluation remained unchanged 1 year later. DISCUSSION CSF accumulation into the orbit is rare. We are aware of only 2 such published reports, both of which occurred after trauma, not surgery (3,4). Many case series that describe outcomes, including complications, fail to identify orbital CSF accumulation (7-12). Although various types of CSF leaks after craniotomies may occur, fluid accumulation within the orbit has not been well described. The rarity of orbital CSF accumulation after pterional- orbitozygomatic craniotomy is likely because of the early tamponade as fluid begins to accumulate within the closed space of the orbit. Egress into the orbit is dependent not only on a physical communication but also on a pressure gradient between the 2 compartments. Without an eleva-tion in intracranial pressure (ICP), CSF does not flow into the orbit. Both of our patients developed orbital collections of CSF after removal of a lumbar drain and both improved with replacement. Postsurgical orbital CSF accumulation contrasts nasal, paranasal sinus, and auditory canal leaks. These air-filled spaces have pressures equal or close to atmo-spheric pressure, and therefore the presence of a fistula with normal CSF pressure can result in a leak. The tissue-filled orbit normally has a pressure greater than the atmospheric pressure. Orbital pressure increases further as fluid accumu-lates (13), explaining why orbital CSF leaks do not occur under normal physiologic conditions. Intracranial pressure is estimated to be elevated in approximately 17% of patients undergoing elective crani-otomy (14). Added risk factors for elevated ICP include surgery for glioblastoma, repeated surgery for recurrent tumor, and surgery duration greater than 6 hours (14). In procedures with a high-risk of postoperative leak, FIG. 1. Case 1: Coronal computed tomography demon-strates lenticular-shaped cerebrospinal fluid accumulation (arrows) in the left superolateral orbit. Yoon et al: J Neuro-Ophthalmol 2014; 34: 346-349 347 Original Contribution Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. neurosurgeons often elect to place a lumbar drains postop-eratively. In our institution, removal of the lumbar drain is typically preceded by clamping the drain and observing the patient for 24 hours. If there is no identifiable leak, the drain is removed. In our first patient, a lumbar drain was placed after craniotomy with ICP maintained less than 15 cm H2O. After removal of the drain, intracranial pressure presumably increased to a level where CSF flowed through the fistula into the orbit. Although ICP was not measured, replace-ment of the drain led to resolution of CSF accumulation in the orbit. Our second patient did not have a lumbar drain placed after hemicraniectomy. Despite the removal of a bone flap, intracranial pressure was presumably elevated, suffi-cient to cause CSF collection within the subgaleal space and orbit. Reducing the gradient by lowering ICP through the extraventricular drain was necessary and sufficient to eliminate the fluid collection within the orbit. Again, ICP was not directly measured, and a precise estimation of the pressure gradient required to cause CSF to flow into the orbit cannot be made. In a case with similarities to ours, delayed development of a posttraumatic orbital CSF collection was reported 10 months after the initial injury (4). Surgical repair of the traumatic communication was attempted with direct closure of the leak in the posterior orbital roof. Despite this, CSF drainage through the incision persisted. Only after the placement of multiple lumbar punctures, presumably low-ering ICP, did resolution occur. Periocular CSF leaks in other clinical settings have been described (15,16). Limawararut et al (16) identified 13 CSF leaks in 376 (3.5%) cases of orbital exenteration, 33 in 1,511 (2.2%) cases of orbital decompression, and no cases in 3,504 dacryocystorhinostomies. With orbital exentera-tion, the orbital pressure is reduced to atmospheric pressure, and closure of the leak is required. Management of CSF leak after orbital decompression is determined in part by the location of the leak. Several reports describe CSF accumu-lating in the eyelid or ocular surface, all of which followed trauma (17-20). When designing a treatment plan for such cases, both the pressure gradient and site of communication should be taken into consideration. This is a small retrospective series and should be interpreted accordingly. Both patients had significant comorbidities, including the need for more than 1 surgery and stroke with aphasia limiting visual assessment in our second patient. Despite this, our report suggests that orbital accumulation of CSF after pterional-orbitozygomatic crani-otomy can be managed by normalizing ICP. REFERENCES 1. Ommaya KA. Cerebrospinal fluid fistula and pneumocephalus. In: Rengachary SS, Wilkins RH, eds. Neurosurgery. New York, NY: McGraw Hill Text, 1996:2273-2282. 2. Perrucci JT. Taveras and Ferrucci's Radiology. Philadelphia, PA: Lippincott Williams & Wilkins, 2003. FIG. 2. Case 2: Orbital cerebrospinal fluid accumulation after pterional-orbitozygomatic craniotomy before (A and B) and after (C and D) normalization of intracranial pressure. Note the fullness of the left temporal scalp (asterisks) denoting subgaleal fluid. FIG. 3. Case 2: Coronal computed tomography shows left orbital fluid collection (arrows) contiguous with intracranial and subgaleal fluid collections. 348 Yoon et al: J Neuro-Ophthalmol 2014; 34: 346-349 Original Contribution Copyright © North American Neuro-Ophthalmology Society. 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