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Show Cerebrospinal Fluid Leak Presenting as Oculorrhea After Blunt Orbitocranial Trauma Alexandra O. Apkarian, MD, Shawn L. Hervey-Jumper, MD, Jonathan D. Trobe, MD Abstract: Cerebrospinal fluid (CSF) leak is an uncommon but well-documented occurrence after blunt head trauma, typically manifesting as otorrhea or rhinorrhea. Blunt cranio-orbital trauma also may cause CSF leak into the orbit, manifesting as orbitocele, blepharocele, chemosis, or tearing ("oculorrhea"). We report a patient who developed oculorrhea after blunt head trauma, and neuroimaging dis-closed comminuted fractures of the left frontal, greater sphenoid wing, nasal, and maxillary bones. Because he also displayed chemosis and markedly reduced ocular duc-tions and periocular pain, carotid-cavernous fistula was sus-pected but appropriate vascular imaging was negative. Aspiration of subconjunctival fluid was positive for beta-2 transferrin, a specific marker for CSF. Chemosis lessened and the oculorrhea ceased spontaneously within 6 days of the trauma. This manifestation of CSF leak must not be overlooked because of the threat of meningitis. Journal of Neuro-Ophthalmology 2014;34:271-273 doi: 10.1097/WNO.0000000000000099 © 2014 by North American Neuro-Ophthalmology Society After head trauma, cerebrospinal fluid (CSF) leak occurs in 2%-3% of cases and usually presents as otorrhea or rhinorrhea (1). It has also been reported after orbital and sinus surgery (2) and penetrating orbital injury (3), manifest-ing as orbitocele (CSF remains trapped posterior to the orbital septum), blepharocele (CSF remains trapped anterior to the orbital septum), chemosis, or tearing ("oculorrhea"). We present a case of a CSF oculorrhea occurring after blunt head trauma in a motor vehicle accident and document how subconjunctival needle aspiration yielded CSF and established the correct diagnosis. CASE REPORT Called to the scene of a traffic accident, a 34-year-old policeman sustained closed head trauma after being struck by an oncoming automobile as he exited the driver's side door of his car. He briefly lost consciousness, and upon awakening, he noted left facial pain. Cranial computed tomography (CT) disclosed multiple comminuted fractures involving the left frontal, orbital, and maxillofacial bones (Fig. 1). After 3 days, before repair of his facial fractures, he experienced an episode of vomiting and suddenly developed marked swelling of soft tissues around the left eye and increased periocular pain. Ophthalmologic examination showed absent abduction and reduced supraduction, infra-duction, and adduction of the left eye. Sensation over the second trigeminal division was decreased on the left side. Bullous chemosis was present in the lower fornix without conjunctival lacerations (Fig. 2). Ophthalmoscopic exami-nation was unremarkable. These findings were suggestive of carotid-cavernous fistula, but CT angiography was unremarkable. A neuro-surgical consultant noted copious tearing coming from the left eye upon leaning the patient's head forward, raising the possibility of a CSF leak through the orbit. The tearing ceased when he lay supine. Subconjunctival fluid aspirated with a 25-guage needle was positive for beta-2-transferrin, confirming the presence of CSF. Although treatment with a lumbar drain was considered, the oculorrhea and chemosis spontaneously resolved on the sixth post-trauma day. The patient's zygomatic and inferior orbital floor fractures were repaired the next week. It was presumed that the frontal bone fracture with an associated dural laceration was the source of the CSF leak. DISCUSSION Skull base fractures result in CSF otorrhea when a dural tear allows CSF to flow through the temporal bone or mastoid fracture into the middle ear. CSF may also appear as rhinorrhea if it flows through a fractured cribriform plate Departments of Ophthalmology and Visual Sciences (AOA, JDT), Neurosurgery (SLH-J, JDT), and Neurology (JDT), University of Michigan Kellogg Eye Center, Ann Arbor, Michigan. The authors report no conflicts of interest. Address correspondence to Jonathan D. Trobe, MD, University of Michigan Kellogg Eye Center, 1000 Wall Street, Ann Arbor, MI 48105; E-mail: jdtrobe@umich.edu Apkarian et al: J Neuro-Ophthalmol 2014; 34: 271-273 271 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. and into the paranasal sinuses and nasal cavity. Otorrhea and rhinorrhea occur readily because of a relatively high-pressure gradient between the intracranial space and the external ear canal or paranasal sinuses. However, the route of CSF leak in oculorrhea is through a dural tear and a fracture in the frontal bone, lateral wall, or roof of the orbit. Orbital manifestations of CSF leak are presumably rare because the pressure gradient between the intracranial subarachnoid space and the intra-orbital space low is low (4-7). As no conjunctival tears have been demonstrated in these cases, the supposition is that the CSF exudes through intact conjunctiva. Establishing the diagnosis of cranio-orbital fistula can be challenging because of the difficulty in differentiating oculorrhea from excessive lacrimation caused by orbital soft tissue trauma or epiphora caused by lacrimal outflow obstruction (4). To date, 10 cases of post-traumatic CSF oculorrhea have been reported (1,4-12). One patient developed increasing and pulsatile proptosis 1 week after a motor vehicle accident. CT showed orbital roof fractures. Orbital ultrasonography demonstrated an orbital cyst. Metrizamide cisternography with CT showed that the cyst communicated with the CSF space (5). In another case, an 8-month-old girl had pulsatile proptosis of the left eye noted 3 days after a motor vehicle accident. A transfemoral retrograde 4-vessel angio-gram in pursuit of a diagnosis of carotid-cavernous fistula was negative. The proptosis increased and chemosis devel-oped. Fluid with high sugar content was found escaping from the left nostril and flowing as tears. She underwent surgical exploration that showed a fracture of the left cribriform plate. The associated dural laceration was repaired (7). Chemical analysis of CSF traditionally involved measure-ment of glucose concentration (13), but has been replaced by immunoanalysis for beta-2-transferrin. The protein is present in CSF but not in nasal secretions or tears (4,6,14,15). Ryall et al (14) retrospectively analyzed 11 consecutive cases of CSF leak after head injury, noting 100% sensitivity and specificity of the beta-2-transferrin test. A CSF leak places the patient at risk for infection within the central nervous system. Regarding CSF oculorrhea, Dryden et al (6) described a 4-year-old boy with 2 years of tearing after a motor vehicle accident and basilar skull frac-ture. The tearing was attributed to nasolacrimal duct obstruction, and the patient underwent dacryocystorhinos-tomy (DCR). He had continued tearing and developed meningitis 1 week postoperatively. Maxillofacial CT showed "a bony defect in the posterior orbit, which did not communicate with the DCR ostium." The "tears" were analyzed for glucose and found consistent with CSF. FIG. 1. Maxillofacial computed tomography. A. Coronal view shows comminuted fractures involving the left frontal bone extending into the orbital roof (arrow) and maxillary sinus (arrowhead). B. Axial view shows left lateral orbital wall fracture (arrow) and fracture involving the greater wing of left sphenoid (arrowhead). FIG. 2. Appearance of the left eye 3 days after head trauma. There is proptosis and lid ecchymosis with marked chemosis. Copious tearing was evident only when the patient bent forward. 272 Apkarian et al: J Neuro-Ophthalmol 2014; 34: 271-273 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Craniotomy performed 10 days later revealed an encephalo-cele extending through a fracture in the posterior medial orbital roof and a silastic implant placed over the defect stop-ped the oculorrhea. Sibony et al (5) reported a 27-year-old man with a post-traumatic CSF cyst of the orbit. Cisternog-raphy showed an orbital cyst communicating with the sub-arachnoid space. The patient developed a cerebral abscess, and CT showed a "large defect in the orbital roof." He was suc-cessfully treated with antibiotics. Because orbital tissues tamponade most CSF leaks, repair of orbital fractures should be deferred so as not to disturb a recently sealed leak. Treatment options for CSF oculorrhea include observation, CSF diversion through lumbar drain placement, and surgical repair, including closure of the dural tear, repair of the fractured bones of anterior skull base, and obliteration of the epidural space with fibrin glue. Many authors recommend an initial 24-hour period of observation because an estimated 85% of fistulas will close spontaneously. The addition of lumbar drain placement results in fistula closure in 95% of patients (16). Surgical repair is recommen-ded for extensive comminuted or displaced skull base frac-tures for leaks persisting more than 48 hours (1). REFERENCES 1. Chandran N, Ojha BK, Chandwani V, Srivastava C, Sing SK, Chandra A. A rare case of posttraumatic eyelid swelling: cerebrospinal fluid blepharocele. 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