CLINICAL STUDY
Iatrogenic Cerebrospinal Fluid Oculorrhea Mijung Chi, MD, PhD,*Þ H. Jane Kim, MD,Þ Bengu E. Koktekir, MD,Þþ Reza Vagefi, MD,Þ and Robert C. Kersten, MDÞ Abstract: Cerebrospinal fluid leakage into the orbit (CSF orbitorrhea) or through the orbit to the exterior (CSF oculorrhea) occurs when there is a communication between orbit and subarachnoid space. It has rarely been described. We report a case of CSF oculorrhea following surgery for recurrent sphenoid wing meningioma. A 67-year-old patient who underwent craniotomy for a recurrent sphenoid wing meningioma complained of tearing from the ipsilateral eye. Fluid collection was observed in the right periorbital area contiguous with craniotomy wound. With wide retraction of eyelids, a 1-cm longitudinal full-thickness laceration through the conjunctiva toward the lateral orbital rim was visualized in the area of the lateral canthal tendon. Steady leakage of pinktinged serous fluid through the forniceal laceration was observed. With a presumed diagnosis of iatrogenic CSF oculorrhea, the patient was treated conservatively with resolution by postoperative day 6. Key Words: Cerebrospinal fluid leak, CSF oculorrhea, CSF orbitorrhea, iatrogenic CSF leak (J Craniofac Surg 2014;25: 469Y470)
C
erebrospinal fluid (CSF) leak is a well-known complication of craniocerebral injury by trauma or surgery.1,2 It occurs in 2% to 3% of craniocerebral injury cases and usually manifests as rhinorrhea or otorrhea. However, leakage of CSF into the orbit (CSF orbitorrhea) or through the orbit to the exterior (CSF oculorrhea) is a rare phenomenon. Fewer than 15 cases of CSF orbitorrhea or CSF oculorrhea have been described, all following head trauma.3Y6 We present a unique case of iatrogenic CSF oculorrhea after surgery for sphenoid wing meningioma. All medical records and other individually identifiable health information used or disclosed in this article are kept properly confidential by the Health Insurance Portability and Accountability Act.
CLINICAL REPORT A 67-year-old man consulted the neurosurgery department for tearing from the right eye noted after lumbar drain was removed on postoperative day 2 following craniotomy.
He had undergone an operation for a right sphenoid wing meningioma via a pterional craniotomy in another institution several years before with resultant no light perception vision. He presented to our institution with a giant recurrence which required reoperation (Fig. 1A). A frontotemporal orbital zygomatic craniotomy with exposure of the roof and lateral wall of the orbit was performed by the neurosurgery team. A standard coronal periosteal flap was reflected, and the temporalis muscle was disinserted from the superior temporal line. Subfascial dissection was used to expose the zygomatic arch, body of the zygoma, and frontozygomatic process, followed by the dissection of the orbital roof and lateral wall. Abundant scar tissue was observed at the site of the previous pterional bone flap, making the elevation of the bone difficult. Zygomatic osteotomy was carried out using an Anspach (DePuy Synthes, West Chester, PA) drill. During tumor removal, dural tears in the area of the lateral wall of the cavernous sinus and right frontal lobe were encountered and repaired using Surgisis (Cook Surgical, Bloomington, IN), DuraGen (Integra LifeSciences Co, Plainsboro, NJ), sponge, and fibrin glue. The osteotomy was repaired with combination of titanium mesh, Medpor orbital implant (Stryker, Fremont, CA), and hydroxyapatite. The patient had unremarkable recovery with exception of fluid collection under the craniotomy wound on the right side. The lumbar drain was removed on postoperative day 2, and ophthalmology was consulted on postoperative day 3 for ‘‘right eye tearing.’’ On examination, vision was no light perception with a relative afferent pupillary defect and intraocular pressure of 30 mm Hg in the right eye. Extraocular muscle movement was mildly restricted in abduction and adduction. There was copious pink-tinged serous fluid flowing from the conjunctival fornices. With wide retraction of the eyelids, a 1-cm longitudinal full-thickness laceration through the conjunctiva toward the lateral orbital wall was discovered in the area of the lateral canthal tendon (Fig. 1B). There was mild chemosis and subconjunctival hemorrhage, but the globe was intact. With a presumed diagnosis of iatrogenic CSF oculorrhea, the patient was treated conservatively with pressure patching, prophylactic systemic antibiotic medication, bed rest, elevation of the head, and stool softener. Without lumbar drainage or systemic diuretics, CSF leakage slowly improved and completely resolved by postoperative day 6.
DISCUSSION From the *Department of Ophthalmology, Gachon University Gil Hospital, Incheon, Korea; †Department of Ophthalmology, University of California, San Francisco, California; and ‡Department of Ophthalmology, Selcuk University, Konya, Turkey. Received October 23, 2013. Accepted for publication November 11, 2013. Address correspondence and reprint requests to Robert C. Kersten, MD, Department of Ophthalmology, University of California, San Francisco, 10 Koret Way, K 304, San Francisco, CA; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000535
The Journal of Craniofacial Surgery
Traumatic CSF leaks usually result from tears in the dura of the skull base that allow CSF to escape from the subarachnoid space into the nasopharynx, the paranasal sinuses, or the subcutaneous plane. The cribriform plate area is thin, and the dura at this site firmly invests the olfactory fissure. Approximately 80% of CSF rhinorrhea is related to fracture of the skull base, and 10% to 30% of all skull base fractures develop CSF fistulae.7,8 Besides trauma, CSF leakage has also been described following oculoplastic surgery. Cerebrospinal fluid rhinorrhea may rarely occur in a dacryocystorhinostomy if bone removal extends to the level of the cribriform plate, or rotational movement during creation of the osteotomy transmits forces to the cribriform plate and causes a fracture.1,9 Cerebrospinal fluid
& Volume 25, Number 2, March 2014
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
469
Chi et al
The Journal of Craniofacial Surgery
FIGURE 1. A, T1 magnetic resonance imaging shows a large heterogeneously enhancing mass arising from the right sphenoid wing, which measures approximately 5.5 3.8 5.5 cm. The mass compresses the right optic apparatus and displaces the optic chiasm to the left. B, Conjunctival laceration (arrow) and serous fluid leakage in the area of lateral canthal tendon in the right eye is shown.
leaks have also been reported in cases of orbital exenteration and orbital decompression.1,10 In case of a fracture of the petrous portion of the temporal bone, CSF from the middle or posterior cranial fossa may escape into the mastoids and produce middle ear fluid mimicking otitis media. Rarely CSF tracks subcutaneously, producing a subepicranial hydroma.11 Cerebrospinal fluid leakage into the orbit is very rare, and it has been described as oculorrhea or orbitorrhea. About 5 patients have presented with epiphora, following blunt head injury.3Y6 In a case of a 4-year-old tearing child, CSF leak was suspected only after a successful dacryocystorhinostomy was performed for posttraumatic lacrimal duct outflow obstruction 3 years after naso-orbital injury.5 Our case of CSF leakage occurred following surgery of a recurrent sphenoid wing meningioma. It is postulated that the experienced neurosurgical team inadvertently created a full-thickness laceration from lateral periorbita to conjunctiva at the time of pterional bone flap elevation to access the large recurrent sphenoid wing meningioma. There was abundant scar tissue at the site of the previous bone flap. Tumor resection around the cavernous sinus and convexities of frontal and temporal lobes proved to be challenging, requiring dural tear repairs. It is likely that CSF leaked from the dural tears, entered into the orbit with final egress through the fullthickness conjunctival laceration, onto the ocular surface. Although it was not undertaken in our case, analysis of the oculorrhea fluid for glucose concentration could further confirm the diagnosis. A glucose level of 30 mg/dL or higher is diagnostic. Immunoassay for A2-transferrin is more specific.1,12
470
& Volume 25, Number 2, March 2014
Although treatment of CSF leakage is controversial, most of the cases improve with only conservative management. Conservative management of CSF oculorrhea may include pressure eye patch, bed rest with head elevation, stool softener, and antibiotics for meningitis prevention. Lumbar drainage and medical treatment with diuretics may be considered, especially if high intracranial pressure is suspected. Surgery is indicated if conservative measures fail.1,3,13 Our patient improved with the conservative treatment. To the best of our knowledge, this is the first case of iatrogenic CSF oculorrhea after a neurosurgical procedure. This rare complication must be considered in any patient who presents with ‘‘tearing’’ following neurosurgery or intracranial trauma.
REFERENCES 1. Badilla J, Dolman PJ. Cerebrospinal fluid leaks complicating orbital or oculoplastic surgery. Arch Ophthalmol 2007;125:1631Y1634 2. Limawararut V, Valenzuela AA, Sullivan TJ, et al. Cerebrospinal fluid leaks in orbital and lacrimal surgery. Surv Ophthalmol 2008;53:274Y284 3. Salame K, Segev Y, Fliss DM, et al. Diagnosis and management of posttraumatic oculorrhea. Neurosurg Focus 2000;9:1Y4 4. Joshi KK, Crockard HA. Traumatic cerebrospinal fluid fistula simulating tears. J Neurosurg 1978;49:121Y123 5. Dryden RM, Wulc AE. Pseudoepiphora from cerebrospinal fluid leak: case report. BJO 1986;70:570Y574 6. Till JS, Marion JR. Cerebrospinal fluid masquerading as tears. South Med J 1987;80:639Y640 7. Loew F, Pertuiset B, Chaumier EE, et al. Traumatic, spontaneous and postoperative CSF rhinorrhea. Adv Tech Stand Neurosurg 1984;11:169Y207 8. Friedman JA, Ebersold MJ, Quast LM. Post-traumatic cerebrospinal fluid leakage. World J Surg 2001;25:1062Y1066 9. Neuhaus RW, Baylis HI. Cerebrospinal fluid leakage after dacryocystorhinostomy. Ophthalmology 1983;90:1091Y1095 10. Wulc AE, Adams JL, Dryden RM. Cerebrospinal fluid leakage complicating orbital exenteration. Arch Ophthalmol 1989;107:827Y830 11. Cullen JR. Unfamiliar swelling after head trauma. Clin Pediatr 1974;13:378 12. Ryall RG, Peacock MK, Simpson DA. Usefulness of beta 2-transferrin assay in the detection of cerebrospinal fluid leaks following head injury. J Neurosurg 1992;77:737Y739 13. Woodworth BA, Prince A, Chiu AG, et al. Spontaneous CSF leaks: a paradigm for definitive repair and management of intracranial hypertension. Otolaryngol Head Neck Surg 2008;138:715Y720
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Iatrogenic Cerebrospinal Fluid Oculorrhea Mijung Chi, MD, PhD,*Þ H. Jane Kim, MD,Þ Bengu E. Koktekir, MD,Þþ Reza Vagefi, MD,Þ and Robert C. Kersten, MDÞ Abstract: Cerebrospinal fluid leakage into the orbit (CSF orbitorrhea) or through the orbit to the exterior (CSF oculorrhea) occurs when there is a communication between orbit and subarachnoid space. It has rarely been described. We report a case of CSF oculorrhea following surgery for recurrent sphenoid wing meningioma. A 67-year-old patient who underwent craniotomy for a recurrent sphenoid wing meningioma complained of tearing from the ipsilateral eye. Fluid collection was observed in the right periorbital area contiguous with craniotomy wound. With wide retraction of eyelids, a 1-cm longitudinal full-thickness laceration through the conjunctiva toward the lateral orbital rim was visualized in the area of the lateral canthal tendon. Steady leakage of pinktinged serous fluid through the forniceal laceration was observed. With a presumed diagnosis of iatrogenic CSF oculorrhea, the patient was treated conservatively with resolution by postoperative day 6. Key Words: Cerebrospinal fluid leak, CSF oculorrhea, CSF orbitorrhea, iatrogenic CSF leak (J Craniofac Surg 2014;25: 469Y470)
C
erebrospinal fluid (CSF) leak is a well-known complication of craniocerebral injury by trauma or surgery.1,2 It occurs in 2% to 3% of craniocerebral injury cases and usually manifests as rhinorrhea or otorrhea. However, leakage of CSF into the orbit (CSF orbitorrhea) or through the orbit to the exterior (CSF oculorrhea) is a rare phenomenon. Fewer than 15 cases of CSF orbitorrhea or CSF oculorrhea have been described, all following head trauma.3Y6 We present a unique case of iatrogenic CSF oculorrhea after surgery for sphenoid wing meningioma. All medical records and other individually identifiable health information used or disclosed in this article are kept properly confidential by the Health Insurance Portability and Accountability Act.
CLINICAL REPORT A 67-year-old man consulted the neurosurgery department for tearing from the right eye noted after lumbar drain was removed on postoperative day 2 following craniotomy.
He had undergone an operation for a right sphenoid wing meningioma via a pterional craniotomy in another institution several years before with resultant no light perception vision. He presented to our institution with a giant recurrence which required reoperation (Fig. 1A). A frontotemporal orbital zygomatic craniotomy with exposure of the roof and lateral wall of the orbit was performed by the neurosurgery team. A standard coronal periosteal flap was reflected, and the temporalis muscle was disinserted from the superior temporal line. Subfascial dissection was used to expose the zygomatic arch, body of the zygoma, and frontozygomatic process, followed by the dissection of the orbital roof and lateral wall. Abundant scar tissue was observed at the site of the previous pterional bone flap, making the elevation of the bone difficult. Zygomatic osteotomy was carried out using an Anspach (DePuy Synthes, West Chester, PA) drill. During tumor removal, dural tears in the area of the lateral wall of the cavernous sinus and right frontal lobe were encountered and repaired using Surgisis (Cook Surgical, Bloomington, IN), DuraGen (Integra LifeSciences Co, Plainsboro, NJ), sponge, and fibrin glue. The osteotomy was repaired with combination of titanium mesh, Medpor orbital implant (Stryker, Fremont, CA), and hydroxyapatite. The patient had unremarkable recovery with exception of fluid collection under the craniotomy wound on the right side. The lumbar drain was removed on postoperative day 2, and ophthalmology was consulted on postoperative day 3 for ‘‘right eye tearing.’’ On examination, vision was no light perception with a relative afferent pupillary defect and intraocular pressure of 30 mm Hg in the right eye. Extraocular muscle movement was mildly restricted in abduction and adduction. There was copious pink-tinged serous fluid flowing from the conjunctival fornices. With wide retraction of the eyelids, a 1-cm longitudinal full-thickness laceration through the conjunctiva toward the lateral orbital wall was discovered in the area of the lateral canthal tendon (Fig. 1B). There was mild chemosis and subconjunctival hemorrhage, but the globe was intact. With a presumed diagnosis of iatrogenic CSF oculorrhea, the patient was treated conservatively with pressure patching, prophylactic systemic antibiotic medication, bed rest, elevation of the head, and stool softener. Without lumbar drainage or systemic diuretics, CSF leakage slowly improved and completely resolved by postoperative day 6.
DISCUSSION From the *Department of Ophthalmology, Gachon University Gil Hospital, Incheon, Korea; †Department of Ophthalmology, University of California, San Francisco, California; and ‡Department of Ophthalmology, Selcuk University, Konya, Turkey. Received October 23, 2013. Accepted for publication November 11, 2013. Address correspondence and reprint requests to Robert C. Kersten, MD, Department of Ophthalmology, University of California, San Francisco, 10 Koret Way, K 304, San Francisco, CA; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000535
The Journal of Craniofacial Surgery
Traumatic CSF leaks usually result from tears in the dura of the skull base that allow CSF to escape from the subarachnoid space into the nasopharynx, the paranasal sinuses, or the subcutaneous plane. The cribriform plate area is thin, and the dura at this site firmly invests the olfactory fissure. Approximately 80% of CSF rhinorrhea is related to fracture of the skull base, and 10% to 30% of all skull base fractures develop CSF fistulae.7,8 Besides trauma, CSF leakage has also been described following oculoplastic surgery. Cerebrospinal fluid rhinorrhea may rarely occur in a dacryocystorhinostomy if bone removal extends to the level of the cribriform plate, or rotational movement during creation of the osteotomy transmits forces to the cribriform plate and causes a fracture.1,9 Cerebrospinal fluid
& Volume 25, Number 2, March 2014
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
469
Chi et al
The Journal of Craniofacial Surgery
FIGURE 1. A, T1 magnetic resonance imaging shows a large heterogeneously enhancing mass arising from the right sphenoid wing, which measures approximately 5.5 3.8 5.5 cm. The mass compresses the right optic apparatus and displaces the optic chiasm to the left. B, Conjunctival laceration (arrow) and serous fluid leakage in the area of lateral canthal tendon in the right eye is shown.
leaks have also been reported in cases of orbital exenteration and orbital decompression.1,10 In case of a fracture of the petrous portion of the temporal bone, CSF from the middle or posterior cranial fossa may escape into the mastoids and produce middle ear fluid mimicking otitis media. Rarely CSF tracks subcutaneously, producing a subepicranial hydroma.11 Cerebrospinal fluid leakage into the orbit is very rare, and it has been described as oculorrhea or orbitorrhea. About 5 patients have presented with epiphora, following blunt head injury.3Y6 In a case of a 4-year-old tearing child, CSF leak was suspected only after a successful dacryocystorhinostomy was performed for posttraumatic lacrimal duct outflow obstruction 3 years after naso-orbital injury.5 Our case of CSF leakage occurred following surgery of a recurrent sphenoid wing meningioma. It is postulated that the experienced neurosurgical team inadvertently created a full-thickness laceration from lateral periorbita to conjunctiva at the time of pterional bone flap elevation to access the large recurrent sphenoid wing meningioma. There was abundant scar tissue at the site of the previous bone flap. Tumor resection around the cavernous sinus and convexities of frontal and temporal lobes proved to be challenging, requiring dural tear repairs. It is likely that CSF leaked from the dural tears, entered into the orbit with final egress through the fullthickness conjunctival laceration, onto the ocular surface. Although it was not undertaken in our case, analysis of the oculorrhea fluid for glucose concentration could further confirm the diagnosis. A glucose level of 30 mg/dL or higher is diagnostic. Immunoassay for A2-transferrin is more specific.1,12
470
& Volume 25, Number 2, March 2014
Although treatment of CSF leakage is controversial, most of the cases improve with only conservative management. Conservative management of CSF oculorrhea may include pressure eye patch, bed rest with head elevation, stool softener, and antibiotics for meningitis prevention. Lumbar drainage and medical treatment with diuretics may be considered, especially if high intracranial pressure is suspected. Surgery is indicated if conservative measures fail.1,3,13 Our patient improved with the conservative treatment. To the best of our knowledge, this is the first case of iatrogenic CSF oculorrhea after a neurosurgical procedure. This rare complication must be considered in any patient who presents with ‘‘tearing’’ following neurosurgery or intracranial trauma.
REFERENCES 1. Badilla J, Dolman PJ. Cerebrospinal fluid leaks complicating orbital or oculoplastic surgery. Arch Ophthalmol 2007;125:1631Y1634 2. Limawararut V, Valenzuela AA, Sullivan TJ, et al. Cerebrospinal fluid leaks in orbital and lacrimal surgery. Surv Ophthalmol 2008;53:274Y284 3. Salame K, Segev Y, Fliss DM, et al. Diagnosis and management of posttraumatic oculorrhea. Neurosurg Focus 2000;9:1Y4 4. Joshi KK, Crockard HA. Traumatic cerebrospinal fluid fistula simulating tears. J Neurosurg 1978;49:121Y123 5. Dryden RM, Wulc AE. Pseudoepiphora from cerebrospinal fluid leak: case report. BJO 1986;70:570Y574 6. Till JS, Marion JR. Cerebrospinal fluid masquerading as tears. South Med J 1987;80:639Y640 7. Loew F, Pertuiset B, Chaumier EE, et al. Traumatic, spontaneous and postoperative CSF rhinorrhea. Adv Tech Stand Neurosurg 1984;11:169Y207 8. Friedman JA, Ebersold MJ, Quast LM. Post-traumatic cerebrospinal fluid leakage. World J Surg 2001;25:1062Y1066 9. Neuhaus RW, Baylis HI. Cerebrospinal fluid leakage after dacryocystorhinostomy. Ophthalmology 1983;90:1091Y1095 10. Wulc AE, Adams JL, Dryden RM. Cerebrospinal fluid leakage complicating orbital exenteration. Arch Ophthalmol 1989;107:827Y830 11. Cullen JR. Unfamiliar swelling after head trauma. Clin Pediatr 1974;13:378 12. Ryall RG, Peacock MK, Simpson DA. Usefulness of beta 2-transferrin assay in the detection of cerebrospinal fluid leaks following head injury. J Neurosurg 1992;77:737Y739 13. Woodworth BA, Prince A, Chiu AG, et al. Spontaneous CSF leaks: a paradigm for definitive repair and management of intracranial hypertension. Otolaryngol Head Neck Surg 2008;138:715Y720
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
