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1
Oculoplastic and Orbital Division, Department of Ophthalmology & Visual Sciences, University of Adelaide, and South
Australian Institute of Ophthalmology, Adelaide, Australia; 2Eyelid, Lacrimal and Orbital Clinic, Department of
Ophthalmology, University of Queensland Medical School, Brisbane, Australia; 3Orbital Plastic and Lacrimal Clinic,
Royal Victorian Eye and Ear Hospital, Melbourne, Australia; 4Corneoplastic Unit, Queen Victoria Hospital, East
Grinstead, United Kingdom; and 5Department of Neurosurgery, Department of Surgery, University of Adelaide, Adelaide,
Australia
Abstract. Cerebrospinal fluid leakage is an uncommon but significant complication of orbital and
rarely lacrimal surgery which may have serious consequences including death. In a retrospective review
of four orbital units, we report an incidence of cerebrospinal fluid leak (diagnosed intraoperatively)
during exenteration, orbital decompression, and dacryocystorhinostomy of 1/154 (0.6%), 4/397 (1%),
and 0/3,504 (0%), respectively. We found two additional cases of cerebrospinal fluid leaks associated
with excision of orbital masses involving the orbital roof. In the literature, the incidence of
cerebrospinal fluid leaks associated with orbital exenterations and decompressions was 1.6--16.7% and
0--10%, respectively. Cerebrospinal fluid leaks occur very rarely in dacryocystorhinostomies with only
a few case reports found in the literature. Preventative measures, diagnosis, and management of this
complication are discussed. Knowledge of anatomy and thorough preoperative assessment may predict
areas at high risk for encountering cerebrospinal fluid leaks. Proper surgical technique further
minimizes the risk for this complication. If a cerebrospinal fluid leak occurs, however, prompt
diagnosis and management usually results in uncomplicated recovery. (Surv Ophthalmol 53:274--284,
2008. Ó 2008 Elsevier Inc. All rights reserved.)
A cerebrospinal fluid (CSF) leak may complicate cerebral abscess, seizures, CSF hypotension with
orbital and, rarely, lacrimal surgery and may have position dependent headache syndrome, occult hem-
serious consequences including meningitis, delayed orrhage, and even death.15,27,42 In many instances, the
274
Ó 2008 by Elsevier Inc. 0039-6257/08/$--see front matter
All rights reserved. doi:10.1016/j.survophthal.2008.02.009
CEREBROSPINAL FLUID LEAKAGE 275
TABLE 1
Incidence of CSF Leaks in Exenteration
Author Incidence Treatment (n) Outcome
13
de Conciliis 5/39 (12.8%) Dural repair with temporalis muscle fascia Uncomplicated recovery (4) Death 15
or lyophilized heterologous dura days postoperatively due to unrelated
packing with autogenous fat or pulmonary complications (1)
temporalis muscle
Bartley5 3/100 (3%) Conservative management-- Spontaneous resolution
Wulc53 3/18 (16.7%) Enlarged bone defect þ primary closure þ Uncomplicated recovery
gelfoam soaked in thrombin (2)
Enlarged bone defect þ primary closure
þ epicranium graft þ lumbar drain (1)
Rahman37 1/64 (1.6%) Local pericranial flap Uncomplicated recovery
Limawararut 1/154 (0.6%) Gelfoam, free flap (1) Uncomplicated recovery
(current study)
exposure of dura with the risk of a subsequent CSF very rarely in DCRs with a few case reports found in the
leak can be predicted by careful pre-operative assess- literature.2,14,17,18,35 CSF leakage is also a well-docu-
ment and knowledge of anatomy. However, if encoun- mented complication among other surgical proce-
tered, exposure of dura is safe if care is taken to avoid dures in the area of the skull base. This is a risk
a tear.22 Even if a dural defect is created, the morbi- particularly during skull base surgery, functional
dity associated with CSF leaks may be minimized if endoscopic surgery (FESS), and open ethmoidectomy.
promptly diagnosed and properly managed. Although We report our experience in four separate orbital
several studies report this complication, the manage- units at the Royal Adelaide Hospital, Adelaide,
ment of CSF leaks in orbital and lacrimal surgery is not Australia; the Corneoplastic Unit, East Grinstead,
well described in the literature. United Kingdom; the Royal Brisbane and Women’s
Hospital, Brisbane, Australia; and the Royal Victorian
Eye and Ear Hospital, Melbourne, Australia. Our
Incidence of CSF Leaks combined incidences of CSF leak (diagnosed intra-
The incidence of CSF leaks associated with orbital operatively) during exenteration, orbital decompres-
and lacrimal surgery is relatively low. We reviewed the sion, and dacryocystorhinostomy are 1/154 (0.6%),
incidence of CSF leaks in orbital exenteration, orbital 4/397 (1%), and 0/3504 (0%), respectively. The sole
decompression, and dacryocystorhinostomy (DCR). case of CSF leak associated with orbital exenteration
Although there is no literature on the incidence of CSF for an orbital desmoplastic melanoma occurred while
leaks in other types of orbital surgery, it is also a risk cutting the optic nerve at the orbital apex. The orbital
when orbital lesions involve the orbital roof or posterior decompressions for thyroid eye disease were per-
lateral wall, especially in erosive processes.13 Such formed medially, either by a transcaruncular or
lesions may include malignant tumors as well as benign swinging eyelid approach, and laterally, by a swinging
lesions such as dermoids, cholesterol granulomas, eyelid or upper skin crease approach. All four cases of
and eosinophilic granulomas. The highest reported CSF leakage during orbital decompression for
incidence of CSF leaks occurred in orbital exenter- thyroid ophthalmopathy occurred while drilling out
ations at a rate of 1.6--16.7% (Table 1).5,13,21,37,53 In the posterior lateral wall. The authors have not
addition, in a series of 39 patients, de Conciliis noted encountered a CSF leak during endonasal, external,
that dural exposure occurred in 20.5% of exentera- redo DCR, or conjunctivo-DCR.
tions and 30.8% of enlarged exenterations which In addition, we found two cases of CSF leak
included excision of one or more orbital walls.13 associated with excision of masses involving the orbital
Furthermore, in that study, 60% of CSF leaks and 75% roof. One of these was an orbital dermoid, and the
of dural exposures occurred in enlarged exenterations other was a recurrent adenoid cystic carcinoma. Both
involving resection of one or more orbital walls. For CSF leaks could have been predicted preoperatively
orbital decompressions, CSF leaks occurred in 0--10% due to the pathologically thinned orbital roof seen on
of cases, with the highest incidence occurring with imaging (Fig. 1).
a coronal approach (Table 2).3,6,16,20,22,23,34,36,38,46,47,48
Most studies reported incidences ranging from 0--4.5%
with transconjunctival, transcaruncular, transantral, Preoperative Assessment
external lateral, swinging eyelid, and endoscopic In many cases, exposure of dura and the risk of
approaches.3,6,16,20,22,23,34,36,38,46,47 CSF leaks occur CSF leak can be predicted by performing a careful
276 Surv Ophthalmol 53 (3) May--June 2008 LIMAWARARUT ET AL
TABLE 2
Incidence of CSF Leaks in Orbital Decompression
Author Approach Incidence Treatment (n) Outcome
3
Bailey Swinging eyelid 2/97 (2%) Conservative management (1) Uncomplicated recovery
Leak patching (1)
Goldberg22 Coronal 2/20 (10%) Tissue glue (2) Uncomplicated recovery
Baylis6 Transantral 0/24 (0%) -- --
Garrity20 Transantral 15/428 (3.5%) Conservative management Uncomplicated recovery
(11) Surgical repair (4)
Fatourechi16 Transantral 1/22 (4.5%) Surgical repair 4 months Uncomplicated recovery
later (1)
Shorr47 Transantral 2/84 (2.4%) Not reported Not reported
Shorr47 Transconjunctival 1/33 (3.0%) Conservative management Uncomplicated
Schaefer38 Combined endoscopic 1/72 (1.4%) Conservative management (1) Uncomplicated recovery
and transconjunctival
Graham23 Transcaruncular or 2/63 (3.1%) Primary repair (2) Uncomplicated recovery
endoscopic
Nadeau34 Endoscopic and 1/73 (1.4%) Nasal turbinate flap and Uncomplicated recovery
external lateral Surgical
Paridaens36 Swinging eyelid 2/198 (1.0%) Temporalis muscle fascia graft Uncomplicated recovery
(1) Lumbar drain (1)
Limawararut External lateral, 4/397 (1.0%) Temporalis muscle graft (1) Uncomplicated recovery
(current study) Swinging eyelid Temporalis muscle graft þ
cyanoacrylate (1) Orbital fat
(1) Bone wax (1)
SURGICAL TECHNIQUE
Using appropriate surgical technique may mini-
mize the risk of dural trauma. Frequent reference to
imaging during the procedure may help guide the
surgeon. Another intraoperative measure which may
play a significant role in avoiding CSF leaks is using
appropriate instrumentation. For example, during
DCRs, sharp rongeurs are important during creation
=
Fig. 4. High-risk areas for encountering dura in the
lateral wall of the orbit. A: Axial view. B: Sagittal view. 1.
While drilling out the marrow space (ms), the inner table
of the greater wing of the sphenoid (gws) bone may be
penetrated. 2. While drilling posterior to the marrow
space the relatively thin bone overlying the middle cranial
fossa (mcf) may be penetrated. 3. While drilling superiorly
through the orbital roof the anterior cranial fossa (acf)
may be entered. iof 5 inferior orbital fissure; sof 5
superior orbital fissure.
CEREBROSPINAL FLUID LEAKAGE 279
Diagnosis
If a CSF leak is encountered during orbital or
lacrimal surgery, prompt diagnosis and treatment
usually leads to favorable results. Diagnosis at the time
of surgery can be made when leakage of clear fluid is
seen in areas at high risk for encountering dura.
When CSF and blood are mixed together, diagnosis
can be difficult. CSF separates from blood when
placed on filter paper and produces a ring or halo
sign. However, this is not exclusive to CSF and can
lead to false-positive results. Glucose content can be
done rapidly; this is also unreliable, however, because
glucose in CSF cannot be distinguished from that in Fig. 6. A stereotactic assistance such as the Stealth system
blood, tears, or nasal secretion. Hence, measuring may be helpful, particularly where normal bony anatomy
glucose from the discharge is generally of little value has been distorted by lesions that involve bone such as
osteomas or fibrous dysplasia, and the skull base is being
in the peri-operative setting. Beta-2 transferrin assay is
approached from below. A: T1-weighted axial MRI of orbit
a marker protein specific to CSF. It has high sensitivity demonstrates a sphenoid wing meningioma. B: The
and specificity, can be performed rapidly, and is the Stealth stereotactic system was used to facilitate tumor
test of choice at most of our institutions. debulking.
280 Surv Ophthalmol 53 (3) May--June 2008 LIMAWARARUT ET AL
Postoperatively for persistent leaks confirmed their use is discouraged, however, due to the risk of
with beta-2 transferrin, the majority of leaks can be Creutzfeldt Jacob or other acquired disease.32
localized with high-resolution CT.42 In a retrospec- Limited exposure of the dura, large areas of dural
tive study, noncontrast high-resolution CT has been loss, or unidentifiable defects may make primary
shown to identify 30/42 (71%) of bone defects with closure impossible. Such leaks may be managed by
CSF leaks.49 If not localized on high-resolution CT, placing an onlay graft against the dural rent or
further interventions that may be useful include suspected area of leakage and suturing the graft to the
intrathecal injection of fluorescein,11,27,52 and surrounding dura. Graft materials described include
radionuclide or contrast-enhanced CT or MRI packing the area with autogenous fat, mucous
cisternography.4,19 membrane, fascia lata, temporalis fascia and muscle,
cartilage, and a osteomucoperiosteal flap.4,17,50 As
described subsequently, these various graft materials
Intraoperative Management can also be combined with a variety of tissue adhesives
EVALUATION OF THE SITE to create a watertight barrier.
In the case of orbital exenteration, packing
Once a CSF leak is identified, appropriate man- material can be placed in the socket to hold the plug
agement may minimize significant morbidity. The of muscle or fascia against the orbital apex.
goal of management with all leaks is to create and
maintain a watertight repair. This will decrease the
risks of excessive CSF drainage, infection (particu-
larly if the leak is into a sinonasal cavity), and aseptic TISSUE ADHESIVES AND ALLOPLASTIC
meningitis due to blood entering the subarachnoid MATERIALS
space.15 Recently, tissue adhesives have been used success-
A careful inspection of the dural laceration and fully in closing dural defects. Although often used in
underlying brain is mandatory. The location and size conjunction with an onlay graft to further seal the
of the dural injury in association with the amount of repair, using an adhesive alone may be adequate in
exposure surrounding the laceration, will determine very small dural defects without tissue loss. Tisseel
the types of repair possible. If the leak into the orbit is fibrin glue22 (Baxter Healthcare Corp., Deerfield, IL)
small, it may close spontaneously3,5,20,35,46 as fat may has been used with favorable results in CSF leaks to
tamponade the area and aid closure. Nevertheless, an the orbit. It consists of human fibrinogen (screened
attempt to repair the leak is advisable to avoid the for human immunodeficiency virus and hepatitis B)
risks of persistent leakage. and bovine thrombin. It is available in four sizes
Neurosurgical consultation is recommended for ranging from 0.5 to 5.0 ml and is in a deep-frozen state
further advice whenever dura is injured. and must be thawed for approximately 20 minutes at
room temperature prior to use. A solid fibrin matrix is
SURGICAL REPAIR created in 5 minutes. The needle must be changed
If there is adequate exposure and the edges of the between applications due to immediate clogging of
dural laceration can be readily apposed, primary the needle (Fig. 7). Cyanoacrylate glue applied to
repair with 5-0 or 6-0 polyglactin or braided nylon a dry surgical field has also been effective in closure of
should be performed to create a watertight seal. Our CSF leaks.50
neurosurgeons recommend removal of enough bone Bone wax23 has also been described in situations
to expose the defect when possible for secure repair. where the leak arises from the depth of a relatively
A 1-mm punch or diamond burr can be used for this small bony defect. The aim is to fill the bone defect
purpose. Many suggest an onlay graft, commonly preventing CSF leakage into the orbit while the dura
a layer of fat, temporalis muscle, or fascia, which may seals itself. However, bone wax should be used with
then be placed over the exposed dura to further caution as Bolger8 reported three cases of CSF leaks
ensure a watertight barrier.15 that were associated with bone wax. The authors
Other sources of autologous grafts include peri- proposed that bone wax controls bleeding during
cranium and periorbita. Additional materials used craniotomy but may also stent a defect open,
effectively include fascia lata, an osteomucoperiosteal preventing fibrin deposition, spontaneous healing,
flap, mucous membrane and lyophilized heterolo- and closure. Furthermore, there have been cases of
gous dura.13 Although mucous membrane has been granulomas associated with bone wax in the orbit.26
used with success,13 its use may carry an increased risk Additional materials used in the repair of CSF leaks at
of infection. Human-derived dehydrated, lyophi- other sites include the use of gelfoam in epidural
lized, and sterilized dura have been used in neuro- blood patches41 and vicryl mesh in the repair of dural
surgery and other surgical specialties for decades; defects.51 Hydroxyapatite cement has been used
CEREBROSPINAL FLUID LEAKAGE 281
Postoperative Management
POSTOPERATIVE PRESENTATION
Following repair of a CSF leak, the patient should
be monitored postoperatively for continued leakage.
Rarely, an unrecognized dural tear may be diagnosed
only in the postoperative period. Persistent CSF
leakage may present as CSF hypotension syndrome.39
This is characterized by postural headaches, which
may be accompanied by neck tenderness, nausea,
vomiting, photophobia, blurry vision, and sixth
cranial nerve palsy.40 Head imaging should be
performed to rule out an intracranial hemorrhage.
Rhinorrhea, and rarely, orbitorrhea may occur. The
latter may simulate tears if conjunctiva has been
damaged; this has been reported only with trauma,
however.1,7,25 If a CSF leak is suspected, patients
should remain under strict neurological observation.
The surgeon should examine the patient carefully for
leakage at the wound site after lateral decompression,
or rhinorrhea in the case of medial decompression or
DCR surgery. After a CSF leak has been confirmed
with studies, neurosurgery and otolaryngology con-
sults should be obtained as appropriate.
CONSERVATIVE MANAGEMENT
Conservative management includes avoiding
straining activities such as nose-blowing or coughing,
and the use of stool softeners. Elevating the head may
reduce venous pressure resulting in reduced CSF
pressure. Prophylactic antibiotics are often used in
cases of CSF leaks; their use remains controversial,
however. Although intravenous penicillin in adults
and ampicillin in children is thought to decrease the
incidence of meningitis,55 Yilmazlar54 found that
Fig. 10. Repair of CSF leak during orbitotomy for prophylactic antibiotics did not affect rates of
a recurrent adenoid cystic carcinoma involving the orbital meningitis in cases of CSF leakage complicating skull
roof. A: The dural defect (arrow) measured approximately base fractures. Many surgeons avoid antibiotics to
10 mm. B: After the bony defect was enlarged with reduce development of resistant organisms.27 The
ronjeurs for adequate exposure, the dural defect was
repaired with primary closure with 6-0 vicryl, an overlying neurosurgeons at our institutions believe that antibi-
fat graft (f), and further seal with Tisseel (t). otics are not necessary; patients are often treated with
antibiotics by other covering physicians, however.
Acetazolamide reduces the production of CSF and
has been used to treat CSF leaks. Of our cases of CSF
CEREBROSPINAL FLUID LEAKAGE 283
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