Original ResearchSinonasal Disorders
Otolaryngology
Increased Intracranial Pressure in
Spontaneous CSF Leak Patients Is
Not Associated with Papilledema
Geoffrey Aaron, MD1, Jennifer Doyle, MD2,
Michael S. Vaphiades2, Kristen O. Riley, MD3, and
Bradford A. Woodworth, MD1
Sponsorships or competing interests that may be relevant to content are dis-
closed at the end of this article.
Abstract
Objective. Spontaneous cerebrospinal fluid (CSF) leaks typi-
cally present in patients with undiagnosed idiopathic intra-
cranial hypertension (IIH) secondary to pressure erosion of
the skull base. Despite elevated intracranial pressure (ICP)
on lumbar puncture or ventriculostomy, patients with spon-
taneous CSF leaks rarely complain of visual disturbances.
The objective of this study is to correlate the presence of
preoperative papilledema with opening ICP in patients
undergoing endoscopic repair of spontaneous CSF leaks.
Study Design. Prospective study.
Setting. Tertiary hospital.
Methods. Prospective evaluation of patients with sponta-
neous CSF leaks was performed over a 1-year period
(December 2012 to December 2013). Fundoscopic exami-
nation for papilledema was completed preoperatively and
CSF pressure measured by lumbar puncture or ventriculost-
omy intraoperatively. Data regarding demographics, nature
of presentation, and body mass index (BMI) were also
recorded and compared to a control cohort of IIH patients
with papilledema.
Results. Sixteen patients (average age 52) were evaluated.
Obesity was present in 94% of individuals (average BMI =
43, range, 27-65). Papilledema was absent preoperatively in
all subjects. Opening pressures via lumbar puncture/ventri-
culostomy were 27.4 6 7.7 cmH20. Following 6 hours of
clamping, measurements significantly increased to 36 6 9.6
cmH20 (P \ .001). IIH controls (average age 33, average
BMI = 36, range, 21-52) exhibited average ICP (36.2 6 11.7)
identical to postclamp measurements in the spontaneous
CSF leak cohort.
Conclusions. Subjects with spontaneous CSF leaks had post-
clamping average ICP identical to controls with IIH and
papilledema. Such evidence suggests that a CSF leak in this
patient population provides sufficient pressure diversion to
avoid the development of papilledema.
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Head and Neck Surgery
2014, Vol. 151(6) 10611066
American Academy of
OtolaryngologyHead and Neck
Surgery Foundation 2014
Reprints and permission:
sagepub.com/journalsPermissions.nav
DOI: 10.1177/0194599814551122
http://otojournal.org
Keywords
CSF leak, cerebrospinal fluid, idiopathic intracranial hyper-
tension, benign intracranial hypertension, pseudotumor cere-
bri, spontaneous CSF leak, acetazolamide, shunt, papilledema.
Received June 3, 2014; revised July 15, 2014; accepted August 22,
2014.
Introduction
Idiopathic intracranial hypertension (IIH) is a disease com-
prised of elevated intracranial pressures without an identifi-
able cause.1 An update of the Modified Dandy Criteria in
2002 defined IIH as: (1) symptoms of generalized intracra-
nial hypertension or papilledema; (2) signs of generalized
intracranial hypertension or papilledema; (3) documented
elevated intracranial pressures in lateral decubitus position;
(4) normal cerebrospinal fluid (CSF) composition; (5) no
evidence of hydrocephalus, mass, structural, or vascular
lesion on magnetic resonance imaging (MRI) or computed
tomography (CT); and (6) no other cause of intracranial
hypertension identified.2 Patients with IIH tend to be obese
females within the childbearing age span.3,4
Papilledema is one of the most common findings seen on
physical examination in individuals diagnosed with IIH. In
many cases, papilledema is the first indication of IIH in the
1
Departments of Surgery/Division of Otolaryngology, University of Alabama
at Birmingham, Birmingham, Alabama, USA
2
Department of Opthalmology, University of Alabama at Birmingham,
Birmingham, Alabama, USA
3
Department of Neurosurgery, University of Alabama at Birmingham,
Birmingham, Alabama, USA
This article was presented at the 2014 AAO-HNSF Annual Meeting & OTO
EXPO; September 21-24, 2014; Orlando, Florida.
Corresponding Author:
Bradford A. Woodworth, MD, OtolaryngologyHead and Neck Surgery,
University of Alabama at Birmingham, BDB 563; 1720 2nd Ave S,
Birmingham, AL 35294, USA.
Email: bwoodwo@hotmail.com
1062
patient and is either diagnosed incidentally on routine
ophthalmologic examination or discovered during the
workup of a visual disturbance. In a recent study, papille-
dema was found in 94.3% of patients with IIH at 1 center.5
Importantly, it is estimated that anywhere between 10% and
25% of patients with papilledema have permanent visual
loss despite therapeutic interventions. 6,7
In the past 10 years, there has been an increased interest
in the association of IIH with spontaneous CSF leaks.
Studies have established a highly suggestive correlation
between the 2 entities through clinical symptoms, radio-
graphic signs, and direct intracranial pressure measure-
ments. A recent literature review published in 2013 found
23 studies that correlated spontaneous CSF leaks with the
diagnosis of IIH.8 Most recently, Chaaban et al9 revealed
that patients with spontaneous CSF leaks undergoing repair
had an opening intrathecal/intracranial pressure of 24.3 cm
H2O that significantly increased to 32.3 cm H2O once the
CSF leak had been sealed.9 Imaging studies often can direct
the source of the CSF leak along with evidence of IIH. MRI
evidence includes partially or totally empty sella, flattening
of the posterior globes, stenosis of the transverse sinus, and
optic nerve distension along with encephaloceles that are
associated with CSF leaks, though not specific to IIH.10,11
Evidence of changes on CT of patients with IIH with spon-
taneous CSF leaks includes erosions and defects of the skull
base with arachnoid pits and enlarged foramen ovales.11,12
Despite the known relationships between IIH and papille-
dema and IIH and spontaneous CSF leaks, there are no
studies examining if there is any correlation between spon-
taneous CSF leaks and papilledema. Patients with sponta-
neous CSF leaks infrequently complain of any visual
changes and are thus rarely evaluated by an ophthalmolo-
gist. The objective of the current study is to correlate the
presence of preoperative papilledema with opening intracra-
nial pressure (ICP) in patients undergoing endoscopic repair
of spontaneous CSF leaks and compare to a control group
of patients with IIH and papilledema.
Materials and Methods
Subjects
Prospective evaluation and data collection of subjects was
approved by the institutional review board at the University
of Alabama at Birmingham and Callahan Eye Foundation.
All patients were treated by a single otolaryngologist
(BAW) for CSF leaks of spontaneous etiology and enrolled
during a 1-year period (December 2012 to December 2013).
CSF leaks were considered spontaneous when there was no
previous history of skull base fracture/trauma, tumor invol-
vement, or other obvious etiology. Preoperative evaluation
of all patients consisted of a thorough history and physical
examination (with nasal endoscopy), including inquiries
about previous history of head trauma, prior sinus or neuro-
logical surgery, congenital abnormalities, prior episodes of
meningitis (or other intracranial events), and obesity. Body
mass index (BMI) calculations were performed in all
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OtolaryngologyHead and Neck Surgery 151(6)
patients. Radiographic imaging assessment included image-
guided surgical navigation CT scans in all cases and MRI
scans. Data regarding demographics, nature of presentation,
location and size of defect, surgical approach, reconstructive
technique, and management of ICP were collected.
The control group was derived from 16 consecutive
patients diagnosed with IIH who had papilledema on fundo-
scopic exam and lumbar puncture to obtain opening pres-
sure measurements during the clinical workup for the
disorder. Demographic data including sex, age, and BMI
were also collected for comparison.
Ophthalmologic Examination
All patients enrolled in the study took part in a complete
ophthalmologic examination at the University of Alabama
at Birminghams Callahan Eye Center. Participants were
examined by a single neuroophthalmologist (MSV) who
specializes in the treatment of IIH. Focus was made primar-
ily on the fundoscopic examination to determine if there
was any evidence of papilledema in both the spontaneous
CSF leak group and the control group.
Surgical Technique
The technique for endoscopic management varied depend-
ing on the site and size of the defect, presentation, and other
factors but generally outlines those previously described.13-18
Lumbar drains (LDs) or ventriculostomies were used in all
surgical patients.19 Fluorescein was also utilized according
to previously published protocols to localize defects, iden-
tify multiple CSF leaks, and inspect for a watertight clo-
sure at the conclusion of the case.20-22 A mixture of 0.1
mL of 10% fluorescein diluted in 10 mL of the patients
CSF is slowly injected over 10 to 15 minutes. Surgical
exposure is characterized according to transethmoid tech-
nique (sphenoethmoidectomy) with additional exposures
(eg, transpterygoid, Draf IIB frontal sinusotomy) as neces-
sary (Figures 1, 2). The transpterygoid approach is per-
formed as previously described for all lateral sphenoid
recess CSF leaks.23
Intracranial Pressure Measurements
Lumbar punctures were performed in the lateral decubitus
position with the spine leveled using a manometer to deter-
mine opening pressures. The control group had a lumbar
puncture performed by radiology, while the surgical cohort
underwent lumbar tap or ventriculostomy under anesthesia
just prior to the procedure. Surgical patients also had LDs
or ventriculostomies placed for postoperative pressure moni-
toring. Although the use of LDs in CSF leak repair has
recently been labeled controversial,17,21,24,25 we utilize
drains when we feel the benefits far outweigh the risks.
Because spontaneous CSF leaks often have multiple defects,
the administration of intrathecal fluorescein is useful for
identifying other leaking sites that may not be readily appar-
ent on preoperative imaging. LDs (or ventriculostomies) are
managed according to previously described protocols with
slight alterations.13 In general, LDs are opened at the time
Aaron et al 1063

Figure 1. Triplanar CT image guidance and endoscopic view of a spontaneous encephalocele and cerebrospinal fluid leak from the right
planum sphenoidale.

Figure 2. The encephalocele is removed with bipolar coblation and the surrounding mucosa stripped (A) The defect is measured and (B)
repaired in multiple layers including a bone graft (C) to provide support against elevated intracranial pressure.

of graft placement, and the height of the collection chamber
is adjusted to maintain drainage at approximately 10 cc/hr.
The drain is clamped on the morning of postoperative day
2, and at least 6 hours is allowed to help equilibrate the
patients CSF volume. A pressure transducer or manometer
is connected to the lumber drain with the patient in the lat-
eral decubitus position zeroed at the spinal column. Normal
CSF pressure is between 5 and 15 cm H2O in this position.
If pressure is elevated, oral acetazolamide (500 mg) is admi-
nistered and read again at 4 to 6 hours to assess the effect
of the medication. Acetazolamide is a carbonic anhydrase
inhibitor that decreases CSF production. In patients with
significantly elevated ICP (generally .35 cm H2O at base-
line), multiple defects, or an inadequate response to medical
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therapy with carbonic anhydrase inhibitors (generally \10
cm H2O decrease), permanent ventriculoperitoneal (VP)
shunting is recommended. If patients refuse the VP shunt,
they are kept on acetazolamide, and electrolytes are checked
periodically to ensure no life-threatening abnormalities.
Data were analyzed for statistical significance with paired
and unpaired t tests.
Postoperative Management
Patients are instructed on movement techniques to avoid
breath holding and Valsalva maneuvers. An antistaphylo-
coccal antibiotic is prescribed until the packing is removed
at the first postoperative visit 9 to 13 days postoperatively.
A stool softener is prescribed for every patient, and light
1064 OtolaryngologyHead and Neck Surgery 151(6)

Table 1. Spontaneous Cerebrospinal Fluid (CAF) Leak Patients.

Age Sex BMI OP CP Defect Sites Size Shunt Papilledema

55 F 30 28 32 L ON sheath 333 mm Yes No

L planum 232 mm
47 F 47 45 45 R AE, PT 535 mm No
61 F 54 28 42 L AE 12 3 10 mm Yes No
67 F 37 26 36 R planum 434 mm No
54 F 47 29 39 R planum 636 mm No
51 F 34 15 23 R LRS 535 mm No
62 F 41 18 35 R crib 334 mm No
41 F 40 35 38 L LRS 333 mm No
44 M 39 34 39 R LRS 8 3 10 mm No
49 F 41 33 54 R AE 232 mm No
R PT 737 mm
50 F 62 22 36 L LRS 838 mm No
49 F 34 19 32 R planum 638 mm Yes No
54 M 35 22 22 R planum 738 mm No
67 F 27 23 17 R crib 737 mm No
46 F 62 35 48 L planum 10 3 8 mm Yes No
33 F 65 27 38 R crib and AE 839 mm No
Abbreviations: AE, anterior ethmoid roof; BMI, body mass index; CP, clamped pressure; crib, cribriform plate; LRS, lateral recess of the sphenoid sinus; ON,
optic nerve; OP, opening pressure; PT, posterior table of the frontal sinus.

activity is continued for 6 weeks after surgery. Patients are pressures were significantly higher than ICP measured in
seen anywhere from 1 to 4 weeks after the first visit. the spontaneous CSF leak cohort (36.2 6 11.7 cm H2O;
P \ .01). However, there was no statistical difference
Results between the 6- hour clamped CSF pressure of the sponta-
neous CSF leak group and the IIH patients with papille-
Over a 1-year period, 16 patients met the criteria of sponta-
dema, which were nearly identical.
neous CSF leaks and were included in the study. Patient
demographics and clinical data are presented in Table 1.
Fourteen (87.5%) of the patients were female (average
age = 52 years old, range, 33-67). Average BMI for patients
with a spontaneous CSF leak was 43 (range, 27-65) with
only 1 patient (BMI = 27) not meeting the definition of obe- Table 2. Idiopathic Intracranial Hypertension Patients.
sity (BMI .30). All patients in the surgical cohort did not Age Sex BMI OP Papilledema
have papilledema during the preoperative assessment. Two
patients had multiple leaks identified during the surgery. 25 F 52 38 Yes
Average defect size was 5.9 3 6.2 mm (length vs width). 32 F 30 49 Yes
The size and location of the individual CSF leaks as well as 23 F 50 60 Yes
specific reconstruction techniques are provided in Table 2. 28 F 21 59 Yes
Two patients required a ventriculostomy when lumbar punc- 25 F 35 35 Yes
tures were unable to be performed secondary to the depth of 30 F 32 21 Yes
adipose tissue in the lower back. Opening pressures were 47 F 35 37 Yes
27.4 6 7.7 cm H2O. The 6-hour postclamp average CSF 29 F 38 34 Yes
pressure taken on postoperative day 2 was 36.0 6 9.6 cm 26 F 41 32 Yes
H2O, which was a statistically significant increase (P \ 34 F 49 22 Yes
.0001). Eleven patients were placed on long-term acetazola- 46 F 22 44 Yes
mide, while 4 individuals received a VP shunt to control 30 F 28 33 Yes
CSF pressure. One patient died from a massive pulmonary 23 F 34 31 Yes
embolism on postoperative day 2. 29 F 35 32 Yes
The control cohort IIH patients with papilledema 43 F 45 25 Yes
included 16 participants. All subjects (100%) were females 54 F 30 27 Yes
with an average age of 33 years old (range, 23-54). Average
Abbreviations: BMI, body mass index; OP, opening pressure.
BMI for this group was 36.0 (range, 21-52). Opening
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Aaron et al
Discussion
IIH is a disease entity that is closely associated to obesity
and has increased in prevalence in our society as the obesity
epidemic continues to rise in Western culture. The incidence
of IIH has been estimated at around 0.9 per 100,000 people
in the United States but increases to 20 per 100,000 people
among the most common demographic affected: obese
women in their childbearing years.3 In the current study,
87.5% of the patients with a spontaneous CSF leak and
100% of the patients in the IIH with papilledema were
females with an average age 51.9 and 32.8 years of age,
respectively. The discrepancy in age suggests the sponta-
neous CSF leak cohort had clinically undiagnosed IIH for
many years that eventually resulted in skull base erosion
and development of a CSF leak, while the IIH cohort pre-
sented early on with papilledema. Both groups had a high
percentage of obesity as defined by BMI .30 (CSF leak =
94%, IIH = 75%) with a very high average BMI (CSF leak
= 43.5, IIH = 36.0). It should be noted that the average BMI
of the IIH control group was very similar to previously
reported studies. The cohort of CSF leak patients in the cur-
rent study was primarily skewed by 2 very obese individuals
(BMI .60) regarding spontaneous CSF leak patients.
Regardless, the 2 groups are representative of what has been
reported in the literature regarding IIH and spontaneous
CSF leak patients.3,4,6,9,10,26,27
To our knowledge, this is the first study to assess papille-
dema in a cohort of spontaneous CSF leak subjects. Despite
objective measurements of elevated ICP, patients did not
have papilledema with concurrent CSF leaks. However,
ICPs significantly increased 6 hours after the lumbar drain
was clamped on postoperative day 2 following repair.
Because there was no difference between the 2 groups (CSF
[postclamp ICP] = 36.0 cm H2O vs IIH = 36.1), this
strongly suggests that the subject populations are representa-
tive of the same etiology. While some patients with IIH may
progress to papilledema, others are clinically undiagnosed
and eventually develop a spontaneous CSF leak due to
chronic pressure erosion of the skull base. The evidence pre-
sented here indicates that a CSF leak allows for sufficient
diversion of ICP so that complications such as papilledema
are avoided. Such a shunting effect has been postulated previ-
ously, but the current study provides the first substantiation
of this theory by directly comparing postclamp elevated ICP
to control subjects with IIH and papilledema. Further support
from the literature includes a recent case report regarding 2
spontaneous CSF leak patients who developed papilledema
after endoscopic repair.8 Though both patients did not have
fundscopic exams prior to the surgical repair of their skull
base defects, neither initially presented with any ocular com-
plaints. Importantly, data presented in the current study sup-
port aggressive control of ICP postoperatively to not only
decrease the risk of developing a second CSF leak but also
reduce the potential of developing papilledema.
IIH is most commonly controlled with acetazolamidea
carbonic anhydrase inhibitor that decreases CSF production.
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1065
It has been previously demonstrated that acetazolamide is
an effective and safe medication to lower CSF pressure
in patients who have undergone closure of CSF leaks
associated with intracranial hypertension.28 The majority of
patients in the current study (68.8%) were controlled with
acetazolamide. In cases where ICPs are very elevated, do
not respond to acetazolamide, multiple CSF leaks are pres-
ent, or the CSF leak recurs, VP shunts are the next line of
therapy.9,17 Shunts, whether VP, ventriculoatrial, or lumbo-
peritoneal, work effectively by regulating ICP by diverting
excessive CSF. Although this measure is effective, it comes
with its own set of risks including surgical site infections,
meningitis, low-pressure headaches, shunt revision, failure
of shunt leading to repeat CSF leaks, and even death.29 Four
patients required VP shunts in the current study.
The current investigation was limited by a relatively
small number of subjects and some incongruity between
groupsspecifically, higher BMI and older age in the spon-
taneous CSF leak cohort. Additionally, fundoscopic exam
was not performed on postoperative day 2 when postclamp
pressure was evaluated. However, papilledema is unlikely to
develop in a 6-hour time span after clamping because it
may take days to weeks to develop30 and subjects were
administered ICP-lowering medications or shunted after
measurements. Additionally, long-term follow-up data have
not been collected on these patients regarding recurrence of
the CSF leak or development of papilledema despite use of
acetazolamide. However, patients receive 6-month clinical
follow-up appointments with neuro-ophthalmology to check
for subclinical papilledema in the setting of long-term aceta-
zolamide therapy.
Conclusion
Patients with spontaneous CSF leaks in the setting of sus-
pected IIH did not demonstrate papilledema on fundoscopic
exam. Once the spontaneous CSF leaks were surgically
repaired, patients had postclamping average ICP identical to
controls with IIH and papilledema. Such evidence suggests
that a CSF leak in this patient population provides sufficient
pressure diversion to avoid the development of papilledema.
Author Contributions
Geoffrey Aaron, data compilation, writing manuscript; Jennifer
Doyle, data compilation, critical review of manuscript; Michael S.
Vaphiades, study design, data acquisition, critical review of manu-
script; Kristen O. Riley, study design, data acquisition, critical
review of manuscript; Bradford A. Woodworth, study design,
data acquisition, critical review of manuscript.
Disclosures
Competing interests: Kristen O. Riley, Cook Medicalroyalty
agreement from OR video. Bradford A. Woodworth, Gyrus/
Olympus, Cook Medical, and ArthroCare ENT consultant.
Sponsorships: None.
Funding source: Research to Prevent Blindness, Inc, none besides
monetary support.
1066 OtolaryngologyHead and Neck Surgery 151(6)

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