Content uploaded by Bronek Boszczyk
Author content
All content in this area was uploaded by Bronek Boszczyk on Aug 25, 2015
Content may be subject to copyright.
GRAND ROUNDS
Subarachnoidal pleural fistula after resection of intradural
thoracic disc herniation and multimodal treatment
with noninvasive positive pressure ventilation (NPPV)
Holger R. Schlag
1
•Samiul Muquit
1
•Tanyo B. Hristov
2
•Guiseppe Morassi
1
•
Bronek Maximilian Boszczyk
1
•Masood Shafafy
1
Received: 2 May 2015 / Revised: 12 July 2015 / Accepted: 14 July 2015
ÓSpringer-Verlag Berlin Heidelberg 2015
Abstract
Subarachnoid pleural fistula (SPF) is a type of cere-
brospinal fluid (CSF) fistula that can arise as a complica-
tion following transthoracic resection of intervertebral disc
herniation in the thoracic spine. It is an abnormal com-
munication between the subarachnoid and pleural space.
Negative intrapleural pressure promotes CSF leak due to a
suction effect into the pleural cavity, with little chance of
spontaneous closure. Due to the risk of severe complica-
tions with CSF leak into the thoracic cavity, early diagnosis
and treatment are mandatory. However, management can
be challenging. We report a case of a 72-year-old woman
who underwent anterior thoracic surgery to treat thoracic
myelopathy caused by an ossified intradural disc hernia-
tion. The postoperative period was complicated by a sub-
arachnoidal pleural fistula. We describe our successful
treatment of this using noninvasive positive pressure ven-
tilation and lumbar CSF drainage and review other meth-
ods reported in the literature.
Keywords Subarachnoidal pleural fistula Intradural disc
herniation Thoracic spine
Case report
A 72-year-old woman, with no significant past medical
history, presented with a 1 year history of thoracic back
pain and a 2 week history of progressive lower extremity
weakness. Neurological examination revealed MRC grade
3/5 power including and distal to the L2 myotome. She had
reduced pin-prick sensation below L1 bilaterally. Reflexes
were brisk in the lower limbs and both planters were
upgoing. Neurological examination of the upper limbs was
normal. Magnetic resonance imaging (Fig. 1) and a CT
scan (Fig. 2) demonstrated a central calcified disc hernia-
tion at the level T9–T10 level causing spinal cord com-
pression with T2 signal change in the spinal cord. The
calcified disc prolapse occupied more than 60 % of the
cross-sectional area of the spinal canal.
We performed transthoracic discectomy with continuous
intraoperative neurophysiologic monitoring (SSEPs and
MEPs). The patient was placed in a right lateral position. A
left-sided minimally invasive lateral transthoracic
transpleural approach between the 9th and 10th ribs was
used. After the retractor system (MaXcess, NuVasive, Inc.,
San Diego, CA, USA) was introduced, the pleura was
&Holger R. Schlag
h.schlag@gmx.net
1
Centre of Spinal Studies and Surgery, Queens Medical
Centre, Campus of Nottingham University Hospitals NHS
Trust, West Block, D Floor, Derby Road,
Nottingham NG7 2UH, UK
2
Department of Neurosurgery, Cologne-Merheim Medical
Center, University of Witten-Herdecke, Campus Cologne-
Merheim, Witten, Germany
123
Eur Spine J
DOI 10.1007/s00586-015-4137-1
incised and dissected. The left 10th rib head was removed
to allow visualisation of the posterolateral aspect of the
disc and the vertebral body. Subsequently, we performed a
wedge-shaped osteotomy of the vertebral body and cranial
aspect of the T10 pedicle to allow exposure of the dura and
calcified disc prolapse. Under the microscope we began
careful dissection of the calcified disc. It became evident at
this stage that the herniated disc prolapse was firmly
adherent to the dura and had a transdural component,
necessitating resection of the surrounding dura to allow
safe and complete removal of the disc prolapse whilst
protecting the spinal cord. The dural defect was sealed with
three layers of dural substitute (TachoSil, Baxter), fibrin
glue (Tisseel, Baxter) and a fourth layer of gelatine sponge
(Spongistan, Johnson & Johnson). After the dural repair
was complete, no CSF leakage was observed with valsalva
manoeuver. The pleura was sutured to provide an addi-
tional layer of coverage. A chest drain with water seal
chamber and without suction was placed prior to the clo-
sure of the thoracotomy wound.
Following surgery, the patient was kept sedated and
ventilated on the intensive care unit for a period of 24 h.
The ventilator was set on Bilevel positive airway pressure
(BiPAP)—Assisted Spontaneous Breathing (ASB) with
FiO
2
of 0.4, Positive End-Expiratory Pressure (PEEP) set
to 6 cmH
2
O and ASB peak of 16 cmH
2
O. Sedation was
stopped and the endotracheal tube was removed on the
second day after surgery. Examination of the chest and
chest radiograph at this stage showed lung re-expansion
and no evidence of pleural effusion.
Fluid output from the chest drain was moderate during
the first 48 h following surgery, measuring a total of
250 ml. Over the subsequent 24 h only a further 20 ml was
produced. The patient was stable and comfortable at this
stage.
On the fourth postoperative day, however, the patient
developed chest pain, dyspnoea and headache. The chest
drain output increased to a total of 700 ml of clear fluid
over the next 24 h and chest radiograph revealed bilateral
pleural effusion (Fig. 3). The fluid drained from the chest
Fig. 1 Preoperative MRI. T2
sagittal (a) and axial (b) images
showing a large thoracic disc
herniation with deformation of
the spinal cord
Fig. 2 Preoperative sagittal
(a) and axial (b) spinal CT scan
showing a calcified disc
herniation at T9–10 level
Eur Spine J
123
drain tested positive for b2-transferrin (a sensitive marker
for CSF). These findings led to the diagnosis of a sub-
arachnoidal pleural fistula.
To manage the CSF fistula, we inserted a lumbar CSF
drain and aimed to drain 10 ml/h. The patient was kept flat
in bed. Despite these measures, the chest drain continued to
drain large amounts of CSF and the patient’s respiratory
function deteriorated, requiring increasing levels of
inspired oxygen. Due to the cerebrospinal fluid depletion,
the patient had worsening headache with nausea and
dizziness. Noninvasive positive pressure ventilation
(NPPV) with a face-mask was applied to counteract the
inspiratory negative intrapleural pressure, which was pro-
moting flow through the CSF fistula. In the following days,
the symptoms resolved and the fluid output through the
chest tube reduced. We were subsequently able to clamp
the chest drain for a couple of days. The patient’s condition
remained stable and the chest radiograph showed a sub-
stantial reduction in the pleural effusion allowing removal
of chest drain and discontinuation of NPPV. The lumbar
drain was removed after a two more days. The follow-up
X-radiographs of the chest revealed no further pleural
collection and the patient recovered well.
Discussion
Subarachnoid pleural fistula (SPF) is an abnormal com-
munication between the subarachnoid and pleural space,
which occurs in the presence of coexisting defects in the
arachnoid and dura mater as well as the parietal pleura [1].
SPF can arise from blunt or penetrating trauma [2], as a
complication of thoracic and spinal surgery [3], or spon-
taneously [4]. Pleural pressure is generally below atmo-
spheric pressure (range between -2 and -8 cmH
2
O).
During inspiration the intrapleural pressure becomes most
negative. Given the positive pressure within the sub-
arachnoid space CSF flows along the pressure gradient in
the presence of a SPF and accumulates within the pleural
space [5]. As 500 ml of CSF is produced per day in human
adults there is a continuous flow of CSF preventing a
spontaneous closure of the fistula. The presence of a chest
tube with water seal chamber promotes flow through the
fistula by removing CSF collecting in thoracic cavity [1].
The pleura is able to absorb CSF but its ability to do so
can be overwhelmed, leading to a pleural collection. An
example of this can be seen with ventriculopleural shunts, a
long established treatment of hydrocephalus. The incidence
of pleural effusion has been reported to range from 2–20 %
in these cases [6]. Conversely small CSF leaks may go
unnoticed. Fluid absorption by the pleural depends on
pleural lymph flow in addition to hydraulic and osmotic
forces. It is estimated that maximum lymph flow can
potentially increase to 700 ml per day [7]. However, as
non-clearance is determined by several factors, this rate of
CSF clearance may not be possible.
In these cases clinical suspicion should be raised when
symptoms of chest pain, dyspnoea, tachypnoea and cough
appear. Chest radiographs usually confirm the presence of
large pleural effusion. If the diagnosis remains uncertain
additional investigation with ultrasound or computer
tomography (CT) is recommended, as they are more sen-
sitive at detecting small effusions [8].
Respiratory compromise due to a substantial pleural
effusion may require thoracentesis or chest tube insertion;
which allows lung re-expansion. To confirm SPF, pleural
fluid should be tested of the presence of b2-transferrin [9],
a biochemical marker that provides a high sensitivity
(94–100 %) and specificity (98–100 %) for confirming
presence of CSF [10]. Small CSF leaks can be difficult to
identify, in which case CT myelography and radionuclide
cisternography are indicated. Both have been reported as
suitable and sensitive investigations to detect a SPF. Of
these, CT myelography provides a better anatomical
description whereas radionuclide cisternography has a
higher sensitivity in detecting small SPF [1].
Drainage of large volumes of CSF through a SPF can
lead to intracranial hypotension [11]; the symptoms of
which are headache, nausea, vomiting, neck pain, changes
in hearing and dizziness. In addition, there is a risk of
subdural hematoma and cerebellar haemorrhage [12].
Disc herniation in the thoracic region comprises only
3 % of all disc herniation in the spine. Intradural disc
herniations (IDH) are rare accounting for 0.26–0.3 % of all
herniated discs [13]. Thoracic disc herniations are
Fig. 3 Chest X-ray AP (antero-posterior) in supine position shows
bilateral pleural effusion (chest drain in situ on the left side)
Eur Spine J
123
frequently calcified [3] which in itself raises the possibility
of intradural encroachment. More than 60 % of calcified
thoracic discs are found intraoperatively to have either an
intradural component or are strongly adherent to (incor-
porated into) the dura [14]. Unfortunately there is no reli-
able diagnostic method to confirm intradural encroachment
of a thoracic disc prior surgery. Magnetic resonance (MRI)
with Gadolinium and CT can only give an indication as to
whether this is likely; unless, as is very rarely encountered,
an extradural CSF collection is visible [11], in which case
one can be certain of a dural defect. It is vital that in all
cases of calcified thoracic disc, one makes provisions for
managing a dural defect and CSF leak in the pre-operative
planning prior to embarking on surgery.
In the case described in our report we chose a
transthoracic approach due to the central location of the
disc herniation, its calcified nature and the patient’s
symptoms of myelopathy. Several authors have reported on
the advantages of an anterolateral transthoracic approach,
particularly for centrally located calcified discs [15–19].
Other approaches have been reported, including costo-
transversectomy and posterior; however, we suggest that
anterior approaches provide the best access to resect almost
all thoracic disc herniations and also provide the best
exposure for repair of a dural defect.
Cerebrospinal fluid leakage during transthoracic disc
surgery has been reported in up to 15 % of the cases [3], and
is either due to intradural disc herniation or iatrogenic causes.
When identified intraoperatively, every effort should be
made to repair the leak so as to prevent serious pulmonary
and neurological complications in the postoperative period.
The dural defect may be large and a watertight primary
closure may not be possible. Alternative means of closure
have been reported including multilayer techniques with the
use of dural patches, fibrin glue or muscle patches. Some
authors recommended the routine intraoperative placement
of a lumbar drain whenever a dural defect is encountered.
However, this is not necessary if a good closure with
watertight seal is achieved during surgery. A lumbar drain
can be subsequently inserted if a significant SPF is diagnosed
post-operatively. If insertion is indicated we agree with the
recommendation that the lumbar drain is kept in for several
days after the chest drain is removed [20].
The use of prophylactic antibiotics is controversial, but
as with other CSF leaks the body of evidence goes against
prophylactic use to avoid infections with resistant organ-
isms [1].
Positive-pressure ventilation (PPV) with a bilevel posi-
tive airway pressure (BiPAP) has been suggested as a
beneficial intervention in managing SPF [21–23]. It is
suggested that this counteracts the negative pleural pres-
sure and promotes a spontaneous closure of the dura [1].
Kurata et al. treated two patients with SPF following
anterior thoracic spine surgery and showed a successful
closure of the CSF leakage after 14 days of NPPV in one
patient and after 5 days in the other [23]. Yoshor et al. also
report a case where use of NPPV was necessary for 5 days
[21]. Our experience was similar, as 6 days of NPPV was
necessary (in addition to lumbar drainage) for the CSF leak
to close. Although only a handful of reports have been
published, the most effective way of conservatively treat-
ing a SPF appears to be with nonivasive positive pressure
ventilation (NPPV) plus lumbar CSF drainage.
If conservative management fails after 7–10 days, if
severe symptoms persist or there is clinical deterioration,
surgical treatment must be considered [1]. The target of
revision surgery is the identification of the leakage and
watertight closure.
Compliance with ethical standards
Conflict of interest The authors report no conflict of interest con-
cerning the materials or methods used in this study or the findings
specified in this paper.
Patient consent statement The patients’ next of kin have consented
to the submission of the case report for submission to the journal.
References
1. Hentschel SJ, Rhines LD, Wong FC et al (2004) Subarachnoid-
pleural fistula after resection of thoracic tumors. J Neurosurg 100
(4 Suppl Spine):332–336
2. Lloyd C, Sahn SA (2002) Subarachnoid pleural fistula due to
penetrating trauma: case report and review of the literature. Chest
122(6):2252–2256
3. McCormick WE, Will SF, Benzel EC (2000) Surgery for thoracic
disc disease. Complication avoidance: overview and manage-
ment. Neurosurg Focus 9(4):e13
4. Kumar V, Bundela YS, Gupta V et al (2010) Spontaneous sub-
arachnoid pleural fistula: a rare complication of lateral thoracic
meningocele. Neurol India 58(3):466–467
5. Shamji MF, Sundaresan S, Da Silva V et al (2011) Subarachnoid-
pleural fistula: applied anatomy of the thoracic spinal nerve root.
ISRN Surg 2011:168959
6. Kupeli E, Yilmaz C, Akcay S (2010) Pleural effusion following
ventriculopleural shunt: case reports and review of the literature.
Ann Thorac Med 5(3):166–170
7. Miserocchi G (1997) Physiology and pathophysiology of pleural
fluid turnover. Eur Respir J 10(1):219–225
8. Moy MP, Levsky JM, Berko NS et al (2013) A new, simple
method for estimating pleural effusion size on CT scans. Chest
143(4):1054–1059
9. Deseyne S, Vanhouteghem K, Hallaert G et al (2015)
Subarachnoidal-pleural fistula (SAPF) as an unusual cause of
persistent pleural effusion. Beta-trace protein as a marker for
SAPF. Case report and review of the literature. Acta Clin Belg
70(1):53–57
10. Huggins JT, Sahn SA (2003) Duro-pleural fistula diagnosed by
beta2-transferrin. Respiration 70(4):423–425
11. Winter SC, Maartens NF, Anslow P et al (2002) Spontaneous
intracranial hypotension due to thoracic disc herniation. Case
report. J Neurosurg 96(3 Suppl):343–345
Eur Spine J
123
12. Paldino M, Mogilner AY, Tenner MS (2003) Intracranial
hypotension syndrome: a comprehensive review. Neurosurg
Focus 15(6):ECP2
13. Epstein NE, Syrquin MS, Epstein JA et al (1990) Intradural disc
herniations in the cervical, thoracic, and lumbar spine: report of
three cases and review of the literature. J Spinal Disord
3(4):396–403
14. Gille O, Soderlund C, Razafimahandri HJ et al (2006) Analysis of
hard thoracic herniated discs: review of 18 cases operated by
thoracoscopy. Eur Spine J 15(5):537–542
15. Nacar OA, Ulu MO, Pekmezci M et al (2013) Surgical treatment
of thoracic disc disease via minimally invasive lateral transtho-
racic trans/retropleural approach: analysis of 33 patients. Neu-
rosurg Rev 36(3):455–465
16. Vollmer DG, Simmons NE (2000) Transthoracic approaches to
thoracic disc herniations. Neurosurg Focus 9(4):e8
17. Ayhan S, Nelson C, Gok B et al (2010) Transthoracic surgical
treatment for centrally located thoracic disc herniations present-
ing with myelopathy: a 5-year institutional experience. J Spinal
Disord Tech 23(2):79–88
18. Quraishi NA, Khurana A, Tsegaye MM et al (2014) Calcified
giant thoracic disc herniations: considerations and treatment
strategies. Eur Spine J 23(Suppl 1):S76–S83
19. Russo A, Balamurali G, Nowicki R et al (2012) Anterior thoracic
foraminotomy through mini-thoracotomy for the treatment of
giant thoracic disc herniations. Eur Spine J 21(Suppl 2):S212–
S220
20. Dickman CA, Rosenthal D, Regan JJ (1999) Reoperation for
herniated thoracic discs. J Neurosurg 91(2 Suppl):157–162
21. Yoshor D, Gentry JB, Lemaire SA et al (2001) Subarachnoid-
pleural fistula treated with noninvasive positive-pressure venti-
lation. Case report. J Neurosurg 94(2 Suppl):319–322
22. Valla FV (2007) Subarachnoid-pleural fistula in an infant treated
with mechanical positive-pressure ventilation. Pediatr Crit Care
Med 8(4):386–388
23. Kurata Y, Yoshimoto M, Takebayashi T et al (2010) Subarach-
noid-pleural fistula treated with noninvasive positive pressure
ventilation: a two-case report and literature review. Spine (Phila
Pa 1976) 35 (18):E908–E911
Eur Spine J
123