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Subarachnoidal pleural fistula after resection of intradural thoracic disc herniation and multimodal treatment with noninvasive positive pressure ventilation (NPPV)

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Abstract

Subarachnoid pleural fistula (SPF) is a type of cerebrospinal fluid (CSF) fistula that can arise as a complication following transthoracic resection of intervertebral disc herniation in the thoracic spine. It is an abnormal communication 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 complications 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 herniation. The postoperative period was complicated by a subarachnoidal pleural fistula. We describe our successful treatment of this using noninvasive positive pressure ventilation and lumbar CSF drainage and review other methods reported in the literature.
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 [1519].
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 [2123]. 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.
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... SPF is an abnormal communication between the subarachnoid and the pleural spaces. This type of fistula has been reported after spine surgery with transthoracic approach [1][2][3][4][5][6][7][8][9][10][11], thoracic surgery [12][13][14][15][16], penetrating trauma or even spontaneously [17]. SPF was found to be a complication of 2.7% of resection of spinal tumors; the incidence after resection of a herniated disc is not determined but is probably lower [2]. ...
... Another interesting proposed strategy was the use of noninvasive positive pressure ventilation (NIPPV) that counters the negative intrapleural pressure and consequently decreases the CSF flow through the fistula (Fig. 4) [24]. Five patients were put on NIPPV and were successfully treated, with no subsequent surgical repair [1,4,5,11]. Treatment duration ranged from 5 [4] to 14 days [5], and patients were most of the time ventilated continuously. ...
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Subarachnoid-pleural fistula (SPF) is a rare complication of spine surgery with a transthoracic approach. The outcome of such an injury is affected by not only the pulmonary status due to the pleural effusion but also the neurological one, secondary to the intracranial hypotension. After reviewing the few published cases of SPF, the journey to diagnosis seams heterogenous and the management plan non-uniform. We report the case of a 48-year old women who underwent a right transthoracic discectomy that was complicated by an SPF. The diagnosis, although suspected perioperatively, was established with the gathering of an abundant post-operative pleural effusion, a subdural hematoma on head Computerized Tomography after drainage and Cerebro-Spinal Fluid markers present in the pleural fluid. The defect was effectively corrected with a radiological procedure. We compare our clinical and paraclinical findings and management plans to those reported in the few other published cases of SPF.
... Successful repair of dorsal fistulas using dural grafts, [10] omental flaps obtained from cadavers, [11] and the application of noninvasive positive pressure ventilation (NPPV) have been described. [12] When the point of the leak is unknown or difficult to treat, an EBP may be performed. [8] Herein, we present the case of a patient who developed an SPF after undergoing transthoracic surgery for a lateral thoracic meningocele. ...
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... A careful monitoring of the cranial symptoms is extremely important and in case of deterioration, application of non-invasive positive pressure ventilation with lumbar CSF drainage has been used successfully in the past. 10 Treatment of the fi stula generally requires surgical revision to seal the breach with glue and fi lling of the dead space with muscle or fat tissue. Postoperatively, non-invasive ventilation with 6 cm H 2 O positive fi nal expiration pressure is recommended, in combination with lumbar drainage. ...
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... The repair can be augmented with a secondary layer of sealants such as fibrin glue, epidural blood patch, Tisseel, DuraSeal (Integra Lifesciences, Plainsboro, NJ), or Onyx (Medtronic-Covidien, Minneapolis, MN) [1,6,8,12]. Alternatively, noninvasive positive pressure mask ventilation combined with chest tube drainage can be used to increase intrathoracic pressure, which decreases CSF flow by decreasing the pressure gradient between the subarachnoid and pleural spaces [5,13]. ...
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A report on 2 cases of subarachnoid pleural fistula (SAPF) treated with noninvasive positive pressure ventilation (NPPV). To highlight the efficacy of NPPV in patients with SAPF. SAPF is a rare but distressing type of cerebrospinal fluid leakage. It is known to be a complication of anterior thoracic spine surgery. The pressure gradient between the subarachnoid space and the pleural cavity maintains the cerebrospinal fluid leakage and precludes the spontaneous closure of the dura. Surgical interventions such as primary repair, patch grafts, muscle flaps, and omental flaps have been advocated. Only limited reports were found with reference to NPPV applied to SAPF. Two patients, a 45-year-old woman and a 39-year-old woman, underwent anterior thoracic spine surgery to treat thoracic myelopathy caused by ossification of the posterior longitudinal ligament. After surgery, they developed SAPF due to perforation of the dura during surgery. Placement of thoracostomy tubes and subarachnoid drains had no effect and an NPPV device was applied. During application of the NPPV device, 14 days in the first patient and 5 days in the second patient, the raised intrapleural pressure obstructed the fluid leakage and successfully treated the fistula. No recurrence of SAPF was observed after removal of the NPPV device and the patients avoided surgical interventions. SAPF is often resistant to conservative therapies and has been treated in an invasive manner. NPPV should be considered as an alternative before such interventions because it is effective, noninvasive, and safe.
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A case of spontaneous subarachnoid pleural fistula following rupture of a thoracic meningocele into the pleural cavity is described in this article. The patient had symptoms of low-pressure headache and difficulty in breathing. The fistulous opening was closed near the foramina by rotating a vascularized muscle flap. After showing initial improvement the patient had a recurrence of symptoms after 6 weeks, with a small leak at the closure site. A lumbar thecoperitoneal shunt led to permanent cure. In this article we discuss the course of the disease, the symptoms, the diagnostic methods, and the various treatment modalities for subarachnoid pleural fistula.