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Thawani, et al., J Spine Neurosurg 2015, 4:2
http://dx.doi.org/10.4172/2325-9701.1000182
Journal of Spine
& Neurosurgery
Case Report
a SciTechnol journal
Microsurgical Treatment of a
Complex Sacral Perineural Cyst
Using Paraspinous Muscle Flap:
Technical Note
Jayesh P Thawani1*, Hovik John Ashchyan1, John Pierce1, Nikhil
R Nayak1, John F Burke1, Suhail K Kanchwala1 and William C
Welch1
Abstract
Dr. I.M. Tarlov first described perineural (Tarlov) cysts in 1938.
Symptomatic Tarlov cysts are common incidental findings yet
uncommon clinical/pathologic entities. Microsurgical treatment
of Tarlov cysts is even more rare. We report the microsurgical
treatment of a symptomatic complex Tarlov cyst as a case. The
patient presented with sacral pain, bilateral sciatica (S1-type),
urinary retention, rectal pain with intercourse, gluteal heaviness,
and intermittent burning in the S2-4 dermatome. MR imaging
revealed a complex sacral Tarlov cyst with bony erosion. The
patient was treated microsurgically and had a paraspinous muscle
flap advancement procedure performed in conjunction with Plastic
Surgery. The patient experienced a positive clinical outcome.
Keywords: Tarlov cyst; Perineural cyst
Introduction
Perineural or Tarlov cysts were first described in 1938 by Dr. I.M
[1,2]. Tarlov. Tarlov cysts are not an uncommon incidental finding
on MR imaging of the spine with a prevalence estimated between
4.6 and 9% of the general adult population [3], Symptomatic Tarlov
cysts are, however, much more rare. They commonly occur in the
sacral spine, arising near the dorsal root ganglion between layers of
perineurium and endoneurium [4]. Given the challenges of treatment
and few series describing therapy, asymptomatic Tarlov cysts are
justifiably not treated.
Symptomatic patients pose diagnostic and therapeutic challenges.
Typical presentations include low back pain, radicular pain, bowel/
bladder/sexual dysfunction, leg weakness, and sensory dysesthesias
[1], Treatment options that have been described include antiinflammatory medications, percutaneous cyst drainage [5], external
CSF-drainage, percutaneous fibrin glue injection [6], insertion of cystsubarachnoid shunt [1], lumboperitoneal shunt [3], cyst-peritoneal
shunt [7], as well as microsurgical treatment (including descriptions
of resection of cyst neck or wall, cyst imbrication, or cauterization
[3,8-11]. We describe the use of an open sacral laminectomy to open
and imbricate the cyst with the aid of autologous muscle, paraspinal
muscle flap, and placement of a lumbar drain.
*Corresponding author: Jayesh P Thawani, MD, Penn Neurosurgery, University
of Pennsylvania, Pennsylvania Hospital, 235 S. 8th Street, Philadelphia, PA, USA
19106, Tel: (800) 789-7366; E-mail: [email protected]
Received: December 24, 2014 Accepted: February 03, 2015 Published: February
02, 2015
International Publisher of
Science, Technology and Medicine
Case Report
Clinical presentation
A 49-year-old female patient presented to the office with a
history of lumbar spondylosis (underwent L4-S1 pedicle fusion
with interbody cages in 2008), endometriosis, surgery for an ectopic
pregnancy incurred in the 1990s, salpingo-oophorectomy, and
removal of scar tissue for endometriosis, open appendectomy, and
laparoscopic placement of a gastric band for obesity. She presented
preoperatively with several years of worsening sacral pain, bilateral
sciatica (S1-type), sensation of urinary retention, rectal pain with
intercourse, gluteal heaviness, intermittent burning in the S2-4 region.
Her pain improved slightly with medication but mostly with lying flat
on her back. It was aggravated with supine position. MR imaging of
the lumbar and sacral spine demonstrated hardware corresponding
to an operation performed as above, as well as a large, cystic structure
encompassing the sacral cistern from S2-S4. The lesion had signal
characteristics of CSF and was bilateral and associated with bony
erosion (Figure 1).
Surgical intervention: microsurgical cyst imbrication
The patient elected to undergo microsurgical treatment, which
was performed by the senior author. A sacral incision was made and
then a bilateral subperiosteal dissection was performed. An S1-S4
osteoplastic laminotomy was carried out using the Misonix (Misonix
Inc, Farmingdale, NY) bone scalpel device. At the S3 level, an obvious
erosion through the sacrum was visible and the patient was clearly
leaking cerebrospinal fluid through the eroded opening. S1-S4 was
removed as a single group exposing an extremely complex Tarlov
cyst. Figure 2 demonstrates the large opening present upon opening
the cyst into the sacral cistern. During removal of the lamina, some
of the thinned dura of the Tarlov cyst was unroofed and removed.
The intradural nerve roots were then identified. A piece of the cyst
wall was removed and sent off for pathological examination. The cyst
was closed by reapproximating the very thinned dura and imbricating
it to the ventral dura. This was done so as not to encompass the
nerve roots and to reduce the size of the cyst. DuraSeal (Covidien
Inc., Dublin, Ireland) was then used to augment closure. DuraGen
(Integra Lifesciences Inc., Plainsboro, New Jersey) was laid onto
the repair. The lamina was replaced and using C-arm fluoroscopy, a
lumbar drain was placed at about the L2 level. Our colleague in Plastic
Surgery (SK) performed a paraspinous muscle flap advancement. The
Figure 1: T2-weighted MR-imaging demonstrating sacral perineural cyst. The
cyst has eroded into the dorsal vertebral bodies of S2 and S3 resulting in
thinning of the sacral laminae.
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Citation: Thawani JP, Ashchyan HJ, Pierce J, Nayak NR, Burke JF (2015) Microsurgical Treatment of a Complex Sacral Perineural Cyst Using Paraspinous Muscle
Flap: Technical Note. J Spine Neurosurg 4:2
doi:http://dx.doi.org/10.4172/2325-9701.1000182
mechanisms relate to cystic dilatation and nerve root irritation and/
or leakage of cerebrospinal fluid causing relative hypotension [10]
The etiology of Tarlov cysts is largely unknown. Some groups
have suggested that they may be a result of trauma, while others have
suggested that the origin of these cysts is due to genetic predisposition
(as may be the case in patients with collagen disorders or collagen
vascular diseases) [14]. Although the mechanism by which the
cysts develop is disputed, a leading theory is that it has to do with
pulsatile and hydrostatic forces of CSF along with a ball-valve effect
(unobstructed inflow of CSF with restricted outflow) allowing for
continuous dilation of the cyst [3,1].
Histopathology
Figure 2: Intraoperative photograph demonstrating fenestrated cyst following
sacral laminectomy.
patient remained in the intensive care unit for 6 days with lumbar
drainage. On post-operative day 6, the lumbar drain was clamped then
removed without incident. The patient went home on post-operative
day 9 with suprafascial Jackson-Pratt drains. Figure 3 demonstrates
histopathological sections from surgery.
Surgical intervention: paraspinous muscle flap advancement
Lumbosacral wounds may utilize gluteal, paraspinous, or
latissimus muscle flaps to augment closure. In patients with Tarlov
cysts, who have often had prior procedures performed, myofascial
planes and vascular pedicles may not be well-defined. Refer to figure 4
for a schematic representing closure. After releasing the paraspinous
musculature from the vertebral elements and undermining above the
fascia, lateral relaxing incisions may be made just above the muscular
fascia to effectively release the paraspinous muscle from the ilium.
We used 3-0 biosyn suture to approximate the paraspinous muscle
fascia, and pulley-type sutures were used to lower the flap as close as
possible to the sacral defect. This was performed in order to minimize
dead space and the potential for a large pseudomeningocele. During
exposure, care should be taken to minimize injury to the stabilizing
ligaments of the bony ilium, spine, and sacrum. Aggressive lateral
dissection may result in injury to the superior cluneal nerves or the
lateral perforating radicular branches.
Though Tarlov cysts are often assessed using MR imaging, the
diagnosis must be confirmed using histopathology. These lesions may
be confused with other clinical entities, including tumors, lipomas, or
arachnoid cysts [11]. Our group encountered a situation in which a
patient presumed to have a Tarlov cyst but had a nerve sheath tumor.
This altered the proposed operative plan and approach. Pathology
plays a very important role in definitive diagnosis. By definition, a
Tarlov cyst needs the presence of spinal nerve root fibers in the wall
or within the cavity of the cyst [9].
Clinical presentation
Tarlov cysts are a symptomatic in the majority of patients [12].
In cases of symptomatic Tarlov cysts, patients experience nonspecific
symptoms similar to those seen in other lumbosacral pathologies;
they commonly experience pelvic/perineal pain, sensory dysesthesias,
low-back pain, radicular pain, sciatic pain, and bowel/bladder/sexual
dysfunction. Patients may also experience tinnitus, orthostasis and
positional headaches [3].
Treatment
Literature on the treatment of symptomatic Tarlov cysts is rather
sparse, and there does not seem to be consensus on an appropriate
Clinical outcome
The patient made steady improvement over the next several
months. Sensory symptoms at several weeks were reported to be quite
minimal compared to before surgery. One year post operatively, the
patient presented to clinic feeling extremely well. The patient had
some sensory symptoms (numbness, infrequent pain) in the left S1
distribution. The patient no longer reported general sacral/pelvic
pain, urinary retention, gluteal heaviness, or sensory dysesthesias of
the low sacral dermatomes.
Discussion
Tarlov cysts are CSF-filled saccular lesions that form in the space
between the perineurium and endoneurium of the nerve root sheaths
of the root ganglion [3]. As previously mentioned, they are one of the
most common incidental findings on MR imaging of the lumbosacral
spine along with fibrolipomas and hemangiomas [12]. It is estimated
that only about 1% of these cysts become symptomatic [13]. Proposed
Volume 4 • Issue 2 • 1000182
Figure 3: Histopathological slides from surgery. A) H&E stain demonstrating
hyalinized paucicellular connective tissue, 100X. B) S-100 stain demonstrating
neural elements within the cyst wall, 100X.
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Citation: Thawani JP, Ashchyan HJ, Pierce J, Nayak NR, Burke JF (2015) Microsurgical Treatment of a Complex Sacral Perineural Cyst Using Paraspinous Muscle
Flap: Technical Note. J Spine Neurosurg 4:2
doi:http://dx.doi.org/10.4172/2325-9701.1000182
Conclusion
Although Tarlov cysts were described close to a century ago, there
is still no consensus on their origin or treatment. Most are benign,
but about 1% are symptomatic and can be managed effectively with
surgery. There are a few studies that have been conducted on the
surgical management of Tarlov cysts and much of this literature
shows positive outcomes [3,15,16]. Here we described a case of a
patient with a symptomatic Tarlov cyst that was treated successfully
with a multi-disciplinary approach incorporating a paraspinous
muscle flap. The long-term outcome for this patient was positive.
References
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Figure 4: Schematic representation of paraspinous muscle flap closure, axial
view. A: The muscular layer is dissected along the posterior vertebral elements
(arrows) and dermal layers are undermined. B: Following fascial detachment
laterally, the muscle flap and overlying dermal/epidermal layers can be reapproximated. C: The flap is extended and the layers are closed.
mode of treatment. A study done by Hendersen et al. recommends
a surgical approach, if the cyst size is ≥ 1.5 cm and associated with
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with microsurgical excision along with duraplasty or placation of
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patients with motor deficits and 6 patients with bladder dysfunction
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that are managed with surgical therapy. The use of a paraspinous
muscle advancement flap may serve to further augment healing
and closure of the opened and imbricated cyst. We suggest that it
may serve to minimize leakage of CSF thus minimizing the size of a
pseudomeningocele.
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Author Affiliation Top
Department of Neurosurgery, Perelman School of Medicine, University of
Pennsylvania, Philadelphia, PA, USA.
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