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Open Journal of Modern Neurosurgery, 2016, 6, 16-19
Published Online January 2016 in SciRes. http://www.scirp.org/journal/ojmn
http://dx.doi.org/10.4236/ojmn.2016.61003
How to cite this paper: Gambhir, S., Winder, M. and Owen, G. (2016) A Rare Case of Anterior Sacral Meningocele (ASM) in
a Young Patient. Open Journal of Modern Neurosurgery, 6, 16-19. http://dx.doi.org/10.4236/ojmn.2016.61003
A Rare Case of Anterior Sacral Meningocele
(ASM) in a Young Patient
Shanu Gambhir, Mark Winder, Gareth Owen
Department of Neurosurgery, St. Vincent’s Hospital, Darlinghurst, Australia
Received 9 November 2015; accepted 3 January 2016; published 6 January 2016
Copyright © 2016 by authors and Scientific Research Publishing Inc.
This work is licensed under the Creative Commons Attribution International License (CC BY).
http://creativecommons.org/licenses/by/4.0/
Abstract
We report a 24-year-old male who presented with abdominal distension, constipation and left
sided groin pain. CT and MRI of the abdomen/spine were performed which showed a large ante-
rior sacral meningocele occupying most of the pelvic and abdominal cavity and displacement of
their respective contents. Initially a posterior approach with lumbosacral laminectomy (L5 - S4)
was performed. The ostium of the meningocele was identified with several nerve roots identified
passing through, adjacent and into the defect. As nerve roots traversed the ostium, watertight clo-
sure was not feasible without sacrificing nerve roots. Subsequent MRI demonstrated recurrence of
approximately 60% of the anterior sacral meningocele. We therefore opted to approach the ASM
anteriorly via an anterior approach with the help of colorectal surgical colleague. The ASM was
completely embedded within the sigmoid and upper to mid rectal mesentry, with its own vascular
supply to the thick walled capsule. This case highlights the need for a combined approach due to
the incorporation of the pseudomeningocele into the omentum with the development of its own
blood supply.
Keywords
Spina Bifida, Sacral, Meningocele, Abdominal Distension
1. Introduction
Anterior sacral meningocele (ASM) is an anomaly where the meninges protrude into retroperitoneal and presa-
cral space through an anterior sacral defect. Most of the cases present in adulthood and diagnosis in childhood is
rare. Common presentations include infection, meningitis and obstetric problems. We report a 24-year-old male
who presented with abdominal distension. This case highlights the need for a combined approach due to the in-
corporation of the pseudomeningocele into the omentum with the development of its own blood supply.
S. Gambhir et al.
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2. Case
2.1. Patient History
A 24-year-old male presented with a two-year history of gradually worsening abdominal distension. Acute
symptoms were related to constipation and left sided groin pain. He reported no nausea, vomiting or weight loss.
He did not have any other gastrological, urological or neurological symptoms or signs and was previously fit and
well. Examination revealed generalized abdominal enlargement that was non-tender with bowel sounds present.
2.2. Findings
CT and MRI of the abdomen/spine were performed which showed a large anterior sacral meningocele occupy-
ing most of the pelvic and abdominal cavity and displacing their respective contents (Figure 1). The meningo-
cele originated from the left anterolateral aspect of sacrum (S2 and S3) with no obvious neural structures passing
through the defect on imaging.
CT brain revealed evidence of prosencephaly without hydrocephalus. There was no evidence of chiari mal-
formation.
3. Surgical Approach
Due to the large size of the meningocele and displacement of the abdominal contents, and progressive symptoms,
a surgical approach was favoured over conservative management. Initially a posterior approach with lumbosa-
cral laminectomy (L5 - S4) was performed. The dura was opened in the midline and the thickened filum termi-
nale was identified and divided. Thickened meninges were encountered with loculated, complex arachnoid lay-
ers surrounding the defect. The ostium of the meningocele was identified with several nerve roots identified
passing through, adjacent and into the defect (Figure 2). As nerve roots traversed the ostium, watertight closure
was not feasible without sacrificing nerve roots. The cerebrospinal fluid (CSF) was aspirated from the menin-
gocele allowing it to collapse. Stay sutures were employed in the extremely thickened pseudomeningocele cap-
sule. The ostium was then closed by laying artificial dural graft (Duragen) over the defect and sutured with 4.0
prolene. Wound was closed in standard fashion.
The patient did not have any post-operative complications, however, subsequent MRI demonstrated recur-
rence of approximately 60% of the anterior sacral meningocele. We therefore opted to approach the ASM ante-
riorly via an anterior approach with the help of colorectal surgical colleague. The ASM was completely embed-
ded within the sigmoid and upper to mid rectal mesentry, with its own vascular supply to the thick walled cap-
sule. The colonic and rectal mesentry was skeletonized off the wall of the ASM, with preservation of the colonic
vascular supply, allowing resection of the ASM to the presacral space. The sac of the ASM was then opened,
and almost all of the sac resected. The small residual sac was closed with a purse string suture (Figure 3).
4. Outcome
The patient made full recovery without deficit and was discharged home one-week post operatively. Follow up
MRI at three months showed complete resolution of meningocele.
Figure 1. MRI abdomen showing large anterior sacral meningocele in sa-
gittal (a), axial (b) and cornoal (c) orientation.
S. Gambhir et al.
18
Figure 2. Demonstrating ostium of the anterior sacral meningocele after sa-
cral laminectomy.
Figure 3. demonstrating meningocele adherent to omentum and the size post
resection.
5. Discussion
ASM is a rare congenital anomaly which was first described in 1837 by Bryant [1]. The congenital malforma-
tion is usually associated with a sacrococcygeal bony defect in which there is a protrusion of dural sac anteriorly
[2]. Fewer than 300 cases of ASM have been reported in literature. It may be associated with a variety of conge-
nital disorders, most frequently the Currarino triad, consisting of congenital canal stenosis, scimitar sacrum and
a presacral mass [3]. The onset of symptoms is usually in early adult life secondary to gradually increasing
pressure on nearby pelvic structures resulting in urinary, bowel and obstetric problems. It has also been reported
to present as cauda equina syndrome [4].
Surgical intervention is almost always necessary as ASM tends to enlarge with time secondary to CSF pulsa-
tions and causes progressive symptoms. If left untreated ASM can result in 30% mortality due to recurrent me-
ningitis as well as obstetric and urinary tract complications [5]. There are no reports of spontaneous regression
suggesting that the risks are unlikely to subside without treatment.
Early intervention is preferred as the meningocele is likely to be smaller, making surgical treatment easier and
also, potentially reducing complications. The favoured approach for treatment of ASM is ligation of the ASM
stalk through sacral laminectomy [6]. Plication of the dura or duraplasty with artificial dural graft may be ne-
cessary where nerve roots pass through the defect as it makes watertight closure impossible, a feature in about
20% of ASMs. Combination with anterior approach may be considered if the stalk is too wide to be ligated via
laminectomy or if watertight closure cannot be accomplished due to ostium containing nerve roots, as performed
in this case. Anterior approach may be preferred as a first line approach if there are other associated abdominal
lesions, for example a tumour or a fistula that can be dealt with at the same time.
6. Conclusion
This case highlights the need for a combined approach due to the incorporation of the pseudomeningocele into
S. Gambhir et al.
19
the omentum with the development of its own blood supply.
References
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