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caSe reportJ Neurosurg Spine 24:862–866, 2016

Anterior sacral meningoceles (ASMs) are rare forms of spinal dysraphism. Their pathomecha-nism involves herniation of the thecal sac through

a bony defect in the anterior sacral wall. Symptoms of ASMS are directly related to their mass effect over sur-rounding pelvic structures causing chronic constipation and urinary disturbances among others.13 The combina-tion of obstructive renal failure and hydronephrosis due to the compressive mass effect of giant ASMs is rare. We present the case of a 37-year-old man with bilateral hy-dronephrosis secondary to the mass effect of a large ASM. Furthermore, we review treatment options and surgical de-cision making for ASMs, describing a novel meningocele closure approach in which an endoscopic cutting stapler is used.

case reportInitial Presentation and Examination

A 37-year-old man with spina bifida occulta, initially

diagnosed at birth, was followed up by the neurosurgery service for a large ASM associated with neurogenic blad-der and recurrent urinary tract infections. As the ASM was initially considered asymptomatic, the patient was fol-lowed up serially until a urinary tract infection progressed to the extent of sepsis that required hospitalization and ad-mission to the intensive care unit. The size of the menin-gocele progressed, obstructing both ureters and causing hydronephrosis and acute renal failure, for which ureteral stents were placed by cystoscopy. Serum creatinine levels had increased to 150–180 mmol/L (1.7–2.0 mg/dl) (normal 50–115 mmol/L or 0.6–1.3 mg/dl).

On examination, he had spastic limbs bilaterally; the strength of the distal muscle groups was normal except he had reduced left plantar flexion resulting from a prior foot surgery. A large mass was both palpable and visible in the midabdominal area. MRI of the lumbar spine and abdomen demonstrated an expansive ASM measuring 25 × 12 × 18 cm, grossly displacing the abdominal structures (Fig. 1).

abbreviatioNS ASM = anterior sacral meningocele.Submitted March 27, 2015.  accepted August 17, 2015.iNclude wheN citiNg Published online January 8, 2016; DOI: 10.3171/2015.8.SPINE15129.

Successful management of a giant anterior sacral meningocele with an endoscopic cutting stapler: case reporttarek p. Sunna, md, harrison J. westwick, md, mSc, Fahed Zairi, md, ilyes berania, md, and daniel Shedid, md, mSc, FrcSc, FabN

Department of Surgery, Division of Neurosurgery, Centre Hospitalier de l’Université de Montréal, Hôpital Notre-Dame, Montreal, Quebec, Canada

Anterior sacral meningoceles (ASMs) are rare defects in the sacrum with thecal sac herniations and symptoms that com-monly include constipation, dysmenorrhea, and urinary disturbances. An ASM causing hydronephrosis and acute renal failure from compression of the lower portion of the urinary tract is a rare clinical entity. Only one other case has been reported. The authors present the case of a 37-year-old man admitted for obstructive renal failure and hydronephrosis due to a giant ASM that measured 25 × 12 × 18 cm and compressed the ureters and bladder. The ASM was successfully treated via an anterior transabdominal approach in which the authors used a novel technique for watertight closure of the meningocele pedicle with an endoscopic cutting stapler. The authors review the literature and discuss the surgical options for the treatment of ASMs, specifically the management of ASMs in the context of obstructive renal failure and hydronephrosis.http://thejns.org/doi/abs/10.3171/2015.8.SPINE15129Key wordS meningocele; spina bifida; hydronephrosis; renal failure; congenital

©AANS, 2016

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OperationThe primary surgical objective was the closure of the

communication between the meningocele and subarach-noid space. This involved direct visualization of the anom-aly, aspiration of the meningocele contents, spinal cord detethering, meningocele pedicle ligation, and watertight closure.22

Repair of the meningocele was first attempted through a sacral laminectomy. Operative findings revealed that the spinal cord was tethered anteriorly at the sacrum with nerve tissue extending into the abdominal portion of the meningocele (confirmed by intraoperative stimulation). With regard to this, it was felt the risks of neurological deterioration were too great with a posterior approach alone. Thus, the procedure was aborted and the meningo-cele would be approached anteriorly in a second operative procedure after discussion with the patient.

One week later, in collaboration with the general sur-geon, the patient was brought back to the operating room and the meningocele was approached via a midline trans-abdominal approach. After dissection of the ASM from the surrounding abdominal structures, the retroperitoneum was opened to reach the base of the ASM at the sacrum.

The ASM was opened to dissect the adherent nerve tis-sue from the dura mater. Watertight closure of the pedicle was achieved with a 45-mm articulating endoscopic linear stapler/cutter (Endopath ETS Compact-Flex 45, Ethicon) with 2.5-mm-height staples and 6 staple rows. Despite the absence of leakage, the staple line was reinforced with ad-ditional suture line and fibrin glue (Fig. 2).

Postoperative CourseThe postoperative course was uneventful and creati-

nine levels rapidly dropped to 115–128 mmol/L (1.3–1.4 mg/dL). The patient has been followed up for a period of 9 months with improvement of urinary symptoms and stable MRI findings (Fig. 3); his leg spasticity, however, has re-mained unchanged.

discussionThe pathogenesis of ASMs is thought to be one of a

congenital neural tube defect arising during embryonic development at the stage of neurulation with a defect in the anterior sacral wall. Meningocele development is ex-plained by CSF pulsation eroding the weakened sacral wall and leading to out-pouching.18,22 ASM formation has

Fig. 1. Preoperative sagittal (a) and axial (b) T2-weighted images of lumbosacral spine, with the ASM measuring 25 × 12 × 18 cm. Preoperative abdominal radiograph (c) demonstrating displacement of ureteral stents by the meningocele.

Fig. 2. Illustration of surgical closure of the meningocele pedicle via a transabdominal approach with an endoscopic articulating cutting stapler after the ASM was opened to ensure no entrapment of neural tissue: stapler placement (a), staple line (b), and pedicle removed (c). Copyright Ilyes Berania. Published with permission.

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also been described in the context of minor trauma to the anterior sacrum, through the same proposed mechanism of CSF pulsation and subsequent erosion.9 Otherwise, some clinical conditions such as Currarino’s triad and Marfan’s have been associated with ASMs. In the present report, our patient did not have any of these associated conditions.10,22

The presentation of ASMs is highly variable; it is be-lieved that the true incidence is underestimated due to their relatively asymptomatic nature. Approximately 300 cases of ASM have been described in the literature.7 The diagnosis is incidental in the context of gynecological investigations or pregnancy in otherwise asymptomatic patients.6,19,27 When present, symptoms are mostly related to the mass effects over surrounding organs in the pel-vis. The local pressure on the rectum, urinary bladder, female genital organs, and sacral nerve roots produces a predictable array of symptoms—chronic constipation,1,14,19 urinary incontinence, dysmenorrhea, and chronic back pain,19 among others, are all well recognized. In extreme cases, fistulization between the meningocele and intes-tines can occur.5 Indeed, ASMs may present with bacterial meningitis, presumably due to a communication between the CSF and an enteric structure.24 Finally, death is also a dreaded complication of ASM, since rare cases of fatal spontaneous rupture have been reported during labor.27

Management of ASMs reported in the literature has been both surgical and observational. It has been acknowl-edged that conservative management is an appropriate op-tion for small, uncomplicated ASMs, without associated tumor or in the context of pregnancy. However, spontane-ous regression of ASMs has not been observed and life-threatening complications such as fistulization, meningi-

tis, and rupture have been described.5,6,21,22 Conversely, a surgical approach is indicated in the case of symptomatic compression of pelvic structures or fistulization. In the present case, obstructive renal failure and hydronephrosis were related to the mass effect of the ASM, and it was felt that the obstructive renal failure would continue to evolve without surgical intervention. Various surgical approach-es have been proposed for the treatment of symptomatic ASMs. Early reports have noted transrectal or transvagi-nal aspiration of cyst contents. However, these approaches were associated with infectious complications, early re-currence, and death.18 Various different conventional sur-gical approaches exist for the treatment of ASMs, but de-cisions are made on a case-by-case basis. Principal factors that influence surgical approach include size of the ASM, size of the ostium, and presence of tumors.25 The anterior transabdominal approach was first introduced as a means of obtaining direct access to the ASM; however, surgical risks of trauma to adjacent structures and high rates of complications due to incomplete “watertight” dural closure have limited acceptance of this approach. However, this technique is highly applicable in the case of large ASMs or large ostium, and with careful dissection of visceral structures, risks can be minimized.2,22,25 The posterior ap-proach, in which sacral laminectomy and ligation of the neck of the meningocele are performed, was introduced in 1938 and was associated with fewer complications than the anterior operation.11,23 In this approach, the goal is to close the communication between the subarachnoid space and meningocele, and spontaneous regression occurs in most cases.2,22 The posterior sagittal approach has been used frequently in the treatment of pediatric colorectal disorders, and it allows good exposure to the pelvic floor while minimizing potential neurological complications.18 The inferior presacral approach has a high risk of visceral injury and offers poor visualization of neural structures. The oblique parasacral approach, used mostly in gluteal meningoceles, does not offer access to the tethering struc-tures or residual meningocele for resection.22 Endoscopy has also been employed for the treatment of ASMs, al-lowing for obliteration of the subarachnoid-meningocele communication with fat grafts. The meningocele was re-ported to regress significantly following fistula closure.15 The endoscopic approach offers the advantages of a small incision and short operative time. Laparoscopic resection of ASMs has been employed to aspirate the cystic con-tents and debulk the pelvic mass of the ASM, with closure of the pedicle with clips and resection of the redundant tissue, but this was done only after the communication was closed via a traditional posterior approach. The au-thors cited easy dissection and decreased risk of damage to adjacent structures as the principal advantages of this approach.26 However, the limited experience explains that the role of endoscopy in the evaluation and treatment of meningoceles is still being assessed.20 Moreover, this type of endoscopy is less familiar to the spine surgeon.22 Fi-nally, lumboperitoneal shunts can be employed as an ad-juvant treatment in the case of failed primary treatment of ASMs, but as is the case when shunting in other contexts, shunt malfunction is a limitation.4,22

Acute urinary complications related to the direct com-

Fig. 3. Postoperative sagittal (a) and axial (b) T2-weighted images of the ASM showing net size reduction from preoperative imaging, and showing stability 8 months postoperatively (c and d).

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pression of an anterior sacral meningocele have been rarely reported in the literature4,7,8,16,17 (Table 1). In the 4 reported cases, symptoms were explained by the mass ef-fect exerted on the bladder, and on initial physical exami-nation, the meningocele could be mistaken for a palpable globe.4,7 Hydronephrosis related to compressive ASM was described in only one other case.7 Various treatment mo-dalities have been reported, but in the majority of circum-stances, surgical intervention was undertaken with resolu-tion of the urinary symptoms. In only one case, described by Lefere and colleagues, an ASM recurred after surgical management, and symptoms resolved once a lumboperi-toneal shunt was placed.17 In these cases, there were no patients with genetic syndromes.

In the present case, obstructive renal failure and hydro-nephrosis were related to the mass effect of the ASM, and it was felt that the symptoms would continue to evolve in the absence of surgical intervention. We proceeded with a dorsal transsacral approach, but after the sacral laminec-tomy was done, we noted that the cord and the nerve tissue had extended beyond the level of the sacrum and into the intraabdominal part of the ASM. Based on evaluation of the imaging, it would have been appropriate to proceed primarily with the transabdominal approach; however, given the advantages of the transsacral approach, we felt it offered advantages. As we continued in the transabdomi-nal approach, we noted, after opening of the ASM, that the neural tissue had migrated to the lower third of the meningocele. With intraoperative nerve stimulation, we identifed that these nerves were functional, and thus it was crucial to decide at which level to close the pedicle. The endoscopic articulating cutting stapler was used to address one of the main shortcomings of the abdominal approach, that of incomplete closure of the dura and intraperitoneal leakage of CSF. This device allowed for watertight clo-sure in a time-efficient manner (Fig. 2). With further re-inforcement of the staple line with additional sutures and fibrin glue, this closure method, we believe, offers advan-tages in the closure of the meningocele pedicle and re-moval of the redundant tissue. The use of gastrointestinal

anastomosis devices has been previously suggested as a successful means of ensuring watertight dural closure in a time-efficient manner.3,12 However, we highlight the ad-vantages of an endoscopic articulating stapler for closing dural pedicles in difficultly accessible situations and with complex conformation.

This report presents an important limitation. Indeed the follow-up duration was limited (9 months), and the slowly progressive nature of this disease does not prevent late recurrence. A close and prolonged monitoring is then highly suitable. As reported by Lefere and colleagues, a shunt could be proposed in such a situation. This treat-ment option has not been proposed as first line given the size of the lesion and the severity of compression, which favored an open surgery to obtain a rapid and significant reduction of the mass effect.

However, this case is remarkable in many aspects. This is only the second reported case of hydronephrosis due to a giant ASM. It also highlights the difficulty of the decision making, with the need to preserve functional neurologi-cal structures to protect the patient’s neurological status. Finally, it demonstrates the value of a novel dural closure technique that provided effective watertight closure, mak-ing the anterior transperitoneal approach safer.

references 1. Amacher AL, Drake CG, McLachlin AD: Anterior sacral

meningocele. Surg Gynecol Obstet 126:986–994, 1968 2. Ashley WW Jr, Wright NM: Resection of a giant anterior

sacral meningocele via an anterior approach: case report and review of literature. Surg Neurol 66:89–93, 2006

3. Banks ND, Redett RJ: Dural repair with grafts using the gastrointestinal anastomosis stapler. J Craniofac Surg 19:1053–1055, 2008

4. Barlow D, Gracey L: Anterior sacral meningocele causing urinary retention. Br J Surg 50:732–734, 1963

5. Bergeron E, Roux A, Demers J, Vanier LE, Moore L: A 40-year-old woman with cauda equina syndrome caused by rectothecal fistula arising from an anterior sacral meningo-cele. Neurosurgery 67:E1464–E1468, 2010

6. Beyazal M: An asymptomatic large anterior sacral meningo-

table 1. Summary of the present case and 5 previous cases of acute urinary presentations associated with aSms

Authors & YearAge (yrs), 

Sex  Clinical Finding Hydronephrosis Treatment Outcome

Present case 37, M Recurrent urinary tract infections, hydronephro-sis & renal failure

Yes Posterior & abdominal Resolution of urinary symptoms

Castelli et al., 2013

37, M Progressive urinary retention symptoms, w/ meningocele compressing bladder, & hydronephrosis

Yes Posterior  Resolution of urinary symptoms

Kubota et al., 2012

31, F Pregnant; 1-yr Hx intermittent urinary difficulty; new-onset acute retention; cesarean delivery

NS None NS

Lefere et al.,  2009

22, M Acute urinary retention w/ 1 L residual NS 1) Surgical NS muscle flap; 2) LP shunt

Failure of surgery; reso-lution w/ shunt

Chovnick, 1971 50, M Chronic urinary retention w/ indwelling catheter NS Abdominal approach; closure of cyst

Recovery of urinary & bowel functions

Barlow & Gracey, 1963

28, F Progressive urinary retention requiring cath-eterization & constipation

NS 1) Lumbar punture; 2) presacral muscle graft 

Resolution of urinary symptoms; procidentia

Hx = history; LP = lumboperitoneal; NS = not specified.

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cele in a patient with a history of gestation: a case report with radiological findings. Case Rep Radiol 2013:842620, 2013

7. Castelli E, Rosso R, Leucci G, Luparello V, Collura D, Gia-cobbe A, et al: Huge anterior sacral meningocele simulating bladder retention. Urology 81:e9–e10, 2013

8. Chovnick SD: Anterior sacral meningocele as a cause of uri-nary retention. J Urol 106:371–374, 1971

9. Cools MJ, Al-Holou WN, Stetler WR Jr, La Marca F, Valdivia-Valdivia JM: Anterior sacral pseudomeningocele following minimal trauma: case report. J Neurosurg Spine 19:384–388, 2013

10. Currarino G, Coln D, Votteler T: Triad of anorectal, sacral, and presacral anomalies. AJR Am J Roentgenol 137:395–398, 1981

11. Dyck P, Wilson CB: Anterior sacral meningocele. Case re-port. J Neurosurg 53:548–552, 1980

12. Epstein NE, Hollingsworth R: Anterior cervical micro-dural repair of cerebrospinal fluid fistula after surgery for ossifica-tion of the posterior longitudinal ligament. Technical note. Surg Neurol 52:511–514, 1999

13. Fitzpatrick MO, Taylor WA: Anterior sacral meningocele associated with a rectal fistula. Case report and review of the literature. J Neurosurg 91 (1 Suppl):124–127, 1999

14. Jaffe R: Anterior sacral meningocele. Report of a case. Ob-stet Gynecol 28:684–688, 1966

15. Jeon BC, Kim DH, Kwon KY: Anterior endoscopic treatment of a huge anterior sacral meningocele: technical case report. Neurosurgery 52:1231–1234, 2003

16. Kubota Y, Kojima Y, Kohri K: Maternal anterior sacral me-ningocele presenting as acute urinary retention in pregnancy. Int J Urol 19:279–280, 2012

17. Lefere M, Verleyen N, Feys H, Somers JF: Anterior sacral meningocoele presenting as acute urinary retention. A case report. Acta Orthop Belg 75:855–857, 2009

18. Massimi L, Calisti A, Koutzoglou M, Di Rocco C: Giant anterior sacral meningocele and posterior sagittal approach. Childs Nerv Syst 19:722–728, 2003

19. Oren M, Lorber B, Lee SH, Truex RC Jr, Gennaro AR: An-terior sacral meningocele: report of five cases and review of the literature. Dis Colon Rectum 20:492–505, 1977

20. Raftopoulos C, Balériaux D, Hancq S, Closset J, David P, Brotchi J: Evaluation of endoscopy in the treatment of rare meningoceles: preliminary results. Surg Neurol 44:308–318, 1995

21. Shastin D, Selvanathan SK, Tyagi AK: A rare case of ante-rior sacral meningocoele presenting as cauda equina syn-drome. Br J Neurosurg 29:428–429, 2015

22. Shedid D, Roger EP, Benzel EC: Presacral meningocele: di-agnosis and treatment. Semin Spine Surg 18:161–167, 2006

23. Smith HP, Davis CH Jr: Anterior sacral meningocele: two case reports and discussion of surgical approach. Neurosur-gery 7:61–67, 1980

24. Somuncu S, Aritürk E, Iyigün O, Bernay F, Rizalar R, Günaydin M, et al: A case of anterior sacral meningocele to-tally excised using the posterior sagittal approach. J Pediatr Surg 32:730–732, 1997

25. Tani S, Okuda Y, Abe T: Surgical strategy for anterior sacral meningocele. Neurol Med Chir (Tokyo) 43:204–209, 2003

26. Trapp C, Farage L, Clatterbuck RE, Romero FR, Rais-Bah-rami S, Long DM, et al: Laparoscopic treatment of anterior sacral meningocele. Surg Neurol 68:443–448, 2007

27. Villarejo F, Scavone C, Blazquez MG, Pascual-Castroviejo I, Perez-Higueras A, Fernandez-Sanchez A, et al: Anterior sacral meningocele: review of the literature. Surg Neurol 19:57–71, 1983

disclosures Dr. Daniel Shedid reports being a consultant for DePuy Synthes and receiving fellowship support from Medtronic and DePuy Synthes. Dr. Westwick reports being a consultant for DePuy Syn-thes and receiving fellowship support from DePuy Synthes.

author contributionsConception and design: Shedid. Acquisition of data: Sunna, Westwick, Berania. Analysis and interpretation of data: West-wick, Zairi, Berania. Drafting the article: Sunna, Westwick, Berania. Critically revising the article: Shedid, Sunna, Westwick, Zairi. Reviewed submitted version of manuscript: Shedid, Sunna, Westwick, Zairi. Approved the final version of the manuscript on behalf of all authors: Shedid. Study supervision: Shedid.

correspondenceDaniel Shedid, Centre Hospitalier de l’Université de Montréal, Chef du Service de Neurochirurgie, 1560 Sherbrooke Est., Mon-treal, QC H2L 4M1, Canada. email: danielshedid@gmail.com.

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