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| LETTER TO EDITOR |
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| Year : 2017 | Volume
: 65
| Issue : 2 | Page : 414-416 |
Occult sacral meningocoele associated with spinal dysraphism: Report of an unusual case and a review of literature
Ravi Dadlani1, Aditya A Atal2
1 Department of Neurosurgery, Sri Sathya Sai Institute of Higher Medical Sciences, Bengaluru, Karnataka; Department of Neurosurgery, Apollo Specialty Hospital, Nellore, India
2 Department of Neurosurgery, Sri Sathya Sai Institute of Higher Medical Sciences, Bengaluru, Karnataka, India
| Date of Web Publication | 10-Mar-2017 |
Correspondence Address:
Dr. Ravi Dadlani
Department of Neurosurgery, Apollo Specialty Hospital, Nellore
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/neuroindia.NI_599_16
How to cite this article:
Dadlani R, Atal AA. Occult sacral meningocoele associated with spinal dysraphism: Report of an unusual case and a review of literature. Neurol India 2017;65:414-6 |
Sir,
Sacral arachnoid cysts (SAC) or occult sacral meningocoeles (OSM) are alternatively used terms to describe cysts of the sacral spine. Nabors et al., classified spinal cysts to eliminate the confusion prevalent in the literature at that time.[1] These may occur in isolation or in close association with spinal dysraphic (SD) states.[1],[2],[3],[4] The latter situation may have a bearing on the etiopathogenesis and management of these cysts. We present a rare case of OSM associated with SD in an adult male who presented with predominant unilateral lower limb motor symptoms.
A 32-year-old gentleman presented with progressive walking difficulty of 3 year-duration associated with urinary incontinence for 9 months. He also developed left foot drop a few months prior to the admission., On examination, he had wasting of the left lower limb [Figure 1]a and [Figure 1]b and a complete foot drop [Figure 1]c. Magnetic resonance imaging (MRI) of the whole spine revealed [Figure 2] a holocord syrinx, low lying conus, and a sacral intradural cyst. He underwent a sacral laminectomy, decompression of the cyst, with partial excision of the cyst wall. Division of the fatty filum was performed to de-tether the cord from the dorsal surface of the cyst [Figure 3]. On retrospective scrutiny of the MRI, the fatty filum could be noted [Figure 4]. It was preoperatively thought to be extradural fat. Postoperatively, the patient had some improvement in his motor symptoms. Postoperative MRI done after a week of surgery demonstrated a reduction in the size of the syringomyelia [Figure 5]. | Figure 1: Predominant motor signs of the patient with gross atrophy of the left thigh and calf muscles (a, b) and a left foot drop (c)
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 | Figure 2: Preoperative magnetic resonance imaging of the spine demonstrating the SAC (a and b). The holocord syrinx is depicted in c and d
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 | Figure 3: Intraoperative image demonstrating the pathological anatomy after sacral laminectomy. The cyst was intradurally located and the fatty filum terminale anchored the thickened filum on to the dorsum of the cyst. Intradural location is demonstrated by the dura being reflected by a dissector on the right
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 | Figure 4: Demonstration in the lumbosacral spine of the fatty filum (black arrow) on the TI-weighted MRI images causing tethering at the dorsal surface of the cyst. This was initially mistaken for epidural fatafter
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 | Figure 5: Reduction in the size of the syrinx (c, d) to about half of preoperative size (a, b) after de-tethering. The syrinx in the preoperative image is marked by a black arrow (a) and by a white arrow in the postoperative image (d)
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The patient corresponded to the type III intradural cyst. A PubMed search revealed 34 articles that were exhibited on typing the key words “occult spinal meningocoele.” Twenty two of the articles were in English language. There were 8 articles from the pre-computed tomography (CT) and pre-MRI era while 5 articles reported OSM in the setting of SD [Table 1].[1],[2],[3],[4] | Table 1: Review of literature of SAC or OSM associated with spinal dysraphism
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Although there have been a few other reports of distant SAC associated with SD, sacral location of the arachnoid cyst is extremely rare. There is usually no anatomical proximity between these two lesions when they co-exist. In our case, however, the fatty filum was terminating on the dorsal aspect of the cyst and there was definite tethering of the cord. The latter lends credence to an etiological relationship between the two conditions. We highlight some of the major differences between an isolated OSM and SAC associated with SD in [Table 2]. | Table 2: Differences between a simple SAC/OSM and one associated with spinal dysraphism
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In conclusion, the diagnosis of OSM requires a high clinical suspicion and MRI screening of the entire spine is mandatory, especially when the clinical syndrome does not correlate with the level of the SAC.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.[5]
| » References | |  |
| 1. | Nabors MW, Pait TG, Byrd EB, Karim NO, Davis DO, Kobrine AI, et al. Updated assessment and current classification of spinal meningeal cysts. J Neurosurg 1988;68:366-77.  |
| 2. | Rabb CH, McComb JG, Raffel C, Kennedy JG. Spinal arachnoid cysts in the pediatric age group: An association with neural tube defects. J Neurosurg 1992;77:369-72. |
| 3. | Atabay H, Ee YE, Mirzai H. Occult intrasacral meningocoele associated with tethered cord syndrome. Turk Neurosurg 1994;4:176-9. |
| 4. | Okada T, Imae S, Igarashi S, Koyama T, Yamashita J. Occult intrasacral meningocele associated with spina bifida: A case report. Surg Neurol 1996;46:147-9. |
| 5. | Tsutsumi S, Wachi A, Uto A, Koike J, Arai H, Sato K. Infantile arachnoid cyst compressing the sacral nerve root associated with spina bifida and lipoma-case report. Neurol Med Chir (Tokyo) 2000;40:435-8. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
[Table 1], [Table 2]
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