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 »  Introduction
 »  Case report
 »  Discussion
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Year : 2000  |  Volume : 48  |  Issue : 3  |  Page : 279-81

Gluteal neuralgia - unusual presentation in an adult with intrasacral meningocele : a case report and review of literature.

Department of Neurosurgery, Khoula Hospital, PO Box - 90, Postal code - 116, Mina AI Fahal, Muscat, Oman.

Correspondence Address:
Department of Neurosurgery, Khoula Hospital, PO Box - 90, Postal code - 116, Mina AI Fahal, Muscat, Oman.

  »  Abstract

A nineteen year old man with intrasacral meningocele is reported, who presented with long standing episodic gluteal pain and progressive muscle wasting. Magnetic resonance imaging established the diagnosis. Surgical excision relieved the pain but muscle wasting persisted. Pertinent literature is reviewed.

How to cite this article:
Mishra G P, Sharma R R, Lad S D, Pawar S J, Mahapatra A K. Gluteal neuralgia - unusual presentation in an adult with intrasacral meningocele : a case report and review of literature. Neurol India 2000;48:279

How to cite this URL:
Mishra G P, Sharma R R, Lad S D, Pawar S J, Mahapatra A K. Gluteal neuralgia - unusual presentation in an adult with intrasacral meningocele : a case report and review of literature. Neurol India [serial online] 2000 [cited 2021 Jan 19];48:279. Available from:

   »   Introduction Top

Symptomatic intrasacral cysts presenting with nerve root or cauda equina compression are rare. A case of an occult intrasacral meningocele is described, who was initially managed as a case of motor neuron disease due to severe atrophy, weakness of the gluteal and posterior thigh muscles and positive neurophysiological investigations. However, magnetic resonance imaging (MRI) scan clinched the diagnosis of an occult intrasacral meningocele.

   »   Case report Top

A nineteen year old football player was referred to neurosurgery department with the complaints of pain in the left gluteal region and the posterior aspect of the thigh for the last 2 years and loss of muscle bulk in the same region for the last one year. Pain fluctuated in its intensity from mild to severe degree and was episodic in nature, lasting for few seconds to few minutes on the superimposed background of ongoing dull pain. Pain used to increase with exertion and was at times involving the posterior aspect of the left lower limb. He had to abandon playing football. There was no sphincter disturbance. Before coming to the neurosurgery, he was managed in various departments with diagnosis of motor neuron disease and muscle dystrophy etc. for two years. On examination there was obvious wasting of left gluteii and thigh muscles and mild atrophy of the left calf muscles. Muscle power in all groups was almost normal. Sensations and deep tendon reflexes were normal. Examinations of rest of the nervous system were normal.
Plain roentgenograms of lumbosacral spine showed scalloping of the posterior aspect of the first and second sacral vertebral bodies and a wide sacral canal. MRI of lumbosacral spine showed space-occupying lesion inside the wide sacral canal [Figure. 1]. It was hypointense on T1 weighted and hyperintense on T2 weighted images. The termination of the thecal sac was pushed upwards to the level of upper border of first sacral vertebra.
First and second sacral laminectomy was performed and the cyst was totally excised microsurgically. Laminae were very thin, perhaps due to the chronic pressure exerted by the underlying sacral cystic lesion. The lesion was a thin walled tense cyst, displacing the sacral thecal sac upwards and stretching the nerve roots present in lateral and anterior aspect, within the sacral canal. The content of the cyst was cerebrospinal fluid (CSF). The post-operative course was uneventful and the patient had satisfactory relief from pain, however, muscle wasting persisted on follow-up, six months following surgery. Histopathological examination revealed the cyst wall consisting of fibrocollagenous tissue.

   »   Discussion Top

Various types of cysts within the spinal canal are detected more frequently nowadays with better methods of imaging. These may be found as incidental findings or as a cause for myelopathy and/or radiculopathy.[1],[2],[3],[6],[7] Common spinal extradural cysts are grouped as (i) congenital spinal extradural cysts (extradural arachnoid cysts), (ii) spinal perineural cysts (Tarlov cysts), (iii) spontaneous meningeal diverticula along spinal nerve roots, and (iv) occult intrasacral meningocele.[8] Location of these cysts seems to be an important differentiating feature. Morphological and histological features are not of much help in differential diagnosis. The shape and size depends on the site. The cyst wall is mainly composed of collagenous connective tissue with or without a lining of cells resembling arachnoid cells.[8] The cyst cavity is mainly filled with a fluid similar to the CSF.
Congenital extradural spinal cysts are most often located in dorsal area, however, lumbar and cervical locations are not unknown. These are attached to the spinal dural sac in the midline posteriorly or to the dura over the dorsal surface of the nerve root posterolaterally. Tarlov cysts usually arise from sacral nerve roots whereas spontaneous meningeal diverticulae occur along the length of spine at the junction of nerve root and thecal sac. Interestingly, intrasacral meningocele is located in the sacral canal below the termination of the thecal sac[8] as found in our case.
Often, even with typical clinical features, the diagnosis of occult intrasacral meningocele is missed or delayed. The factors contributing to such delay in the diagnosis are (i) lack of awareness of this rare entity, (ii) incomplete radiological studies and (iii) early myelogram may not fill the meningocele and may even show a virtually normal thecal sac.
The true occult intrasacral meningoceles are rare.[1],[2],[3],[4],[6],[7] Forty four cases had been reported between 1932 and 1972[9] and, since then, forty more cases have been added. The delayed presentation is due to slow expansion of meningocele over a period of several years. Therefore, symptoms and signs are usually present for several years before the final diagnosis.[3] In an extensive literature review, pain in the low back, buttocks and legs was the initial presenting symptom in 69 percent of cases, weakness in the lower limbs was noted in 56 percent and bowel and bladder dysfunction was present in 44 per cent of patients.[3] Lamas et al pointed out association of occult intrasacral meningocele with chronic neuromusculoskeletal syndrome in one case.[5] Interestingly, our patient had no sensory deficit except severe gluteal neuralgia. MRI of the lumbo-sacral spine is the investigation of choice, by which the CSF intensity lesion situated inside the wide sacral canal is clearly identified. However, the degree of communication between the subarachnoid space and the meningocele may not be clear even on the MR study. In the case reported here, the diagnosis of intrasacral meningocele came into picture only after MRI scan. All cases of long standing gluteal and lower extremity pain with or without neurological deficit such as muscle wasting must be subjected to MRI examination of the lumbo-sacral spine at the earliest possible to rule out rare lesions like intrasacral meningocele.


  »   References Top

1.Abbott KH, Retter RH, Leimbach WH : The roles of perineural sacral cysts in the sciatic and sacrococcygeal syndromes: A review of the literature and report of 9 cases. J Neurosurg 1957; 14 : 5-21.   Back to cited text no. 1    
2.Crellin RQ, Jones ER : Sacral extradural cysts : A rare cause of low backache and sciatica. J Bone Joint Surg 1973; 55B : 20-31.   Back to cited text no. 2    
3.French BN : Midline fusion defects and defects of formation. In : Neurological surgery. JR Youmans (Ed.) Third edition, W.B. Saunders company, Philadelphia 1990; 2 : 1081-1235.   Back to cited text no. 3    
4.Joseph RA, McKenzie T : Occult intrasacral meningocele. J Neurol Neurosurg Psychiatry 1970; 33 :493-496.   Back to cited text no. 4    
5.Lamas E, Lobato RD, Armor T: Occult intrasacral meningocele. Surg Neurol1977; 8 :181-184.   Back to cited text no. 5    
6.Pool JL : Spinal cord and local signs secondary to occult sacral meningocele in adults. Bull N Y Acad Med 1952; 28 : 655-663.   Back to cited text no. 6    
7.Reddy DR, Sathyanarayana K, Krishnamurthy D : Occult intrasacral meningocele: Report of a case. Aust N Z J Surg 1974; 44 : 273-275.   Back to cited text no. 7    
8.Wilkins RH : Intraspinal cysts. In: Neurosurgery. RS Wilkins , SS Rengachary (eds) Second edition, McGraw-Hill, New Yark. 1996; 3 : 3509-3519.   Back to cited text no. 8    
9.Wilkins RH, Odom GL : Spinal extradural cysts. In : Handbook of Clinical Neurology. Vinken PJ, Bruyn GW (eds) North Holland, Amsterdam. 1976; 20 : 137-175.   Back to cited text no. 9    


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