J Neurosurg 72:951-954, 1990

Giant sacrolumbar meningioma Case report JOHN A. FELDENZER, M.D., JOHN E. McGILLICUDDY, M.D., AND JONATHAN W. HOPKINS, M.D.

Section of Neurosurgery, University of Michigan Medical Center, Ann Arbor, and Bronson Medical Center, Kalamazoo, Michigan A case of giant sacral meningioma with presacral and lumbar extension is presented. The difficulties in diagnosis and management are emphasized including the staged multidisciplinary surgical approaches and preoperative tumor embolization. KEY W O R D S

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spinal neoplasm

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ENINGIOMAS account for 25% of intraspinal neoplasms, with the thoracic area being the most c o m m o n location (61% to 95% of reported cases).4-8'11.12,14,16,20,22-24Meningiomas at the caudal aspect of the spine, however, are exceedingly unusual. To our knowledge, a sacral meningioma without lumbar extension has not previously been reported, although two cases have been documented at the L 5 S1 level. 4'29 Primary t u m o r s of the sacrum are also rare. 3'9'1~ This case o f a giant sacral meningioma extending into the l u m b a r subarachnoid and presacral spaces demonstrates the difficulty in diagnosing sacral lesions at an early stage. The value of a multidisciplinary approach to the successful managem e n t of this large sacral t u m o r is emphasized. Case Report

This 39-year-old w o m a n was first evaluated at the University of Michigan in July, 1987, for low-back and bilateral radicular leg pain. She also described mildly decreased perineal sensation, decreased sensation of bowel movements, and m a r k e d limitation of mobility of the lower back. At the time o f initial examination, she was unable to lie fiat on her back or sit because of severe low-back and radiating bilateral buttock pain. The patient first noticed the pain 9 years prior to our evaluation when it was localized to the coccygeal area. She failed conservative t r e a t m e n t of her coccydynia and in 1979 underwent a coccygectomy, which was complicated by w o u n d infection. The low-back pain J. Neurosurg. / Volume 72/June, 1990

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was mild but persistent until an exacerbation in 1983 after the patient fell on the seat o f an exercise bicycle. After that, she noted a gradual progression o f her lowback and bilateral leg pain. H e r initial neurosurgical evaluation, in 1987, included a computerized tomography (CT) scan and m y e l o g r a m a n d she was subsequently referred to the University o f Michigan Medical Center. E x a m i n a t i o n . Physical e x a m i n a t i o n revealed a well-developed w o m a n in no acute distress. H e r general physical examination was within n o r m a l limits. H e r gait was slightly broad-based a n d she had very limited m o t i o n of the lumbosacral spine, with 15* o f forward flexion, 20* of lateral flexion, a n d 10 ~ o f extension. There was a firm, tender, palpable subcutaneous mass at the lumbosacral junction a n d a firm presacral mass was detected on rectal e x a m i n a t i o n . Neurological exa m i n a t i o n revealed n o r m a l m e n t a l status and cranial nerve function. There was no m o t o r , sensory, or reflex deficit in the u p p e r extremities. L o w e r extremity examination, which was limited by the patient's pain, demonstrated m i n i m a l diffuse weakness; however, there was significant weakness o f the gluteus m a x i m u s and medius muscles bilaterally. Reflexes were n o r m a l at the knees and ankles bilaterally. Sensory examination was normal except for decreased pinprick sensation in the $3-5 d e r m a t o m e s bilaterally. Routine laboratory values, chest x-ray film, a n d electrocardiogram were normal. An e l e c t r o m y o g r a m showed no radioculopathy, plexopathy, or m o n o n e u r o p a t h y . 951

J. A. Feldenzer, J. E. McGillicuddy, and J. W. Hopkins

FIG. 1. Left: Anteroposterior radiograph of the lumbosacral spine showing erosion of S-l, S-2, and S-3 on the right (arrow). Right: Lumbosacral myelogram demonstrating a complete block at the mid L-3 level.

Plain spinal radiographs demonstrated sacral erosion posteriorly (Fig. l left). Spinal radiographs obtained at the onset o f s y m p t o m s in 1979 showed no evidence of coccygeal fracture or bone erosion of the sacrum. The lumbosacral m y e l o g r a m showed a complete block, suggestive of an intradural mass, at the L-3 level (Fig. 1 right). T h e initial C T scan demonstrated an intrasacral t u m o r with erosion o f the posterior sacral wall and presacral extension. Pelvic arteriography demonstrated a vascular presacral mass. Operations. A n open biopsy of the posterior subcutaneous mass was p e r f o r m e d and the final pathology was consistent with a poorly differentiated meningioma. There was no neurological change after this procedure and 4 weeks later the patient underwent an L 2 - 5 lami n e c t o m y with gross total removal o f the intradural l u m b a r c o m p o n e n t o f the tumor. The t u m o r was easily separated f r o m the nerve roots but was very vascular (1900 cc blood loss). Once again, the pathological diagnosis was m e n i n g i o m a . N o mitosis or necrosis was seen (Fig. 2). Postoperatively, the patient had a new left L-4 sensory deficit, decreased rectal tone, and absent ankle reflexes. Because o f the t u m o r ' s size, location, and vascularity, an embolization procedure was attempted prior to extirpation o f the sacral and presacral components. The patient u n d e r w e n t embolization o f the right L-5 and both internal iliac vessels with coils and Ivalon synthetic polyvinyl formyl alcohol f o a m sponge* (Fig. 3). The left L-5 a n d middle sacral arteries could not be catheterized deeply enough to permit safe embolization. The patient experienced severe back and radiating leg pain with increased neurological deficits and fever within 12 * Ivalon sponge manufactured by Unipoint Industries, High Point, North Carolina. 952

FIG. 2. Photomicrograph showing a giant sacrolumbar meningioma with focal calcification (arrow). There was no evidence of necrosis or mitosis. H & E (magnification not available).

hours of the embolization procedure. Steroid therapy provided minimal relief and she required large doses o f narcotic analgesics. A C T scan after t u m o r embolization demonstrated the extent of the intrasacral and pelvic c o m p o n e n t s (Fig. 4 upper and center) and multiple low-density areas of infarction within the t u m o r (Fig. 4 lower). Increased pain and neurological deficit after embolization were thought to be related to t u m o r swelling and further nerve root compression as a result of t u m o r infarction. Three days postembolization, the patient underwent a combined posterior transsacral (neurosurgical) and lateral transabdominal (general surgical) approach in the right lateral decubitus position. Cystometric a n d anal sphincter monitoring was utilized during surgery. The initial approach was posterior and the dura was noted to be disrupted (anteriorly and posteriorly) at the S-1 level and below. Nearly all of the posterior wall o f the sacrum was eroded. The left S-2 and S-3 foramina were enlarged and the anterior wall of the sacrum on the fight was destroyed at this level with t u m o r extending into the pelvis. A gross total excision o f the intrasacral portion was accomplished through this approach. All of the right sacral nerves and the left S-1 and S-2 nerves were preserved. The general surgeons then entered the pelvis through an incision in the left lower quadrant, and a transperitoneal approach to the retrorectal space revealed the presacral component. Dissection of the last fragments of t u m o r off of the sacral nerves exiting the anterior foramina was performed by the neurosurgeons. The patient was then returned to the prone position and a fascia lata graft was used to close the dura. The graft was sewn to the dural margins laterally and to the dura covering the root sleeve at S-3 on the right and S-2 on the left. Decompression of the sacral nerves and a gross total excision of the giant meningioma was accomplished through this c o m b i n e d anterior and posterior approach.

J. Neurosurg. / Volume 72~June, 1990

Giant sacrolumbar meningioma

FIG. 3. Postembolization pelvic angiogram demonstrating reduced blood flow to the tumor through the occluded internal iliac arteries,

Postoperative Course. After surgery, the patient had complete relief of her back and radiating leg pain. She had normal right lower-extremity strength except for 4/ 5 strength of the right dorsal and plantar foot flexor muscles. In the left lower extremity, she had no dorsiflexion and only trace plantar flexion in addition to 3/5 hamstring and 3 - / 5 gluteus medius and maximus muscle function. Sensory examination showed normal sensation to the S-1 level and diffuse hypalgesia from S-3 to S-5 on the right. Sensation f r o m L-5 to S-5 on the left was decreased. The patient underwent an intensive course of physical therapy, and 1) years postoperatively she has no back or leg pain, is able to walk, and has had return of bladder and sexual function. She is still unable to dorsiflex the left foot. A postoperative CT scan in May, 1988, showed no t u m o r recurrence. An asymptomatic persistent fluid collection, suggesting a meningocele in the floor o f the pelvis, is being monitored conservatively. Discussion The clinical course o f this patient emphasizes the problem of diagnosing a sacral t u m o r at an early stage. Sacral tumors are rare. In addition, the capacity of the sacral spinal canal to a c c o m m o d a t e a slow-growing t u m o r m a y result in the absence o f localizing symptoms and cause a delay in diagnosis. S y m p t o m s of low-back and/or radicular pain are often misdiagnosed as evidence of intervertebral disc d i s e a s e , l~ In our series of nine patients with sacral tumors, sphincter and sacral sensory disturbances were i m p o r t a n t clinical findings in association with low-back and radicular pain. j~ A palpable rectal mass was noted in 67% of these patients. 1o J. Neurosurg. / Volume 72/June, 1990

FIG. 4. Upper." Pelvic computerized tomography scan demonstrating the intrasacral component and the extent of posterior erosion into the subcutaneous space (arrow). Center: Scan showing erosion of the anterior aspect of the right S-3 level (arrowhead) with a large presacral component on the left emerging from the S-3 foramen (arrow). Lower: Postembolization scan showing the large presacral tumor with multiple areas of low density due to infarction (arrow).

Standard sacral radiographs are often difficult to interpret due to p o o r quality or overlying bowel gas or fecal material. Early bone erosion changes m a y be missed) 7,27 The CT scan is an excellent diagnostic tool but the standard scan to rule out disc herniation often ends at the S-1 level, thus potentially missing a lower sacral tumor. The choice of surgical approach is determined by the type of lesion, the age and physical condition of the 953

J. A. Feldenzer, J. E. McGillicuddy, and J. W. Hopkins patient, and the a m o u n t of sacral destruction. The surgical approach to large sacral and presacral tumors has evolved from an exclusively posterior approach to one that involves anterior, posterior, or combined operations. 1.2.15.18.26 The posterior approach, popularized by MacCarty, et al., TM is limited due to poor control of sacral arteries and presacral veins. Due to the lumbar, sacral, and presacral extent o f this t u m o r in addition to its vascularity, we utilized a staged multidisciplinary surgical approach. After removal of the lumbar component and confirmation of the diagnosis o f meningioma, it was decided to reduce the vascularity with embolization prior to combined anterior and posterior approaches to the sacral component. After embolization, the significant increase in low-back pain and the new neurological deficit were attributed to infarction of the t u m o r with edema, causing compression of the nerve roots. After this experience, we would encourage surgeons to operate within 12 to 18 hours after embolization to avoid further neurological deterioration. This patient did not require a fusion procedure as the sacroiliac joints were not significantly involved on either side. Diffuse destruction o f the sacrum by t u m o r or a radical sacral resection may require a stabilization procedure. Abernathey, et aL, 1 r e c o m m e n d e d evaluation of sacroiliac joint stability 9 to 12 weeks after tumor resection. In summary, this giant sacral t u m o r with presacral extension was diagnosed at a late stage due to nonspecific symptoms and signs. Important to the diagnosis was a careful history and physical examination with attention to the sacral sensory and rectal examination findings. The CT scan was the diagnostic procedure of choice but angiography and myelography added important information in the management o f this patient. Sacral tumors of this size often require a multidirectional approach and combined surgical specialty management. These cases should be individualized, with consideration given to the patient's age, general physical condition, neurological status, and prognosis based on a pathological diagnosis. References 1. Abernathey CD, Onofrio BM, Scheithauer B, et al: Surgical management of giant sacral schwannomas. J Neurosurg 65:286-295, 1986 2. Adson AW: Intraspinal tumors: surgical consideration. Int Abstr Surg 67:225-237, 1938 3. Adson AW, Moersch FP, Kernohan JW: Neurogenic tumors arising from the sacrum. Arch Neurol Psychiatry 41:535-555, 1939 4. Brown MH: Intraspinal meningiomas. A clinical and pathologic study. Arch Nearol Psychiatry 47:271-292, 1942 5. Buchstein HF: Meningiomas of the spinal cord. Minn Med 24:539-545, 1941 6. Bull JWD: Spinal meningiomas and neurofibromas. Acta Radiol 40:283-300, 1953 7. Cushing HW, Eisenhardt L: Meningiomas, Their Classi-

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fication, Regional Behaviour, Life History, and Surgical End Results. Springfield, Ill: Charles C Thomas, 1938, pp 74-99 Davis RA, Washburn PL: Spinal cord meningiomas. Surg Gynecol Obstet 130:15-21, 1970 Elsberg CA: Extradural spinal tumors; primary, secondary, metastatic. Surg Gynecol Obstet 46:1-20, 1928 Feldenzer JA, McGauley JL, McGillicuddy JE: Sacral and presacral tumors: problems in diagnosis and management. Neurosurgery 25:884-891, 1989 Horrax G, Poppen JL, Wu WQ, et al" Meningiomas and neurofibromas of the spinal cord. Certain clinical features and end results. Surg Clin North Am 29:659-665, 1949 Iraci G, Peserico L, Salar G: Intraspinal neurinomas and meningiomas. A clinical survey of 172 cases. Iut Surg 56: 289-303, 1971 Lesoin F, Krivosic I, Cama A, et al: A giant intrasacral schwannoma revealed by lumbosacral pain. Neurochirargia 27:23-24, 1984 Levy WJ, Bay J, Dohn D: Spinal cord meningioma. J Neurosurg 57:804-812, 1982 Localio SA, Eng K, Ranson JHC: Abdominosacral approach for retrorectal tumors. Ann Surg 191:555-560, 1980 Lombardi G, Passerini A: Spinal cord tumors. Radiology 76:381-392, 1961 Luken MG III, Michelson WJ, Whelan MA, et al: The diagnosis of sacral lesions. Surg Neurol 15:377-383, 1981 MacCarty CS, Waugh JM, Coventry MB, et al: Surgical treatment of sacral and presacral tumors other than sacrococcygeal chordoma. J Nearosurg 22:458-464, 1965 McColl I: The classification of presacral cysts and tumours. Proc R Soc Med 56:797-798, 1963 Namer IJ, Pamir MN, Benli K, et al: Spinal meningiomas. Neurochirurgia 30:11-15, 1987 Norstrom CW, Kernohan JW, Love JG: One hundred primary caudal tumors. JAMA 178:1071-1077, 1961 Oddsson B: Spinal Meningioma. Copenhagen: Einhard Munskgaard, 1947 Rand CW: Surgical experiences with spinal cord tumors. A survey over a forty-year period. Bull Los Angeles Neurol Soc 28:260-268, 1940 Rasmussen TB, Kernohan JW, Adson AW: Pathologic classification, with surgical consideration, of intraspinal tumors. Ann Surg 111:513-530, 1940 Ross ST: Sacral and presacral tumors. Am J Surg 76: 687-693, 1948 Stener B, Gunterberg B: High amputation of the sacrum for extirpation of tumors. Principles and techniques. Spine 3:351-366, 1978 Turner ML, Mulhern CB, Dalinka MK: Lesions of the sacrum. Differential diagnosis and radiological evaluation. JAMA 245:275-277, 1981 Whittaker LD, Pemberton JJ: Tumors ventral to the sacrum. Ann Surg 107:96-106, 1938 Wood JB, Wolpert SM: Lumbosacral meningioma. AJNR 6:450-451, 1985

Manuscript received August 8, 1989. Accepted in final form November 20, 1989. Address reprint requests to: John A. Feldenzer, M.D., Roanoke Neurological Center, 2601 Franklin Road S.W., Roanoke, Virginia 24014.

J. Neurosurg. / Volume 72~June, 1990

Giant sacrolumbar meningioma. Case report.

A case of giant sacral meningioma with presacral and lumbar extension is presented. The difficulties in diagnosis and management are emphasized includ...
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