J Neurosurg 48:645-648, 1978
Spinal subaraehnoid hemorrhage during myelography Case report
JOHN F. RICE, M . D . , CHRISTOPHER B. SHIELDS, M . D . , F.R.C.S.(C), CHARLES F. MORRIS, M.D., AND BYRON D. NEELY, M . D .
Department of Radiology and the Section of Neurological Surgery, Department of
Surgery, University o f Louisville School of Medicine, and the Louisville Veterans Administration Hospital. Louisville, Kentucky
Neoplasms of the conus medullaris and cauda equina have been shown to cause subarachnoid hemorrhage (SAH). In such instances, the hemorrhage has been the presenting symptom. A patient with a neurofibroma at T12-LI experienced spinal SAH during myelography for suspected lumbar disc disease. The roentgenograms showing the lesion itself first and the subarachnoid clot subsequently may be unique. The cause of hemorrhage in this patient may have been the shearing of bridging capsular veins because of diminished cerebrospinal fluid pressure below the almost obstructing lesion. ~ myelography subarachnoid hemorrhage KEY WORDS
R
ECENTLY a patient undergoing examination for suspected lumbar disc disease developed subarachnoid hemorrhage (SAH) during myelogram. This complication of myelography has not been reported previously; its occurrence in association with a neurofibroma at the conus level prompted this report. Case Report
A 50-year-old man was admitted to the Louisville Veterans Administration Hospital in November, 1976, with a 4-year history of thoracolumbar pain. The pain radiated into the left buttock and anteromedial thigh, with extension to the knee. These complaints were worse at night, causing him to sleep in a semiJ. Neurosurg. / Volume 48 / April, 1978
9 neurofibroma
9
Fowler position, and were aggravated by coughing and straining. In November, 1976, the pain progressed to involve the left scrotal and both inguinal areas. There were no complaints in the right leg, nor was there any disturbance of sphincter control. The patient had undergone lumbar discoidectomy at L4-5 in 1954. Examination. Physical examination on admission disclosed pain bilaterally on straightleg raising at 70 ~ with radiation of pain to the lumbar region. Deep tendon reflexes were symmetric and equal, with plantar flexor responses. Sensory and motor examination was unremarkable in the lower limbs. There was loss of lumbar lordosis, bilateral paravertebral muscle spasm, and muscle tenderness. 645
J. F. Rice, C. B. Shields, C. F. Morris and B. D. Neely
FIG. I. Frontal view shows the sharply marginated intradural extramedullary mass at T12-LI (white arrow), displacing the upper cauda equina roots from left.to right (black arrow).
Plain radiographs showed straightening of the lumbar spine but no enlargement of the spinal canal, pediculate erosion, or calcification. Lumbar myelography using Pantopaque (iophendylate) was performed after a careful midline puncture with a No. 20 spinal needle at L3-4. An oval intradural, extramedullary mass, approximately 31/2 cm in length, was seen at T12-L1, producing an almost complete block (Fig. 1). The conus and upper roots of the cauda equina were displaced from left to right. During myelography the patient complained of a dramatic exacerbation in back and groin pain. The pain had an intense burning character and persisted for the next 8 hours, during which time the patient was quite restless. No attempt was made to remove the contrast medium. 646
Cerebrospinal fluid (CSF) samples taken before instillation of contrast material showed slight xanthochromia, with protein level 518 mg%, glucose 34 mg%, and 100 lymphocytes and 37 red blood cells/cu mm. Blood coagulation studies were normal with prothrombin time of 10.9 seconds (normal 12.0 seconds), partial thromboplastin time of 32 seconds (normal 30 to 34 seconds), and hemoglobin of 14.9 gm%, with normal findings on hemoglobin electrophoresis. The patient underwent operation 10 days after myelogram, a delay necessitated by urinary tract infection. On the evening before operation, while the operative level was being marked under fluoroscopic control, an illdefined intradural defect was noted immediately inferior to the previously demonstrated intradural mass (Fig. 2). Operation. Thoracolumbar laminectomy disclosed a firm, typical intradural neurofibroma just inferior and to the left of the conus medullaris. The nerve roots were separated easily from the tumor. A small nerve root was seen passing into the upper pole of the tumor. Immediately caudal and attached to the tumor, also pushing the nerve roots from left to right, was a relatively fresh, solid hemorrhagic clot, approximately 8 ml in volume, which was removed easily in one section. A thrombosed vessel, probably related to the filum terminale, passing through the hemorrhagic mass was thought to represent the source of bleeding. Microscopic evaluation of the tumor demonstrated small foci of necrosis and hemorrhage. The latter was not in continuity with the subarachnoid clot. Postoperatively, the patient has done well with marked improvement in back and leg pain and normal neurological findings in the lower limbs. Discussion
Spinal SAH is an infrequent presentation of spinal cord tumor. The clinical presentation of spinal SAH was first described by Michon 7 and subsequently Andr6-Thomas, et al. 1 The most common cause of spinal SAH is arteriovenous malformation? ,13 The clinical manifestations of such a hemorrhage in patients with spinal cord tumors were characterized as a syndrome by Fincher? The most recent series of spinal neoplasms presenting with spinal SAH lists 31 cases in J. Neurosurg. / Volume 48 / April, 1978
Spinal subaraehnoid hemorrhage during myelography
FIG. 2. Left." Spot film l0 days after myelography shows the irregular filling defect at I_-I (arrowheads) below tumor site. Right: At operation a typical neurofibroma (white arrows) was seen displacing the conus and upper cauda equina from left to right (black arrows). Immediately below the tumor is a blood clot (arrowheads) corresponding to the irregular filling defect (left).
FIG. 3. Left." Before myelography. Diagram shows relationship of tumor, conus, cauda equina, and dura. Right: After myelography the tumor has descended, and the clot is adjacent to the lower pole of the tumor.
J. Neurosurg. / Volume 48 / April, 1978
647
J. F. Rice, C. B. Shields, C. F. Morris and B. D, Neely the world literature." These patients presented with generalized SAH; the spinal origin of the hemorrhages often was unsuspected initially, and only the persistent back and leg pain provoked radiographic examination of the spinal subarachnoid space. 2,8,6''~ The most common tumor types associated with spinal S A H are ependymomas. In none of the reported patients with tumors did myelographic study precede the hemorrhage. Although a traumatic spinal tap may induce some bleeding into the CSF, clinical S A H is not a documented consequence of m y e l o g r a p h y ? 2 The precise etiology .of hemorrhage in this patient is speculative, but h e m o r r h a g e was precipitated by violent physical exertion in several of the 31 cases reported previously. Such hemorrhage has been attributed to the well known mobility of the spinal contents relative to the spinal canal in different positions) Either intrinsically friable tumor vessels or bridging vessels from the tumor capsule to surrounding membranes rupture as a consequence of differential movement of spinal contents relative to the spinal canal? In our patient one of two factors may have been causative (Fig. 3). The introduction of Pantopaque below the almost obstructing lesion may have distended the lumbar theca sufficiently to push the tumor upward. More likely, the spinal puncture caused a minor decrease in C S F pressure below the lesion, and the tumor was pushed caudally by the higher column of C S F pressure above. In either event, bridging vessels, which were identified between the tumor and adjacent membranes, were torn by the shearing force of tumor displacement relative to the surrounding theca. The clot and, presumably, all the subarachnoid bleeding were confined to the subarachnoid space below the tumor. This correlates with the patient's symptoms of a focal spinal S A H rather than a generalized hemorrhage as seen in previously reported cases. References
1. Andr6-Thomas F, Schaeffer H, DeMartel T: Syndrome d'hemorragie m6ning6e r6alis6 par une tumeur de la queue de cheval. Paris Med 77:292-296, 1930 648
2. Bernell WR, Kepes J J, Clough CA: Subarachnoid hemorrhage from malignant schwannoma of cauda equina. Tex Med 69: 101-104, 1973 3. Bhandari YS: Subarachnoid hemorrhage due to cervical cord tumor in a child. Case report. J Neurosurg 30:749-751, 1969 4. Fincher EF: Spontaneous subarachnoid hemorrhage in intradural tumors of the lumbar sac: a clinical syndrome. J Neurosurg 8: 576-584, 1951 5. Grollmus J: Spinal subarachnoid hemorrhage with schwannoma. Acta Neurochir 31:253256, 1975 6. Halpern L, Feldman S, Peyser E: Subarachnoid hemorrhage with papilledema due to spinal neurofibroma. Arch Neurol Psychiatry 79:138-141, 1958 7. Michon P: Le coup de poignard rachidien. Sympt6me initial de certaines h6morragies sous-arachno'idiennes; Essai sur les h6morragies m6ning6es spinales. Presse Med 36: 964-966, 1928 8. Nassar SI, Correll JW: Subarachnoid hemorrhage due to spinal cord tumors. Neurology 18:87-94, 1968 9. Nijensohn DE, Laventman J, Miller RH, et al: Repeated "occult" spinal subarachnoid hemorrhage and spinal cord ependymoma. Minn Med 57:697-699, 1974 10. Prieto A Jr, Cantu RC: Spinal subarachnoid hemorrhage associated with neurofibroma of the cauda equina. Case report. J Neurosurg 27:63-69, 1967 11. Runnels JB, Hanbery JW: Spontaneous subarachnoid hemorrhage associated with spinal cord tumor. Case report. J Neurosurg 40:252-254, 1974 12. Shapiro RS: Myelography, ed 2. Chicago: Year Book Medical Publishers, 1968, pp 455-461 13. Walton JN: Subarachnoid haemorrhage of unusual aetiology. Neurology 3:517-543, 1953
This paper was presented at the 15th Annual Meeting of the American Society of Neuroradiology, Hamilton, Bermuda, March 27-April 1, 1977. Address reprint requests to: Christopher B. Shields, M.D., F.R.C.S.(C), Department of Surgery, University of Louisville School of Medicine, Health Sciences Center, Louisville, Kentucky 40201. d. Neurosurg. / Volume 48 / April, 1978
Spinal subaraehnoid hemorrhage during myelography Case report
JOHN F. RICE, M . D . , CHRISTOPHER B. SHIELDS, M . D . , F.R.C.S.(C), CHARLES F. MORRIS, M.D., AND BYRON D. NEELY, M . D .
Department of Radiology and the Section of Neurological Surgery, Department of
Surgery, University o f Louisville School of Medicine, and the Louisville Veterans Administration Hospital. Louisville, Kentucky
Neoplasms of the conus medullaris and cauda equina have been shown to cause subarachnoid hemorrhage (SAH). In such instances, the hemorrhage has been the presenting symptom. A patient with a neurofibroma at T12-LI experienced spinal SAH during myelography for suspected lumbar disc disease. The roentgenograms showing the lesion itself first and the subarachnoid clot subsequently may be unique. The cause of hemorrhage in this patient may have been the shearing of bridging capsular veins because of diminished cerebrospinal fluid pressure below the almost obstructing lesion. ~ myelography subarachnoid hemorrhage KEY WORDS
R
ECENTLY a patient undergoing examination for suspected lumbar disc disease developed subarachnoid hemorrhage (SAH) during myelogram. This complication of myelography has not been reported previously; its occurrence in association with a neurofibroma at the conus level prompted this report. Case Report
A 50-year-old man was admitted to the Louisville Veterans Administration Hospital in November, 1976, with a 4-year history of thoracolumbar pain. The pain radiated into the left buttock and anteromedial thigh, with extension to the knee. These complaints were worse at night, causing him to sleep in a semiJ. Neurosurg. / Volume 48 / April, 1978
9 neurofibroma
9
Fowler position, and were aggravated by coughing and straining. In November, 1976, the pain progressed to involve the left scrotal and both inguinal areas. There were no complaints in the right leg, nor was there any disturbance of sphincter control. The patient had undergone lumbar discoidectomy at L4-5 in 1954. Examination. Physical examination on admission disclosed pain bilaterally on straightleg raising at 70 ~ with radiation of pain to the lumbar region. Deep tendon reflexes were symmetric and equal, with plantar flexor responses. Sensory and motor examination was unremarkable in the lower limbs. There was loss of lumbar lordosis, bilateral paravertebral muscle spasm, and muscle tenderness. 645
J. F. Rice, C. B. Shields, C. F. Morris and B. D. Neely
FIG. I. Frontal view shows the sharply marginated intradural extramedullary mass at T12-LI (white arrow), displacing the upper cauda equina roots from left.to right (black arrow).
Plain radiographs showed straightening of the lumbar spine but no enlargement of the spinal canal, pediculate erosion, or calcification. Lumbar myelography using Pantopaque (iophendylate) was performed after a careful midline puncture with a No. 20 spinal needle at L3-4. An oval intradural, extramedullary mass, approximately 31/2 cm in length, was seen at T12-L1, producing an almost complete block (Fig. 1). The conus and upper roots of the cauda equina were displaced from left to right. During myelography the patient complained of a dramatic exacerbation in back and groin pain. The pain had an intense burning character and persisted for the next 8 hours, during which time the patient was quite restless. No attempt was made to remove the contrast medium. 646
Cerebrospinal fluid (CSF) samples taken before instillation of contrast material showed slight xanthochromia, with protein level 518 mg%, glucose 34 mg%, and 100 lymphocytes and 37 red blood cells/cu mm. Blood coagulation studies were normal with prothrombin time of 10.9 seconds (normal 12.0 seconds), partial thromboplastin time of 32 seconds (normal 30 to 34 seconds), and hemoglobin of 14.9 gm%, with normal findings on hemoglobin electrophoresis. The patient underwent operation 10 days after myelogram, a delay necessitated by urinary tract infection. On the evening before operation, while the operative level was being marked under fluoroscopic control, an illdefined intradural defect was noted immediately inferior to the previously demonstrated intradural mass (Fig. 2). Operation. Thoracolumbar laminectomy disclosed a firm, typical intradural neurofibroma just inferior and to the left of the conus medullaris. The nerve roots were separated easily from the tumor. A small nerve root was seen passing into the upper pole of the tumor. Immediately caudal and attached to the tumor, also pushing the nerve roots from left to right, was a relatively fresh, solid hemorrhagic clot, approximately 8 ml in volume, which was removed easily in one section. A thrombosed vessel, probably related to the filum terminale, passing through the hemorrhagic mass was thought to represent the source of bleeding. Microscopic evaluation of the tumor demonstrated small foci of necrosis and hemorrhage. The latter was not in continuity with the subarachnoid clot. Postoperatively, the patient has done well with marked improvement in back and leg pain and normal neurological findings in the lower limbs. Discussion
Spinal SAH is an infrequent presentation of spinal cord tumor. The clinical presentation of spinal SAH was first described by Michon 7 and subsequently Andr6-Thomas, et al. 1 The most common cause of spinal SAH is arteriovenous malformation? ,13 The clinical manifestations of such a hemorrhage in patients with spinal cord tumors were characterized as a syndrome by Fincher? The most recent series of spinal neoplasms presenting with spinal SAH lists 31 cases in J. Neurosurg. / Volume 48 / April, 1978
Spinal subaraehnoid hemorrhage during myelography
FIG. 2. Left." Spot film l0 days after myelography shows the irregular filling defect at I_-I (arrowheads) below tumor site. Right: At operation a typical neurofibroma (white arrows) was seen displacing the conus and upper cauda equina from left to right (black arrows). Immediately below the tumor is a blood clot (arrowheads) corresponding to the irregular filling defect (left).
FIG. 3. Left." Before myelography. Diagram shows relationship of tumor, conus, cauda equina, and dura. Right: After myelography the tumor has descended, and the clot is adjacent to the lower pole of the tumor.
J. Neurosurg. / Volume 48 / April, 1978
647
J. F. Rice, C. B. Shields, C. F. Morris and B. D, Neely the world literature." These patients presented with generalized SAH; the spinal origin of the hemorrhages often was unsuspected initially, and only the persistent back and leg pain provoked radiographic examination of the spinal subarachnoid space. 2,8,6''~ The most common tumor types associated with spinal S A H are ependymomas. In none of the reported patients with tumors did myelographic study precede the hemorrhage. Although a traumatic spinal tap may induce some bleeding into the CSF, clinical S A H is not a documented consequence of m y e l o g r a p h y ? 2 The precise etiology .of hemorrhage in this patient is speculative, but h e m o r r h a g e was precipitated by violent physical exertion in several of the 31 cases reported previously. Such hemorrhage has been attributed to the well known mobility of the spinal contents relative to the spinal canal in different positions) Either intrinsically friable tumor vessels or bridging vessels from the tumor capsule to surrounding membranes rupture as a consequence of differential movement of spinal contents relative to the spinal canal? In our patient one of two factors may have been causative (Fig. 3). The introduction of Pantopaque below the almost obstructing lesion may have distended the lumbar theca sufficiently to push the tumor upward. More likely, the spinal puncture caused a minor decrease in C S F pressure below the lesion, and the tumor was pushed caudally by the higher column of C S F pressure above. In either event, bridging vessels, which were identified between the tumor and adjacent membranes, were torn by the shearing force of tumor displacement relative to the surrounding theca. The clot and, presumably, all the subarachnoid bleeding were confined to the subarachnoid space below the tumor. This correlates with the patient's symptoms of a focal spinal S A H rather than a generalized hemorrhage as seen in previously reported cases. References
1. Andr6-Thomas F, Schaeffer H, DeMartel T: Syndrome d'hemorragie m6ning6e r6alis6 par une tumeur de la queue de cheval. Paris Med 77:292-296, 1930 648
2. Bernell WR, Kepes J J, Clough CA: Subarachnoid hemorrhage from malignant schwannoma of cauda equina. Tex Med 69: 101-104, 1973 3. Bhandari YS: Subarachnoid hemorrhage due to cervical cord tumor in a child. Case report. J Neurosurg 30:749-751, 1969 4. Fincher EF: Spontaneous subarachnoid hemorrhage in intradural tumors of the lumbar sac: a clinical syndrome. J Neurosurg 8: 576-584, 1951 5. Grollmus J: Spinal subarachnoid hemorrhage with schwannoma. Acta Neurochir 31:253256, 1975 6. Halpern L, Feldman S, Peyser E: Subarachnoid hemorrhage with papilledema due to spinal neurofibroma. Arch Neurol Psychiatry 79:138-141, 1958 7. Michon P: Le coup de poignard rachidien. Sympt6me initial de certaines h6morragies sous-arachno'idiennes; Essai sur les h6morragies m6ning6es spinales. Presse Med 36: 964-966, 1928 8. Nassar SI, Correll JW: Subarachnoid hemorrhage due to spinal cord tumors. Neurology 18:87-94, 1968 9. Nijensohn DE, Laventman J, Miller RH, et al: Repeated "occult" spinal subarachnoid hemorrhage and spinal cord ependymoma. Minn Med 57:697-699, 1974 10. Prieto A Jr, Cantu RC: Spinal subarachnoid hemorrhage associated with neurofibroma of the cauda equina. Case report. J Neurosurg 27:63-69, 1967 11. Runnels JB, Hanbery JW: Spontaneous subarachnoid hemorrhage associated with spinal cord tumor. Case report. J Neurosurg 40:252-254, 1974 12. Shapiro RS: Myelography, ed 2. Chicago: Year Book Medical Publishers, 1968, pp 455-461 13. Walton JN: Subarachnoid haemorrhage of unusual aetiology. Neurology 3:517-543, 1953
This paper was presented at the 15th Annual Meeting of the American Society of Neuroradiology, Hamilton, Bermuda, March 27-April 1, 1977. Address reprint requests to: Christopher B. Shields, M.D., F.R.C.S.(C), Department of Surgery, University of Louisville School of Medicine, Health Sciences Center, Louisville, Kentucky 40201. d. Neurosurg. / Volume 48 / April, 1978
