The Journal of Emergency Medicine, Vol. 47, No. 5, pp. 552–556, 2014 Copyright Ó 2014 Elsevier Inc. Printed in the USA. All rights reserved 0736-4679/$ - see front matter
http://dx.doi.org/10.1016/j.jemermed.2014.06.030
Selected Topics: Neurological Emergencies
SPONTANEOUS SPINAL SUBDURAL HEMATOMA OF INTRACRANIAL ORIGIN PRESENTING AS BACK PAIN Judy C. Lin, MD and Kerri Layman, MD Emergency Medicine Department, Medstar Georgetown University Hospital and Medstar Washington Hospital Center, Washington, District of Columbia Reprint Address: Judy C. Lin, MD, Emergency Medicine Department, Medstar Georgetown University Hospital and Medstar Washington Hospital Center, 110 Irving Street, NW, Washington, DC 20010
, Abstract—Background: Spinal subdural hematoma (SDH) is an uncommon condition mainly associated with bleeding dyscrasias, use of anticoagulants, trauma, iatrogenic procedures, and vascular malformations. Prompt diagnosis and treatment are recommended to prevent progressive neurologic compromise. Spinal SDH concomitant with intracranial SDH is an even rarer entity, with few cases reported in the English literature. Here we present a case of spontaneous spinal SDH with intracranial SDH presenting as sacral back pain in a 70-year-old man. We also describe the potential mechanism, treatment, and prognosis of concomitant spinal and intracranial SDH. Case Report: We report an unusual case of spontaneous spinal SDH concomitant with intracranial SDH and discuss the epidemiology, clinical presentation, potential etiology, treatment, and prognosis of this disease. Why Should an Emergency Physician Be Aware of This?: Awareness of the association between spinal SDH and intracranial SDH can expedite appropriate imaging of both brain and spine, which can lead to a more complete diagnosis and require changes in patient management in the emergency setting. Ó 2014 Elsevier Inc.
INTRODUCTION Spinal hematomas are rare, but the acute progression of spinal hemorrhage can lead to devastating neurologic sequelae. Therefore, prompt identification and treatment is essential. Spinal subdural hematomas (SDHs) account for < 4.1% of all spinal hematomas and are mainly associated with bleeding dyscrasias, use of anticoagulants, trauma, iatrogenic procedures, and vascular malformations (1). Most present as acute to subacute pain at the level of the spinal hemorrhage and can have neurologic symptoms, including sensory disturbance, progressive weakness, urine and stool retention or incontinence, and paralysis. To our knowledge spinal SDH concomitant with intracranial SDH is even rarer, with cases more commonly a result of trauma and less commonly due to coagulopathy or without any identifiable cause (2 27). Here we present an unusual case of spontaneous spinal SDH concomitant with intracranial SDH followed by a discussion on the epidemiology, clinical presentation, potential etiology, treatment, and prognosis of this disease.
, Keywords—spinal hematoma; intracranial hemorrhage; subdural hematoma; spontaneous hemorrhage; back pain
CASE REPORT A 70-year-old man presented to the emergency department with a chief complaint of back pain radiating down to his toes and bilateral lower-extremity weakness for 5 days. Medical history was significant
Institutional Review Board exemption was assumed because this study was a case report without any personal identifiers.
RECEIVED: 2 October 2013; FINAL SUBMISSION RECEIVED: 16 May 2014; ACCEPTED: 30 June 2014 552
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for hypertension, hyperlipidemia, benign prostatic hypertrophy, metastatic non small cell lung cancer diagnosed in 2007 with metastasis to the left adrenal gland status post chemotherapy, radiation therapy, left adrenalectomy, and left lower lobectomy in 2008, and partial colectomy in 2006. The patient was on tamsulosin 0.4 mg daily. There was no history of trauma or use of aspirin, clopidogrel, or warfarin. Review of systems was positive for urinary hesitancy and mild, chronic headaches, and negative for fevers, chills, nausea, vomiting, visual changes, numbness, or incontinence. The patient’s initial temperature was 36 C, heart rate was 74 beats/min, blood pressure was 167/82 mm Hg, respiratory rate was 19 breaths/min, and oxygen saturation was 96% on room air. The patient was alert and oriented to person, place, and time. Cardiovascular, pulmonary, and abdominal examinations were normal. On neurologic examination, pupils were 4 to 2 mm bilaterally, extraocular movements were intact, cranial nerves were intact with normal deep tendon reflexes, strength was 5 out of 5 bilaterally, and sensation was normal to light touch, temperature, and vibration. Back examination revealed exquisite tenderness to palpation at the tailbone. Rectal examination showed good strength and tone with a nontender prostate. The patient was given morphine 4 mg i.v. for pain. White blood cell count was 8500/mL (reference range 4,000 to 10,800/mL), with a neutrophil percentage of 84.1% (reference range 43% to 75%). Platelets were 189,000/mL (reference range 145,000 to 400,000/mL). Electrolytes and hemoglobin were normal. Urinalysis showed a specific gravity of 1.014, but was otherwise unremarkable. International normalized ratio was 1.1 (reference range 0.8 to 1.2). Given the patient’s cancer history, magnetic resonance imaging (MRI) of the lumbosacral spine with and without gadolinium was ordered to evaluate for metastases. Review of the MRI by neuroradiology revealed an SDH along the posterior spinal canal from L4 to S1 with resultant tapered compression of the thecal sac at these levels with moderate to severe narrowing and nerve root compression at L5 to S1 (Figure 1). Consequently, a computed tomography (CT) of the head was ordered, which showed bilateral convexity SDHs, right larger than left, without significant midline shift, as well as mild mass effect upon underlying parenchyma, lateral, and third ventricles (Figure 2). The patient was admitted, placed on levetiracetam 500 mg twice daily, and bilateral subdural drains were placed by the neurosurgical service. The spinal SDH was conservatively managed. The subdural drains were removed after repeat head CT showed decreased SDHs. Serial head CTs showed moderate resolution of the SDHs. The patient’s pain decreased and he was discharged on levetiracetam 500 mg twice daily 4 days after initial admission.
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Figure 1. Sagittal T1-weighted magnetic resonance imaging of lumbar spine without contrast with arrow indicating area of high-signal intensity at L4 to S1 consistent with subdural hematoma.
Figure 2. Axial head computed tomography without contrast performed after admission showing bilateral subdural hematomas.
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One month later, the patient presented to the emergency department with 1 week of increasing gait instability and mild headache. The patient was found to have acute on chronic SDH with mass effect and shift and underwent right craniotomy for hematoma evacuation. Postoperative head CT showed adequate removal of the clot. The patient’s pain and ambulation improved, and he was later discharged. DISCUSSION Nontraumatic spontaneous spinal SDHs are rare and can cause compression of the spinal cord or cauda equina. Cases have been reported in ages 1 through 79 years, with almost half presenting between ages 45 and 60 years (1). There is no sex difference, and most occur in the thoracolumbar and lumbar regions (1,29). More than half of these cases are due to anticoagulant use and iatrogenic procedures, such as lumbar punctures and peridural anesthesia (29). However, in a study done by Domenicucci et al., a significant number of patients had other predisposing factors: 19% had disorders of hemostasis, such as leukemia, hemophilia, thrombocytopenia, cryoglobulinemia, and polycythemia; 4% had an intradural tumor; 4% had a ruptured arteriovenous malformation; and 14% had no identifiable cause (29). The exact mechanism of nontraumatic spontaneous spinal SDHs is unclear because the spinal subdural space is relatively avascular and lacks bridging veins found in the intracranial space (1,28,29). Two origins of hemorrhage in the spinal subdural space have been proposed. In the first theory, rapid changes in intra-abdominal and thoracic pressures in combination with a lag in cerebrospinal fluid pressure generates a pressure gradient that causes subarachnoid vessels to rupture and dissect into the subdural space. Blood in the subarachnoid space is then cleared by cerebrospinal fluid, leaving only blood in the subdural space (1,30 32). The risk of vessel rupture and dissection is further increased with iatrogenic procedures, disorders of hemostasis, and use of anticoagulants. In the second theory, intracranial hemorrhage in the subdural space extends and migrates as a result of gravity to the spinal subdural space, causing compression of the spinal cord (11,29). Continuity of these two spaces has been demonstrated by postmortem studies in which injection of air into the subdural spinal space was found later in the subdural intracranial space (33). This continuity is further supported by MRI evidence of hemorrhagic collections connecting these two spaces (11). Our patient reported no trauma or invasive procedures, was not on anticoagulation, and his laboratory values did not reveal any hemostatic abnormalities. Therefore, we postulate migration of intracranial subdural blood into the lumbar spine led to the spinal SDH.
Spinal SDHs present first as acute, severe localized back pain followed by radicular pain radiating to the trunk or upper or lower extremities (34). A review of case reports reveals that the radicular pain is most commonly bilateral (2,6,17,24). Within hours or, less commonly, days, varying degrees of neurologic symptoms develop, including paresthesias, sensory loss, weakness, and cauda equina compression causing bowel or bladder dysfunction and paraplegia (28). In traumatic spinal SDHs, symptoms develop immediately after the injury. However, in case reports describing spontaneous spinal SDH concomitant with intracranial SDH, the time to development of symptoms ranges from a few days to 1 week for acute spinal SDH and up to 3 weeks for chronic spinal SDH (10,13,14,17,19). We hypothesize this delay may be the time period during which the effect of gravity causes intracranial blood to migrate to the more gravity-dependent thoracolumbar spinal subdural space. Nontraumatic spontaneous intracranial SDHs are most commonly caused by bleeding due to shearing forces on bridging veins or venous sinuses. Patients with cerebral atrophy, low intracranial pressure, coagulopathies, or alcoholic hepatopathy are at increased risk of bridging vein rupture (35). Other less common causes include aneurysm or arteriovenous malformation rupture, hypertensive hemorrhage into the subdural space, dura mater tumor hemorrhage, and cortical artery rupture (36). Both intracranial and spinal SDHs are surgical emergencies. MRI is the recommended method of imaging for spinal SDH, as it more accurately evaluates the size and extent of hemorrhage and differentiates subdural from epidural hematoma. Non-contrast head CT is recommended for intracranial SDH, although brain MRI can also evaluate presence of SDH. Patients with large or acute intracranial SDH or with progressive neurologic deterioration should be treated with craniotomy and evacuation of the hematoma. Patients with spinal SDH who have stable, minimal sensorimotor deficit without cauda equina symptoms can be conservatively managed, while surgical decompression is recommended for those with progressive and severe sensorimotor impairment and cauda equina symptoms (5,11,17,27). Spinal SDH outcomes are related to the severity, location, and timing of surgical decompression (1,37). Prognosis is poor for severe neurologic deficit, such as paralysis, for cervical or thoracic lesions, and for symptoms longer than 3 months. Prognosis is good for minimal neurologic impairment, for lumbar lesions, and when there is surgical intervention before permanent paralysis (4,22). There are several case reports of concomitant intracranial and spinal SDHs with stable neurologic examinations managed conservatively with good recovery, including our patient (4 7,11,16,18 20,23,25,26,29).
Spontaneous Spinal Subdural Hematoma
A review of the literature indicates that most patients with concomitant intracranial and spinal SDH have symptoms of both headache and back pain. However, it might be possible that we are underestimating the incidence of this condition if imaging studies are done of only the head or the spine. Wong et al. presented a case report of a patient who had intracranial SDH but an asymptomatic spinal SDH found on screening MRI (38). Kokubo et al. performed spinal MRI screening on 168 patients with intracranial SDH and found that 2 patients had concomitant intracranial and spinal SDH (9). Both had direct head and spinal trauma and neither had neurologic deficits. The case report and study suggest that screening for spinal SDH in patients with intracranial SDH in patients without back pain or sensorimotor deficits would likely not change management, as these asymptomatic SDHs would most likely do well with observation. Although asymptomatic spinal SDH can exist with intracranial SDH, the management of these patients without any symptoms of back pain or sensorimotor deficits would still be observation. However, if a patient has suspected or confirmed intracranial SDH and has developed new-onset back pain or any neurologic deficits, then MRI imaging of the spine should be considered to exclude the diagnosis of spinal SDH. Symptomatic patients might progress and need screening for hemostatic abnormalities and interventions such as surgical decompression and anticoagulant reversal. Conversely, we can also ask whether finding a spinal SDH necessitates imaging of the brain. No recommendations exist regarding this question. Some case reports describe patients in whom the spinal SDH was found before the intracranial SDH. Imaging of the brain was prompted by patient confusion or complaints of increased headache, nausea, or vomiting (11,14,16). In two of these cases, brain imaging might have been delayed because of the chronic nature of headache and lack of confusion or cranial nerve deficits. In all cases, the intracranial SDH was treated with craniotomy. Therefore, when you diagnose or are suspicious of spinal SDH, it seems prudent to identify if the patient also has headache, nausea, or vomiting. If intracranial symptoms are present, CT or MRI should be included in the workup. In patients with spinal SDH but without intracranial symptoms, there are no recommendations regarding brain imaging. Jibu et al. describe a patient who had spinal SDH with an asymptomatic intracranial SDH found on screening brain MRI (6). However, this patient’s intracranial SDH was managed conservatively and the patient did well. In patients with intracranial SDH, mortality rates are higher for those on anticoagulation than those who are not (39,40). For such patients, a panel of experts on stroke, neurologic intensive care, and hematology
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recommend urgent reversal of warfarin, but do not come to a consensus as to the best reversal agent. Three experts recommend prothrombin complex concentrates (PCC) only, two recommend recombinant factor VIIa (rFVIIa) only, one expert recommends rFVIIa with fresh frozen plasma (FFP), and one expert recommends either PCC or FFP (39). There are no randomized controlled trials evaluating the clinical outcomes of different reversal strategies for patients on warfarin with intracranial hemorrhage (39). Vitamin K should be given as well, although it does not normalize the international normalized ratio (INR) for 6 to 24 h. FFP can also take several hours to normalize the INR in addition to requiring 2 to 4 L volume for full reversal (39). PCCs normalize INR faster than FFP, but there are no data showing an effect on patient outcomes. Finally, rFVIIa has been shown to decrease the INR in minutes. However, rFVIIa is expensive, has a short half-life, and, after its administration, the INR might not be accurately followed to guide treatment (39). Given that even small amounts of blood can cause permanent neurologic injury in the spinal subdural space, most reported cases of patients with symptomatic spinal SDH on anticoagulation also received anticoagulation reversal (34,41,42). WHY SHOULD AN EMERGENCY PHYSICIAN BE AWARE OF THIS? This case report reveals an interesting and rare case of intracranial SDH presenting as lower back pain. In our patient, an MRI demonstrated spinal SDH and additional investigation revealed intracranial SDH. It is postulated that blood in the intracranial subdural space can migrate due to gravity and cause spinal SDH. A diagnosis of spinal SDH and knowledge of this mechanism should prompt the emergency physician to inquire about a history of headaches, nausea, or vomiting, which, if present, should heighten suspicion for intracranial pathology (28). Likewise, spinal SDH should be a differential diagnosis in a patient with a recent history of intracranial SDH who presents with back pain or sensorimotor deficits. Awareness of concomitant intracranial and spinal SDH can aid in a more timely and complete diagnosis, and can ultimately change management in emergency department patients requiring anticoagulant reversal or surgical intervention. REFERENCES 1. Kreppel D, Antoniadis G, Seeling W. Spinal hematoma: a literature survey with meta-analysis of 613 patients. Neurosurg Rev 2003;26: 1–49. 2. Bartolotti C, Wang H, Fraser K, et al. Subacute spinal subdural hematoma after spontaneous resolution of cranial subdural hematoma: causal relationship or coincidence? J Neurosurg 2004;100:372–4.
556 3. Hagihara N, Abe T, Kojima K, et al. Coexistence of cranial and spinal subdural hematomas: case report. Neurol Med Chir (Tokyo) 2010;50:333–5. 4. Hung KS, Lui CC, Wang CH, et al. Traumatic spinal subdural hematoma with spontaneous resolution. Spine 2002;27:E534–8. 5. Jain V, Singh J, Sharma R. Spontaneous concomitant cranial and spinal subdural haematomas with spontaneous resolution. Singapore Med J 2008;49:e53–8. 6. Jibu KJ, Pranesh MB, Prakash B, et al. Bilateral intracranial and spinal subdural hematoma presenting as bilateral sciatica. J Neurosci Rural Pract 2012;3:97–8. 7. Kim K, Katsuno M, Isu T, et al. concomitant cranial and lumbar subdural hematomas: case report. Neurol Med Chir (Tokyo) 2010;50: 402–4. 8. Kim MS, Chung CK, Hur JW, et al. Spinal subdural hematoma following craniotomy: case report. Surg Neurol 2004;61:288–92. 9. Kokubo R, Kim K, Mishina M, et al. Prospective assessment of concomitant lumbar and chronic subdural hematoma: is migration from the intracranial space involved in their manifestation? J Neurosurg Spine 2014;20:157–63. 10. Leber KA, Pendi G, Kogler S, et al. Simultaneous spinal and intracranial chronic subdural hematoma. J Neurosurg 1997;87:644. 11. Lecouvet FE, Annet L, Duprez TP, et al. Uncommon magnetic resonance imaging observation of lumbar subdural hematoma with cranial origin. J Comput Assist Tomogr 2003;27:530–3. 12. Lee TH, Su TM, Wang KW, et al. Lumbosacral spinal subdural hematoma following burr hole craniotomy: case report and literature review. Clin Neurol Neurosurg 2007;109:282–96. 13. Liu J, Wu B, Feng H, et al. Spinal subdural hematoma following cranial surgery: a case report and review of the literature. Neurol India 2011;59:281–4. 14. Moon W, Joo W, Chough J, et al. Spontaneous spinal subdural hematoma concurrent with cranial subdural hematoma. J Korean Neurosurg Soc 2013;54:68–70. 15. Morishige M, Abe T, Ishii K, et al. Spontaneous chronic head and spinal subdural haematoma. Acta Neurochir (Wien) 2007;149: 1081–2. 16. Nagashima H, Tanida A, Hayashi I, et al. Spinal subdural haematoma concurrent with cranial subdural haematoma: report of two cases and review of literature. Br J Neurosurg 2010;24:537–41. 17. Sari A, Sert B, Dinc H, et al. Subacute spinal subdural hematoma associated with intracranial subdural hematoma. J Neuroradiol 2006;33:67–9. 18. Silver JM, Wilkins RH. Spinal subdural hematoma formation following ventriculoperitoneal shunting for hydrocephalus. Acta Neurochir (Wien) 1991;108:159–62. 19. Shimuzu S, Tachibana S, Maezawa H, et al. Lumbar spinal subdural hematoma following craniotomy: case report. Neurol Med Chir (Tokyo) 1999;39:299–301. 20. Sirin NG, Yesilot N, Ekizoglu E, et al. A case report of cerebral venous thrombosis in polycythemia vera presenting with intracranial and spinal subdural hematoma. Case Rep Neurol 2010; 2:37–45. 21. Swann KW, Ropper AH, New PFJ, et al. Spontaneous spinal subarachnoid hemorrhage and subdural hematoma. J Neurosurg 1984;61:975–80.
J. C. Lin and K. Layman 22. Tillich M, Kammerhuber F, Reittner P, et al. Chronic spinal subdural haematoma associated with intracranial subdural hematoma: CT and MRI. Neuroradiology 1999;41:137–9. 23. Wajima D, Yokota H, Ida Y, et al. Spinal subdural hematoma associated with traumatic intracranial interhemispheric subdural hematoma. Neurol Med Chir (Tokyo) 2012;52:636–9. 24. Wang US, Ju CI, Kim SW, et al. Spontaneous concomitant intracranial and spinal subdural hematomas in association with anticoagulation therapy. J Korean Neurosurg Soc 2012;51:237–9. 25. Wurm G, Pogady P, Lungenschmid K, et al. Subdural hemorrhage of cauda equina. A rare complication of cerebrospinal fluid shunt. Case report. Neurosurg Rev 1996;19:113–7. 26. Yamaguchi S, Kurisu K, Arita K, et al. Simultaneous cranial and spinal subdural hematoma. Neurol Med Chir (Tokyo) 2005;45:645–9. 27. Yang MS, Tung YW, Yang TH, et al. Spontaneous spinal and intracranial subdural hematoma. J Formos Med Assoc 2009;108:258–61. 28. Russell NA, Benoit BG. Spinal subdural hematoma: a review. Surg Neurol 1983;20:133–7. 29. Domenicucci M, Ramieri A, Ciappetta P, et al. Nontraumatic acute spinal subdural hematoma: report of five cases and review of the literature. J Neurosurg 1999;91:65–73. 30. Morandi X, Riffaud L, Chabert E, et al. Acute nontraumatic spinal subdural hematomas in three patients. Spine (Phila Pa 1976) 2001; 26:E547–51. 31. Masdeu JC, Breuer AC, Schoene WC. Spinal subarachnoid hematomas: clues to a source of blooding in traumatic lumbar puncture. Neurology 1979;29:872–6. 32. Rader JP. Chronic subdural hematoma of the spinal cord: report of a case. N Engl J Med 1955;374–6. 33. Stone RS, Jones OW. Encephalopathy: a review of 113 cases. And a report of postmortem studies on the injection of air. Radiology 1933;21:411–9. 34. Mattle H, Sieb JP, Rohner M, et al. Nontraumatic spinal epidural and subdural hematomas. Neurology 1987;37:1351–6. 35. Markwalder TM. Chronic subdural hematomas: a review. J Neurosurg 1981;54:637–45. 36. McDermott M, Fleming JFR, Vanderlinden RG, et al. Spontaneous arterial subdural hematoma. Neurosurgery 1984;14:13–8. 37. Groen RJM, van Alpen HAM. Operative treatment of spontaneous spinal epidural hematomas: a study of the factors determining postoperative outcome. Neurosurgery 1996;39:494–508. 38. Wong ST, Yuen MK, Fok KF, et al. Redistribution of hematoma to spinal subdural space as a mechanism for the rapid spontaneous resolution of posttraumatic intracranial acute subdural hematoma: case report. Surg Neurol 2009;71:99–102. 39. Aguilar MI, Hart RG, Kase CS, et al. Treatment of warfarinassociated intracerebral hemorrhage: literature review and expert opinion. Mayo Clin Proc 2007;8:387. 40. Ivascu F, Howells GA, Junn FS, et al. Rapid warfarin reversal in anticoagulated patients with traumatic intracranial hemorrhage reduces hemorrhage progression and mortality. J Trauma 2005;59:1131–9. 41. Miller DR, Ray A, Hourihan MD. Spinal subdural haematoma: how relevant is the INR? Spinal Cord 2004;42:477–80. 42. Haji Mohd Yasin NA, Donato-Brown D, Taha A. Non-traumatic spontaneous spinal subdural haematoma. N Z Med J 2012;125: 77–80.
http://dx.doi.org/10.1016/j.jemermed.2014.06.030
Selected Topics: Neurological Emergencies
SPONTANEOUS SPINAL SUBDURAL HEMATOMA OF INTRACRANIAL ORIGIN PRESENTING AS BACK PAIN Judy C. Lin, MD and Kerri Layman, MD Emergency Medicine Department, Medstar Georgetown University Hospital and Medstar Washington Hospital Center, Washington, District of Columbia Reprint Address: Judy C. Lin, MD, Emergency Medicine Department, Medstar Georgetown University Hospital and Medstar Washington Hospital Center, 110 Irving Street, NW, Washington, DC 20010
, Abstract—Background: Spinal subdural hematoma (SDH) is an uncommon condition mainly associated with bleeding dyscrasias, use of anticoagulants, trauma, iatrogenic procedures, and vascular malformations. Prompt diagnosis and treatment are recommended to prevent progressive neurologic compromise. Spinal SDH concomitant with intracranial SDH is an even rarer entity, with few cases reported in the English literature. Here we present a case of spontaneous spinal SDH with intracranial SDH presenting as sacral back pain in a 70-year-old man. We also describe the potential mechanism, treatment, and prognosis of concomitant spinal and intracranial SDH. Case Report: We report an unusual case of spontaneous spinal SDH concomitant with intracranial SDH and discuss the epidemiology, clinical presentation, potential etiology, treatment, and prognosis of this disease. Why Should an Emergency Physician Be Aware of This?: Awareness of the association between spinal SDH and intracranial SDH can expedite appropriate imaging of both brain and spine, which can lead to a more complete diagnosis and require changes in patient management in the emergency setting. Ó 2014 Elsevier Inc.
INTRODUCTION Spinal hematomas are rare, but the acute progression of spinal hemorrhage can lead to devastating neurologic sequelae. Therefore, prompt identification and treatment is essential. Spinal subdural hematomas (SDHs) account for < 4.1% of all spinal hematomas and are mainly associated with bleeding dyscrasias, use of anticoagulants, trauma, iatrogenic procedures, and vascular malformations (1). Most present as acute to subacute pain at the level of the spinal hemorrhage and can have neurologic symptoms, including sensory disturbance, progressive weakness, urine and stool retention or incontinence, and paralysis. To our knowledge spinal SDH concomitant with intracranial SDH is even rarer, with cases more commonly a result of trauma and less commonly due to coagulopathy or without any identifiable cause (2 27). Here we present an unusual case of spontaneous spinal SDH concomitant with intracranial SDH followed by a discussion on the epidemiology, clinical presentation, potential etiology, treatment, and prognosis of this disease.
, Keywords—spinal hematoma; intracranial hemorrhage; subdural hematoma; spontaneous hemorrhage; back pain
CASE REPORT A 70-year-old man presented to the emergency department with a chief complaint of back pain radiating down to his toes and bilateral lower-extremity weakness for 5 days. Medical history was significant
Institutional Review Board exemption was assumed because this study was a case report without any personal identifiers.
RECEIVED: 2 October 2013; FINAL SUBMISSION RECEIVED: 16 May 2014; ACCEPTED: 30 June 2014 552
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for hypertension, hyperlipidemia, benign prostatic hypertrophy, metastatic non small cell lung cancer diagnosed in 2007 with metastasis to the left adrenal gland status post chemotherapy, radiation therapy, left adrenalectomy, and left lower lobectomy in 2008, and partial colectomy in 2006. The patient was on tamsulosin 0.4 mg daily. There was no history of trauma or use of aspirin, clopidogrel, or warfarin. Review of systems was positive for urinary hesitancy and mild, chronic headaches, and negative for fevers, chills, nausea, vomiting, visual changes, numbness, or incontinence. The patient’s initial temperature was 36 C, heart rate was 74 beats/min, blood pressure was 167/82 mm Hg, respiratory rate was 19 breaths/min, and oxygen saturation was 96% on room air. The patient was alert and oriented to person, place, and time. Cardiovascular, pulmonary, and abdominal examinations were normal. On neurologic examination, pupils were 4 to 2 mm bilaterally, extraocular movements were intact, cranial nerves were intact with normal deep tendon reflexes, strength was 5 out of 5 bilaterally, and sensation was normal to light touch, temperature, and vibration. Back examination revealed exquisite tenderness to palpation at the tailbone. Rectal examination showed good strength and tone with a nontender prostate. The patient was given morphine 4 mg i.v. for pain. White blood cell count was 8500/mL (reference range 4,000 to 10,800/mL), with a neutrophil percentage of 84.1% (reference range 43% to 75%). Platelets were 189,000/mL (reference range 145,000 to 400,000/mL). Electrolytes and hemoglobin were normal. Urinalysis showed a specific gravity of 1.014, but was otherwise unremarkable. International normalized ratio was 1.1 (reference range 0.8 to 1.2). Given the patient’s cancer history, magnetic resonance imaging (MRI) of the lumbosacral spine with and without gadolinium was ordered to evaluate for metastases. Review of the MRI by neuroradiology revealed an SDH along the posterior spinal canal from L4 to S1 with resultant tapered compression of the thecal sac at these levels with moderate to severe narrowing and nerve root compression at L5 to S1 (Figure 1). Consequently, a computed tomography (CT) of the head was ordered, which showed bilateral convexity SDHs, right larger than left, without significant midline shift, as well as mild mass effect upon underlying parenchyma, lateral, and third ventricles (Figure 2). The patient was admitted, placed on levetiracetam 500 mg twice daily, and bilateral subdural drains were placed by the neurosurgical service. The spinal SDH was conservatively managed. The subdural drains were removed after repeat head CT showed decreased SDHs. Serial head CTs showed moderate resolution of the SDHs. The patient’s pain decreased and he was discharged on levetiracetam 500 mg twice daily 4 days after initial admission.
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Figure 1. Sagittal T1-weighted magnetic resonance imaging of lumbar spine without contrast with arrow indicating area of high-signal intensity at L4 to S1 consistent with subdural hematoma.
Figure 2. Axial head computed tomography without contrast performed after admission showing bilateral subdural hematomas.
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One month later, the patient presented to the emergency department with 1 week of increasing gait instability and mild headache. The patient was found to have acute on chronic SDH with mass effect and shift and underwent right craniotomy for hematoma evacuation. Postoperative head CT showed adequate removal of the clot. The patient’s pain and ambulation improved, and he was later discharged. DISCUSSION Nontraumatic spontaneous spinal SDHs are rare and can cause compression of the spinal cord or cauda equina. Cases have been reported in ages 1 through 79 years, with almost half presenting between ages 45 and 60 years (1). There is no sex difference, and most occur in the thoracolumbar and lumbar regions (1,29). More than half of these cases are due to anticoagulant use and iatrogenic procedures, such as lumbar punctures and peridural anesthesia (29). However, in a study done by Domenicucci et al., a significant number of patients had other predisposing factors: 19% had disorders of hemostasis, such as leukemia, hemophilia, thrombocytopenia, cryoglobulinemia, and polycythemia; 4% had an intradural tumor; 4% had a ruptured arteriovenous malformation; and 14% had no identifiable cause (29). The exact mechanism of nontraumatic spontaneous spinal SDHs is unclear because the spinal subdural space is relatively avascular and lacks bridging veins found in the intracranial space (1,28,29). Two origins of hemorrhage in the spinal subdural space have been proposed. In the first theory, rapid changes in intra-abdominal and thoracic pressures in combination with a lag in cerebrospinal fluid pressure generates a pressure gradient that causes subarachnoid vessels to rupture and dissect into the subdural space. Blood in the subarachnoid space is then cleared by cerebrospinal fluid, leaving only blood in the subdural space (1,30 32). The risk of vessel rupture and dissection is further increased with iatrogenic procedures, disorders of hemostasis, and use of anticoagulants. In the second theory, intracranial hemorrhage in the subdural space extends and migrates as a result of gravity to the spinal subdural space, causing compression of the spinal cord (11,29). Continuity of these two spaces has been demonstrated by postmortem studies in which injection of air into the subdural spinal space was found later in the subdural intracranial space (33). This continuity is further supported by MRI evidence of hemorrhagic collections connecting these two spaces (11). Our patient reported no trauma or invasive procedures, was not on anticoagulation, and his laboratory values did not reveal any hemostatic abnormalities. Therefore, we postulate migration of intracranial subdural blood into the lumbar spine led to the spinal SDH.
Spinal SDHs present first as acute, severe localized back pain followed by radicular pain radiating to the trunk or upper or lower extremities (34). A review of case reports reveals that the radicular pain is most commonly bilateral (2,6,17,24). Within hours or, less commonly, days, varying degrees of neurologic symptoms develop, including paresthesias, sensory loss, weakness, and cauda equina compression causing bowel or bladder dysfunction and paraplegia (28). In traumatic spinal SDHs, symptoms develop immediately after the injury. However, in case reports describing spontaneous spinal SDH concomitant with intracranial SDH, the time to development of symptoms ranges from a few days to 1 week for acute spinal SDH and up to 3 weeks for chronic spinal SDH (10,13,14,17,19). We hypothesize this delay may be the time period during which the effect of gravity causes intracranial blood to migrate to the more gravity-dependent thoracolumbar spinal subdural space. Nontraumatic spontaneous intracranial SDHs are most commonly caused by bleeding due to shearing forces on bridging veins or venous sinuses. Patients with cerebral atrophy, low intracranial pressure, coagulopathies, or alcoholic hepatopathy are at increased risk of bridging vein rupture (35). Other less common causes include aneurysm or arteriovenous malformation rupture, hypertensive hemorrhage into the subdural space, dura mater tumor hemorrhage, and cortical artery rupture (36). Both intracranial and spinal SDHs are surgical emergencies. MRI is the recommended method of imaging for spinal SDH, as it more accurately evaluates the size and extent of hemorrhage and differentiates subdural from epidural hematoma. Non-contrast head CT is recommended for intracranial SDH, although brain MRI can also evaluate presence of SDH. Patients with large or acute intracranial SDH or with progressive neurologic deterioration should be treated with craniotomy and evacuation of the hematoma. Patients with spinal SDH who have stable, minimal sensorimotor deficit without cauda equina symptoms can be conservatively managed, while surgical decompression is recommended for those with progressive and severe sensorimotor impairment and cauda equina symptoms (5,11,17,27). Spinal SDH outcomes are related to the severity, location, and timing of surgical decompression (1,37). Prognosis is poor for severe neurologic deficit, such as paralysis, for cervical or thoracic lesions, and for symptoms longer than 3 months. Prognosis is good for minimal neurologic impairment, for lumbar lesions, and when there is surgical intervention before permanent paralysis (4,22). There are several case reports of concomitant intracranial and spinal SDHs with stable neurologic examinations managed conservatively with good recovery, including our patient (4 7,11,16,18 20,23,25,26,29).
Spontaneous Spinal Subdural Hematoma
A review of the literature indicates that most patients with concomitant intracranial and spinal SDH have symptoms of both headache and back pain. However, it might be possible that we are underestimating the incidence of this condition if imaging studies are done of only the head or the spine. Wong et al. presented a case report of a patient who had intracranial SDH but an asymptomatic spinal SDH found on screening MRI (38). Kokubo et al. performed spinal MRI screening on 168 patients with intracranial SDH and found that 2 patients had concomitant intracranial and spinal SDH (9). Both had direct head and spinal trauma and neither had neurologic deficits. The case report and study suggest that screening for spinal SDH in patients with intracranial SDH in patients without back pain or sensorimotor deficits would likely not change management, as these asymptomatic SDHs would most likely do well with observation. Although asymptomatic spinal SDH can exist with intracranial SDH, the management of these patients without any symptoms of back pain or sensorimotor deficits would still be observation. However, if a patient has suspected or confirmed intracranial SDH and has developed new-onset back pain or any neurologic deficits, then MRI imaging of the spine should be considered to exclude the diagnosis of spinal SDH. Symptomatic patients might progress and need screening for hemostatic abnormalities and interventions such as surgical decompression and anticoagulant reversal. Conversely, we can also ask whether finding a spinal SDH necessitates imaging of the brain. No recommendations exist regarding this question. Some case reports describe patients in whom the spinal SDH was found before the intracranial SDH. Imaging of the brain was prompted by patient confusion or complaints of increased headache, nausea, or vomiting (11,14,16). In two of these cases, brain imaging might have been delayed because of the chronic nature of headache and lack of confusion or cranial nerve deficits. In all cases, the intracranial SDH was treated with craniotomy. Therefore, when you diagnose or are suspicious of spinal SDH, it seems prudent to identify if the patient also has headache, nausea, or vomiting. If intracranial symptoms are present, CT or MRI should be included in the workup. In patients with spinal SDH but without intracranial symptoms, there are no recommendations regarding brain imaging. Jibu et al. describe a patient who had spinal SDH with an asymptomatic intracranial SDH found on screening brain MRI (6). However, this patient’s intracranial SDH was managed conservatively and the patient did well. In patients with intracranial SDH, mortality rates are higher for those on anticoagulation than those who are not (39,40). For such patients, a panel of experts on stroke, neurologic intensive care, and hematology
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recommend urgent reversal of warfarin, but do not come to a consensus as to the best reversal agent. Three experts recommend prothrombin complex concentrates (PCC) only, two recommend recombinant factor VIIa (rFVIIa) only, one expert recommends rFVIIa with fresh frozen plasma (FFP), and one expert recommends either PCC or FFP (39). There are no randomized controlled trials evaluating the clinical outcomes of different reversal strategies for patients on warfarin with intracranial hemorrhage (39). Vitamin K should be given as well, although it does not normalize the international normalized ratio (INR) for 6 to 24 h. FFP can also take several hours to normalize the INR in addition to requiring 2 to 4 L volume for full reversal (39). PCCs normalize INR faster than FFP, but there are no data showing an effect on patient outcomes. Finally, rFVIIa has been shown to decrease the INR in minutes. However, rFVIIa is expensive, has a short half-life, and, after its administration, the INR might not be accurately followed to guide treatment (39). Given that even small amounts of blood can cause permanent neurologic injury in the spinal subdural space, most reported cases of patients with symptomatic spinal SDH on anticoagulation also received anticoagulation reversal (34,41,42). WHY SHOULD AN EMERGENCY PHYSICIAN BE AWARE OF THIS? This case report reveals an interesting and rare case of intracranial SDH presenting as lower back pain. In our patient, an MRI demonstrated spinal SDH and additional investigation revealed intracranial SDH. It is postulated that blood in the intracranial subdural space can migrate due to gravity and cause spinal SDH. A diagnosis of spinal SDH and knowledge of this mechanism should prompt the emergency physician to inquire about a history of headaches, nausea, or vomiting, which, if present, should heighten suspicion for intracranial pathology (28). Likewise, spinal SDH should be a differential diagnosis in a patient with a recent history of intracranial SDH who presents with back pain or sensorimotor deficits. Awareness of concomitant intracranial and spinal SDH can aid in a more timely and complete diagnosis, and can ultimately change management in emergency department patients requiring anticoagulant reversal or surgical intervention. REFERENCES 1. Kreppel D, Antoniadis G, Seeling W. Spinal hematoma: a literature survey with meta-analysis of 613 patients. Neurosurg Rev 2003;26: 1–49. 2. Bartolotti C, Wang H, Fraser K, et al. Subacute spinal subdural hematoma after spontaneous resolution of cranial subdural hematoma: causal relationship or coincidence? J Neurosurg 2004;100:372–4.
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