Gen Thorac Cardiovasc Surg DOI 10.1007/s11748-014-0401-2
CASE REPORT
Spontaneous spinal epidural hematoma presenting as paraplegia after cardiac surgery Hajime Kin • Masayuki Mukaida • Junichi Koizumi Takeshi Kamada • Yoshino Mitsunaga • Tomoyuki Iwase • Akio Ikai • Hitoshi Okabayashi
•
Received: 28 January 2014 / Accepted: 27 March 2014 Ó The Japanese Association for Thoracic Surgery 2014
Abstract An 86-year-old woman was scheduled to undergo aortic valve replacement and coronary artery bypass graft. On postoperative day 3, she developed sudden-onset neck pain followed by weakness in the right arm. Her symptoms worsened with time, and she developed paraplegia. At 60 h after the first complaint, spontaneous spinal epidural hematoma (SSEH) from C2 to C6 with spinal cord compression was diagnosed from a magnetic resonance image of the cervical region. We decided on conservative therapy because operative recovery was impossible. Delayed diagnosis led to grievous results in the present case. When neurological abnormalities follow neck or back pain after open heart surgery, SSEH must be considered in the differential diagnosis. Further, if it is suspected, early cervical computed tomography/magnetic resonance imaging and surgery should be considered. Keywords Spinal epidural hematoma Paraplegia Cardiac surgery Magnetic resonance imaging
Introduction Spontaneous spinal epidural hematoma (SSEH) is an uncommon occurrence, with an estimated incidence of 0.1 per 100,000 individuals [1], and SSEH presenting as neurological deficits after cardiac surgery is sporadic even rarer [2–6]. This hematoma usually presents as sudden-
H. Kin (&) M. Mukaida J. Koizumi T. Kamada Y. Mitsunaga T. Iwase A. Ikai H. Okabayashi Department of Cardiovascular Surgery, Iwate Medical University Memorial Heart Center, 19-1 Uchimaru, Morioka 020-8505, Iwate, Japan e-mail: [email protected]
onset neck or back pain followed by the signs and symptoms of rapidly evolving nerve root and spinal cord compression. Although early diagnosis and prompt management are correlated with a good outcome, they remain a challenge for physicians. We encountered the case of a patient who developed paraplegia with SSEH of the cervical spine after conventional cardiac surgery.
Case An 86-year-old woman with a history of hypertension and chronic renal dysfunction visited the hospital for recurrent episodes of congestive heart failure. Echocardiography showed severe aortic stenosis (peak and mean pressure gradients, 94 and 65 mmHg, respectively; aortic valve area, 0.67 cm2) but normal left ventricular function (ejection fraction 75 %). Coronary angiography showed stenosis of the left anterior descending artery (LAD) and the right coronary artery. Preoperative neck and head magnetic resonance angiography showed no abnormal findings. The patient was scheduled to undergo aortic valve replacement (AVR) and coronary artery bypass graft (CABG). On the day of the surgery, activated partial thromboplastin time (APTT), APTT-international normalized ratio (INR), prothrombin time (PT), and PT-INR were 34.0 s, 1.22, 10.6 s, and 0.95, respectively, and the platelet count was 316 9 103/lL. The patient underwent CABG (left internal thoracic artery—LAD, saphenous vein graft—posterior descending artery) and AVR (19 mm—CEP MAGNA EASE; Edwards Lifescience, Irvine, CA) through a standard median sternotomy under mild hypothermic (35 °C) cardiopulmonary bypass (CPB). The patient weaned from the CPB smoothly.
123
Gen Thorac Cardiovasc Surg
The operating, CPB, and cross-clamp times were 270, 113, and 55 min, respectively. Intraoperative transfusion with 4 units of red cell concentrates and 4 units of fresh frozen plasma was performed. Initial neurological assessment under anesthesia yielded normal findings. Tracheal extubation was carried out successfully 17 h after the patient arrived in the intensive care unit. Although the postoperative course was uneventful and there was no report of cervical injury, the patient complained of sudden neck pain on postoperative day 3. One hour after the first complaint, the patient reported weakness of the right upper limb. On neurological examination, the patient was alert and fully oriented. Motor examination showed loss of motor power (grade 3/5) in the right upper and lower limbs. At this time, her blood pressure was 160/80 mmHg and PT-INR was 2.39; the patient had been administered warfarin from postoperative day 1. An emergency brain computed tomography (CT) scan indicated no bleeding or infarction. After 4 h, on brain CT evaluation, her systolic blood pressure was 60 mmHg and heart rate had decreased to 50 beats/min. The patient’s respiration rate had also decreased and PCO2 level was elevated (PCO2 = 85.5 mmHg) in the blood gas analysis. Tracheal intubation was carried out, and 17 h after the first complaint, the neurological deficit had worsened as indicated by the greater loss of motor power (grade 1/5) in the upper and lower limbs on both sides. A neurologist and orthopedician were consulted, and transverse myelopathy at the C4 cord level was diagnosed. Magnetic resonance image (MRI) of the cervical region was carried out, and spinal epidural hematoma from C2 to C6 with spinal cord compression was diagnosed (Fig. 1) at 60 h after the first complaint. At this time, the patient suffered from complete paraplegia with no reflexes of the anal sphincter muscle (proctoparalysis). We decided to administer conservative therapy because operative recovery was impossible. After tracheotomy, the patient was transferred to another hospital.
Discussion The characteristic clinical symptom of SSEH is local pain of acute onset. Within minutes or hours, signs of spinal cord compression appear as progressive paraplegia and the loss of sensory function. Some precipitating factors associated with SSEH include vascular malformation, anticoagulant therapy for prosthetic implanted valves, thrombolysis therapy for acute myocardial infarction, uncontrolled hypertension, and end-stage renal disease receiving hemodialysis [1]. In the present case, just before the symptoms appeared, hemodialysis was needed for renal dysfunction, and the blood pressure was relatively high. In
123
Fig. 1 A sagittal T1-weighted magnetic resonance image of the spine showing an anterior epidural hematoma extended from C2 to C6 (arrow)
addition, before symptom appearance, the PT-INR and platelet count were 2.39 and 55 9 103/lL, respectively, while after symptom appearance, they were 3.42 and 57 9 103/lL, respectively. The main cause underlying the SSEH in this case is unclear, although the patient had some risk factors for SSEH, including high blood pressure, hemodialysis therapy, and a bleeding tendency. One study showed that 20–30 % of spinal epidural hematomas occur as a result of anticoagulant therapy [7]. Recent studies [8, 9] have suggested that CT angiography and MRA are useful for diagnosing spinal vascular malformation. Preoperative MRA did not show an arteriovenous malformation in this case; therefore, we believe that it was not an issue. However, Shin et al. [10] suggested that further research may be required to determine if and how vascular anomalies cause SSEH. Although SSEH shows distinct characteristics, its early and accurate diagnosis remains a challenge for physicians. Further, SSEH presenting solely as neurological deficits after cardiac surgery is extremely rare [2–6]. A previous study of 5 such cases reported that 4 cases were spontaneous, while in 1 case, SSEH occurred after removal of a catheter under epidural analgesia [6]. In another study, except for idiopathic spinal epidural hematoma, one patient recovered spontaneously [2] while 3 patients underwent laminectomy [3–5]. After this operation, 1 patient recovered partially, but the others did not. The patient who partially recovered was operated on 14 h after the initial appearance of symptoms [4]. Of the remaining 2 patients, 1
Gen Thorac Cardiovasc Surg
was operated on 40 h after initial appearance of symptoms [3], while the other was operated on 60 h after undergoing cardiac surgery [5]. Shin et al. [10] reported a good outcome when patients were operated on within 24 h of initial symptom appearance. In particular, when patients with incomplete injury were operated on within 12 h, all of them returned to normal conditions. Therefore, Shin et al. [10] suggested that the final outcome was related to two factors: the preoperative neurological status and the time from the initial onset of symptoms to operation. As reported for differential diagnosis of epidural hematoma from cerebral infarction [11, 12], we also initially performed brain CT, since we suspected the patient had cerebral infarction, but we found no abnormalities. Kameda et. al [12] reported that it was impossible to conduct MRI for any reasons; cervical CT could have diagnosed SSEH in some cases. If we had conducted cervical CT for suspicions of SSEH at this point, we could have diagnosed SSEH faster. Subsequently, we performed MRI and diagnosed SSEH. However, the interval from initial symptom onset until this point was 60 h, during which time no improvements in patient condition were achieved. If we experience this kind of case in the future, we will not hesitate to choose surgical treatment. On the other hand, in the present case, neck pain appeared initially along with symptoms such as neurological abnormalities and acute respiratory disorder related to a high-position cervical cord lesion, which suggest diseases of the spinal cord, such as epidural hematoma. Thus, a delayed diagnosis led to grievous results in the present case. We recommend that when neurological abnormalities accompanied with neck or back pain are observed after open heart surgery, SSEH must be considered as part of the differential diagnosis, even though it is a rare condition. Further, if SSEH is suspected, early cervical CT/MRI and surgery should be considered.
Conflict of interest
None declared.
References 1. Holtas S, Heiling M, Lonntoft M. Spontaneous spinal epidural hematoma: findings at MR imaging and clinical correlation. Radiology. 1996;199:409–13. 2. Hayashi I, Shimamura Y, Maehara M. Paraplegia due to spinal epidural hematoma after mitral valve surgery: report of a case. Surg Today. 2011;41:704–6. 3. Karlekar A, Dutta D, Dev Arora K, Mishra yK. Spinal-epidural hematoma presenting as paraplegia following mitral valve surgery: a case report. J Cardiothorac Vasc Anesth. 2013;. doi:10. 1053/j.jvca.2013.07.012 (Epub ahead of print). 4. Imanaka K, Kyo S, Yokote Y, Asano H, Tanabe H, Ohuchi H. Paraplegia due to acute spinal epidural hematoma after routine cardiac surgery. Intensive Care Med. 2000;26:826. 5. Nakaya M, Kawazoe K, Ohara K, Kosakai Y, Kito Y, Kawashima Y. A case report of spinal epidural hematoma complicated after open heart surgery. Jpn J Cardiothorac Surg. 1992;40:116–8. 6. Rosen DA, Hawkinberry DW, Rosen KR, Gustafson RA, Hogg JP, Broadman LM. An epidural hematoma in an adolescent patient after cardiac surgery. Anesth Analg. 2004;98:966–9. 7. Baena RR, Gaetani P, Tancioni F, Tartara F. Spinal epidural hematoma during anticoagulant therapy. A case report and review of the literature. J Neurosurg Sci. 1995;39(1):87–94. 8. Toni F, Cirillo L, Marliani AF, Albini Riccioli L, Leonardi M. Role of contrast-enhanced magnetic resonance angiography in spinal dural arteriovenous fistula. Neuroradiol J. 2010;23(2):234–43. 9. Sharma AK, Westesson PL. Preoperative evaluation of spinal vascular malformation by MR angiography: how reliable is the technique: case report and review of literature. Clin Neurol Neurosurg. 2008;110(5):521–4. 10. Shin JJ, Kuh SU, Cho YE. Surgical management of spontaneous spinal epidural hematoma. Eur Spine J. 2006;15:998–1004. 11. Son S, Kong DH, Choi DS, Kim SK, Lim BH, Choi NC. A case of spontaneous spinal epidural hematoma mimicking a stroke. Neurologist. 2012;18:41–3. 12. Kameda T, Doi H, Sugiyama M, Ueda N, Kugimoto C, Baba Y, et al. Three cases of cervical epidural hematoma mimicking acute ischemic stroke. Brain Nerve. 2009;6(12):1429–33.
123
CASE REPORT
Spontaneous spinal epidural hematoma presenting as paraplegia after cardiac surgery Hajime Kin • Masayuki Mukaida • Junichi Koizumi Takeshi Kamada • Yoshino Mitsunaga • Tomoyuki Iwase • Akio Ikai • Hitoshi Okabayashi
•
Received: 28 January 2014 / Accepted: 27 March 2014 Ó The Japanese Association for Thoracic Surgery 2014
Abstract An 86-year-old woman was scheduled to undergo aortic valve replacement and coronary artery bypass graft. On postoperative day 3, she developed sudden-onset neck pain followed by weakness in the right arm. Her symptoms worsened with time, and she developed paraplegia. At 60 h after the first complaint, spontaneous spinal epidural hematoma (SSEH) from C2 to C6 with spinal cord compression was diagnosed from a magnetic resonance image of the cervical region. We decided on conservative therapy because operative recovery was impossible. Delayed diagnosis led to grievous results in the present case. When neurological abnormalities follow neck or back pain after open heart surgery, SSEH must be considered in the differential diagnosis. Further, if it is suspected, early cervical computed tomography/magnetic resonance imaging and surgery should be considered. Keywords Spinal epidural hematoma Paraplegia Cardiac surgery Magnetic resonance imaging
Introduction Spontaneous spinal epidural hematoma (SSEH) is an uncommon occurrence, with an estimated incidence of 0.1 per 100,000 individuals [1], and SSEH presenting as neurological deficits after cardiac surgery is sporadic even rarer [2–6]. This hematoma usually presents as sudden-
H. Kin (&) M. Mukaida J. Koizumi T. Kamada Y. Mitsunaga T. Iwase A. Ikai H. Okabayashi Department of Cardiovascular Surgery, Iwate Medical University Memorial Heart Center, 19-1 Uchimaru, Morioka 020-8505, Iwate, Japan e-mail: [email protected]
onset neck or back pain followed by the signs and symptoms of rapidly evolving nerve root and spinal cord compression. Although early diagnosis and prompt management are correlated with a good outcome, they remain a challenge for physicians. We encountered the case of a patient who developed paraplegia with SSEH of the cervical spine after conventional cardiac surgery.
Case An 86-year-old woman with a history of hypertension and chronic renal dysfunction visited the hospital for recurrent episodes of congestive heart failure. Echocardiography showed severe aortic stenosis (peak and mean pressure gradients, 94 and 65 mmHg, respectively; aortic valve area, 0.67 cm2) but normal left ventricular function (ejection fraction 75 %). Coronary angiography showed stenosis of the left anterior descending artery (LAD) and the right coronary artery. Preoperative neck and head magnetic resonance angiography showed no abnormal findings. The patient was scheduled to undergo aortic valve replacement (AVR) and coronary artery bypass graft (CABG). On the day of the surgery, activated partial thromboplastin time (APTT), APTT-international normalized ratio (INR), prothrombin time (PT), and PT-INR were 34.0 s, 1.22, 10.6 s, and 0.95, respectively, and the platelet count was 316 9 103/lL. The patient underwent CABG (left internal thoracic artery—LAD, saphenous vein graft—posterior descending artery) and AVR (19 mm—CEP MAGNA EASE; Edwards Lifescience, Irvine, CA) through a standard median sternotomy under mild hypothermic (35 °C) cardiopulmonary bypass (CPB). The patient weaned from the CPB smoothly.
123
Gen Thorac Cardiovasc Surg
The operating, CPB, and cross-clamp times were 270, 113, and 55 min, respectively. Intraoperative transfusion with 4 units of red cell concentrates and 4 units of fresh frozen plasma was performed. Initial neurological assessment under anesthesia yielded normal findings. Tracheal extubation was carried out successfully 17 h after the patient arrived in the intensive care unit. Although the postoperative course was uneventful and there was no report of cervical injury, the patient complained of sudden neck pain on postoperative day 3. One hour after the first complaint, the patient reported weakness of the right upper limb. On neurological examination, the patient was alert and fully oriented. Motor examination showed loss of motor power (grade 3/5) in the right upper and lower limbs. At this time, her blood pressure was 160/80 mmHg and PT-INR was 2.39; the patient had been administered warfarin from postoperative day 1. An emergency brain computed tomography (CT) scan indicated no bleeding or infarction. After 4 h, on brain CT evaluation, her systolic blood pressure was 60 mmHg and heart rate had decreased to 50 beats/min. The patient’s respiration rate had also decreased and PCO2 level was elevated (PCO2 = 85.5 mmHg) in the blood gas analysis. Tracheal intubation was carried out, and 17 h after the first complaint, the neurological deficit had worsened as indicated by the greater loss of motor power (grade 1/5) in the upper and lower limbs on both sides. A neurologist and orthopedician were consulted, and transverse myelopathy at the C4 cord level was diagnosed. Magnetic resonance image (MRI) of the cervical region was carried out, and spinal epidural hematoma from C2 to C6 with spinal cord compression was diagnosed (Fig. 1) at 60 h after the first complaint. At this time, the patient suffered from complete paraplegia with no reflexes of the anal sphincter muscle (proctoparalysis). We decided to administer conservative therapy because operative recovery was impossible. After tracheotomy, the patient was transferred to another hospital.
Discussion The characteristic clinical symptom of SSEH is local pain of acute onset. Within minutes or hours, signs of spinal cord compression appear as progressive paraplegia and the loss of sensory function. Some precipitating factors associated with SSEH include vascular malformation, anticoagulant therapy for prosthetic implanted valves, thrombolysis therapy for acute myocardial infarction, uncontrolled hypertension, and end-stage renal disease receiving hemodialysis [1]. In the present case, just before the symptoms appeared, hemodialysis was needed for renal dysfunction, and the blood pressure was relatively high. In
123
Fig. 1 A sagittal T1-weighted magnetic resonance image of the spine showing an anterior epidural hematoma extended from C2 to C6 (arrow)
addition, before symptom appearance, the PT-INR and platelet count were 2.39 and 55 9 103/lL, respectively, while after symptom appearance, they were 3.42 and 57 9 103/lL, respectively. The main cause underlying the SSEH in this case is unclear, although the patient had some risk factors for SSEH, including high blood pressure, hemodialysis therapy, and a bleeding tendency. One study showed that 20–30 % of spinal epidural hematomas occur as a result of anticoagulant therapy [7]. Recent studies [8, 9] have suggested that CT angiography and MRA are useful for diagnosing spinal vascular malformation. Preoperative MRA did not show an arteriovenous malformation in this case; therefore, we believe that it was not an issue. However, Shin et al. [10] suggested that further research may be required to determine if and how vascular anomalies cause SSEH. Although SSEH shows distinct characteristics, its early and accurate diagnosis remains a challenge for physicians. Further, SSEH presenting solely as neurological deficits after cardiac surgery is extremely rare [2–6]. A previous study of 5 such cases reported that 4 cases were spontaneous, while in 1 case, SSEH occurred after removal of a catheter under epidural analgesia [6]. In another study, except for idiopathic spinal epidural hematoma, one patient recovered spontaneously [2] while 3 patients underwent laminectomy [3–5]. After this operation, 1 patient recovered partially, but the others did not. The patient who partially recovered was operated on 14 h after the initial appearance of symptoms [4]. Of the remaining 2 patients, 1
Gen Thorac Cardiovasc Surg
was operated on 40 h after initial appearance of symptoms [3], while the other was operated on 60 h after undergoing cardiac surgery [5]. Shin et al. [10] reported a good outcome when patients were operated on within 24 h of initial symptom appearance. In particular, when patients with incomplete injury were operated on within 12 h, all of them returned to normal conditions. Therefore, Shin et al. [10] suggested that the final outcome was related to two factors: the preoperative neurological status and the time from the initial onset of symptoms to operation. As reported for differential diagnosis of epidural hematoma from cerebral infarction [11, 12], we also initially performed brain CT, since we suspected the patient had cerebral infarction, but we found no abnormalities. Kameda et. al [12] reported that it was impossible to conduct MRI for any reasons; cervical CT could have diagnosed SSEH in some cases. If we had conducted cervical CT for suspicions of SSEH at this point, we could have diagnosed SSEH faster. Subsequently, we performed MRI and diagnosed SSEH. However, the interval from initial symptom onset until this point was 60 h, during which time no improvements in patient condition were achieved. If we experience this kind of case in the future, we will not hesitate to choose surgical treatment. On the other hand, in the present case, neck pain appeared initially along with symptoms such as neurological abnormalities and acute respiratory disorder related to a high-position cervical cord lesion, which suggest diseases of the spinal cord, such as epidural hematoma. Thus, a delayed diagnosis led to grievous results in the present case. We recommend that when neurological abnormalities accompanied with neck or back pain are observed after open heart surgery, SSEH must be considered as part of the differential diagnosis, even though it is a rare condition. Further, if SSEH is suspected, early cervical CT/MRI and surgery should be considered.
Conflict of interest
None declared.
References 1. Holtas S, Heiling M, Lonntoft M. Spontaneous spinal epidural hematoma: findings at MR imaging and clinical correlation. Radiology. 1996;199:409–13. 2. Hayashi I, Shimamura Y, Maehara M. Paraplegia due to spinal epidural hematoma after mitral valve surgery: report of a case. Surg Today. 2011;41:704–6. 3. Karlekar A, Dutta D, Dev Arora K, Mishra yK. Spinal-epidural hematoma presenting as paraplegia following mitral valve surgery: a case report. J Cardiothorac Vasc Anesth. 2013;. doi:10. 1053/j.jvca.2013.07.012 (Epub ahead of print). 4. Imanaka K, Kyo S, Yokote Y, Asano H, Tanabe H, Ohuchi H. Paraplegia due to acute spinal epidural hematoma after routine cardiac surgery. Intensive Care Med. 2000;26:826. 5. Nakaya M, Kawazoe K, Ohara K, Kosakai Y, Kito Y, Kawashima Y. A case report of spinal epidural hematoma complicated after open heart surgery. Jpn J Cardiothorac Surg. 1992;40:116–8. 6. Rosen DA, Hawkinberry DW, Rosen KR, Gustafson RA, Hogg JP, Broadman LM. An epidural hematoma in an adolescent patient after cardiac surgery. Anesth Analg. 2004;98:966–9. 7. Baena RR, Gaetani P, Tancioni F, Tartara F. Spinal epidural hematoma during anticoagulant therapy. A case report and review of the literature. J Neurosurg Sci. 1995;39(1):87–94. 8. Toni F, Cirillo L, Marliani AF, Albini Riccioli L, Leonardi M. Role of contrast-enhanced magnetic resonance angiography in spinal dural arteriovenous fistula. Neuroradiol J. 2010;23(2):234–43. 9. Sharma AK, Westesson PL. Preoperative evaluation of spinal vascular malformation by MR angiography: how reliable is the technique: case report and review of literature. Clin Neurol Neurosurg. 2008;110(5):521–4. 10. Shin JJ, Kuh SU, Cho YE. Surgical management of spontaneous spinal epidural hematoma. Eur Spine J. 2006;15:998–1004. 11. Son S, Kong DH, Choi DS, Kim SK, Lim BH, Choi NC. A case of spontaneous spinal epidural hematoma mimicking a stroke. Neurologist. 2012;18:41–3. 12. Kameda T, Doi H, Sugiyama M, Ueda N, Kugimoto C, Baba Y, et al. Three cases of cervical epidural hematoma mimicking acute ischemic stroke. Brain Nerve. 2009;6(12):1429–33.
123
