Case Reports / Journal of Clinical Neuroscience 22 (2015) 1515–1518
infiltration into the optic nerves. He died of CNS lymphoma before an optic nerve biopsy was obtained. Consent was obtained from his family to perform a post mortem optic nerve biopsy which showed infiltrative malignant lymphocytes causing vessel occlusion. It appears that although optic nerve biopsies are highly invasive and potentially blinding, they offer the best diagnostic results for LON. Many reports of elusive LON patients with negative MRI and LP in the literature have advocated for optic nerve sheath fenestration for diagnosis [5]. Diagnostic confirmation is also important as intrathecal chemotherapy and local radiotherapy can lead to toxicity and permanent damage to the brain. Our patient developed progressive diffuse white matter changes on his MRI secondary to chemotoxicity. Therefore, the radiation oncologists were initially reluctant to start radiotherapy. Moreover, the MRI raised the possibility of optic neuritis and all other investigations had been negative so far. When the ophthalmologists found similar progressive changes in the left eye to those that had occurred in the right eye which were causing no perception to light, local radiotherapy was administered to both eyes. Despite this treatment, our patient developed lymphomatous meningitis and died. Treatment of LON should not be delayed if typical fundus findings are present despite negative MRI and LP investigations. An
1515
optic nerve biopsy should be considered, especially in a blind eye, to confirm diagnosis if required.
Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.
References [1] Kim UR, Shah AD, Arora V, et al. Isolated optic nerve infiltration in systemic lymphoma – a case report and review of literature. Ophthal Plast Reconstr Surg 2010;26:291–3. [2] Shukla D, Arora A, Hadi KM, et al. Combined central retinal and vein occlusion secondary to systemic non-Hogkin’s lymphoma. Indian J Ophthalmol 2006;54:204–6. [3] Lee LC, Howes EL, Bhisitkul RB. Systemic non-Hodgkin’s lymphoma with optic nerve infiltration in a patient with AIDS. Retina 2002;22:75–9. [4] Fierz AB, Sartoretti S, Thoelen AM. Optic neuropathy and central retinal artery occlusion in non-Hodgkin lymphoma. J Neuroophthalmol 2001;21:103–5. [5] Behbehani RS, Vacerezza N, Sergott RC, et al. Isolated optic nerve lymphoma diagnosed by optic nerve biopsy. Am J Ophthalmol 2005;139:1128–30. [6] Tatsugawa M, Noma H, Mumura T, et al. Unusual orbital lymphoma undetectable by magnetic resonance imaging: a case report. J Med Case Rep 2009;3:104.
http://dx.doi.org/10.1016/j.jocn.2015.03.022
Posterior circulation cerebral hyperperfusion syndrome after high flow external carotid artery to middle cerebral artery bypass Eric T. Quach a,b,⇑, Andres A. Gonzalez c, Parastou Shilian c, Jonathan J. Russin b a b c
Temple University School of Medicine, 3500 North Broad Street, Philadelphia, PA 19140, USA Department of Neurological Surgery, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA Department of Neurology, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA
a r t i c l e
i n f o
Article history: Received 10 March 2015 Accepted 21 March 2015
Keywords: Cerebral hyperemia Cerebral hyperperfusion syndrome High flow extracranial-intracranial arterial bypass Revascularization
a b s t r a c t We present the first report, to our knowledge, in which revascularization of the middle cerebral artery (MCA) with a high flow extracranial-intracranial procedure resulted in symptomatic hyperemia of the posterior circulation. Cerebral hyperperfusion syndrome (CHS) is a poorly understood phenomenon that is classically seen in the distribution of a revascularized artery. A 37-year-old woman presented with a 3 month history of cognitive and speech difficulties, persistent headaches, weakness, numbness, and paresthesia which was worse in the right extremities and face. She was found to have bilateral watershed infarcts worse in the left cerebral hemisphere, severe bilateral stenosis of the supraclinoid internal carotid artery, and a small left superior hypophyseal aneurysm. The patient underwent left cerebral hemisphere revascularization with a high flow external carotid artery to MCA bypass with aneurysm trapping. During skin closure, significant changes were seen in her bilateral upper extremity motor-evoked potentials. The patient’s postoperative exam was noted for an intermittent inability to follow commands, bilateral upper extremity weakness, vertical nystagmus, and alogia that all dramatically improved with strict blood pressure control. Postoperative perfusion imaging revealed posterior circulation hyperemia. This patient highlights the potential for hyperemic complications outside the revascularized arterial territory. Strict blood pressure control is recommended in order to prevent and manage hyperemia-associated symptoms. Improving our understanding of CHS may assist in identifying at risk patients and at risk arterial territories in order to optimize CHS prevention and management strategies. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction
⇑ Corresponding author. Tel.: +1 94 92444780. E-mail address: [email protected] (E.T. Quach).
Cerebral hyperemia is a poorly understood phenomenon that is classically seen in the distribution of a revascularized artery. We present the first report, to our knowledge, in which revascularization of the middle cerebral artery (MCA) with a high flow
1516
Case Reports / Journal of Clinical Neuroscience 22 (2015) 1515–1518
extracranial-intracranial procedure resulted hyperemia of the posterior circulation.
in
symptomatic
2. Case report A 37-year-old woman presented with a 3 month history of cognitive and speech difficulties, persistent headaches, weakness, numbness, and paresthesia which was worse in the right extremities and face. Imaging revealed bilateral watershed infarcts with the left hemisphere more greatly affected than the right (Fig. 1A–C; Supp. Fig. 1A–D). A cerebral vessel angiogram demonstrated severe, bilateral stenosis of the supraclinoid internal carotid artery (ICA) consistent with a Moyamoya-like disease (Fig. 1D–E). Furthermore, a small, downward pointing left superior hypophyseal aneurysm was discovered (Fig. 1D). Due to the right-sided lateralization of the majority of the symptoms and the presence of a left superior hypophyseal aneurysm, a decision was made to revascularize the left cerebral hemisphere with an external carotid artery (ECA) to MCA bypass utilizing a radial artery graft (RAG) with proximal left ICA occlusion.
During the procedure, the patient’s motor evoked potentials (MEP) and somatosensory evoked potentials (SSEP) were monitored. After anastomosis of the RAG to the M3 branch of the MCA and the ECA, a significant reduction in amplitude was seen in bilateral intrinsic hand MEP. No changes were seen in SSEP. Typically, intraoperative monitoring changes during this procedure are related to hypoperfusion in a particular vascular territory resulting in neurophysiological changes contralateral to the injured area. In this woman, amplitude reduction was seen bilaterally. In the immediate postoperative period, the woman had a fluctuating neurological examination characterized by an intermittent inability to follow commands, significant bilateral upper extremity weakness, a coarse upbeat nystagmus, and alogia. A postoperative perfusion CT scan demonstrated increased cerebral blood flow (CBF), mean transit time (MTT), and cerebral blood volume (CBV) in the posterior circulation consistent with cerebral hyperemia (Supp. Fig. 1E–G). The revascularized left MCA territory showed increased CBF with normal MTT and CBV (Supp. Fig. 1E–G). The patient’s neurological exam returned to baseline after strict normotensive blood pressure control. Electroencephalography monitoring was negative for epileptiform activity. Follow-up imaging was negative for hemorrhage and
Fig. 1. Preoperative axial MRI demonstrating watershed infarcts and restricted diffusion on T2-weighted fluid-attenuated inversion recovery sequence (A), diffusionweighted imaging (B), and apparent diffusion coefficient map (C) affecting the left hemisphere greater than the right. Lateral views of left (D) and right (E) internal carotid artery preoperative angiogram revealing severe bilateral supraclinoid stenosis along with a small left superior hypophyseal aneurysm (arrow).
Case Reports / Journal of Clinical Neuroscience 22 (2015) 1515–1518
1517
Fig. 2. 3D CT scan reconstruction (A) and conventional angiography (B) demonstrating radial artery graft patency (arrow) along with occlusion of the left ICA with an aneurysm clip (arrowhead). Frontal (C) and lateral view (D) of preoperative vertebral angiogram demonstrating bilateral filling of the middle cerebral artery territories from the posterior communicating arteries. This figure is available in colour at www.sciencedirect.com.
confirmed graft patency (Fig. 2A–B). The symptoms improved by postoperative day 6 and she was discharged with plans to revascularize her right hemisphere. 3. Discussion Cerebral hyperperfusion syndrome (CHS), most often defined as a symptomatic increase in cerebral blood flow, has been reported in 0.4–38% of patients across all methods of carotid artery revascularization [1–3]. A recent patient series reported the incidence of CHS specifically after high flow extracranial-intracranial bypass to be 4%, while other patient series examining hyperemia reported no incidences [4,5]. In contrast, studies examining cerebral ischemia explicitly after high flow bypass reported that 60% of patients demonstrated transient elevations of mean CBF postoperatively with 17–23% found to have intracerebral hyperemia with hemorrhage [6–8]. However, to our knowledge, there are no reports of symptomatic cerebral hyperemia in the posterior circulation after high flow bypass into the MCA territory. Although CBF increased in the left MCA territory, the perfusion evidence of hyperemia was only seen in the posterior circulation. Our patient’s preoperative angiogram showed significant filling of the bilateral MCA territories via the posterior communicating arteries (Fig. 2C–D). Given the watershed infarcts, it is likely that the posterior circulation was maximally dilated for some time and likely had dysfunction
of the normal autoregulatory mechanisms. Additionally, the vertebrobasilar system has decreased sympathetic innervation in comparison to other vascular cerebral territories, further explaining the hyperemic pattern in our patient [1,9]. Regarding the timing of hyperemic complications, Stiver et al. described a patient with CHS which occurred intraoperatively during a high flow bypass for carotid stenosis [10]. In light of their experience, our patient’s intraoperative neurophysiological changes may have been a manifestation of cerebral hyperemia. Although patients with severe cases may progress to intracerebral hemorrhage, hyperemic symptoms often resolve once vigilant blood pressure control is instituted with care taken to avoid cerebral vasodilating medications such as hydralazine, nitrates, and calcium channel blockers which may worsen cerebral edema [1,2]. Cerebral edema and seizures should be treated in the usual fashion as no data exists for the treatment of these entities in the context of CHS [1,2]. 4. Conclusion A high index of suspicion for cerebral hyperemia should be maintained both during and after high flow extracranial-intracranial bypass, especially in patients with chronic cerebral ischemia. The woman reported here highlights the potential for hyperemic complications outside the revascularized arterial territory. Improving our understanding of CHS may assist in identifying at risk patients
1518
Case Reports / Journal of Clinical Neuroscience 22 (2015) 1518–1520
and at risk arterial territories in order to optimize CHS prevention and management strategies. Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. Appendix A. Supplementary material Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.jocn.2015.03.022. References [1] van Mook WN, Rennenberg RJ, Schurink GW, et al. Cerebral hyperperfusion syndrome. Lancet Neurol 2005;4:877–88. [2] Moulakakis KG, Mylonas SN, Sfyroeras GS, et al. Hyperperfusion syndrome after carotid revascularization. J Vasc Surg 2009;49:1060–8.
[3] Yamaguchi K, Kawamata T, Kawashima A, et al. Incidence and predictive factors of cerebral hyperperfusion after extracranial-intracranial bypass for occlusive cerebrovascular diseases. Neurosurgery 2010;67:1548–54 [discussion 1554]. [4] Morton RP, Moore AE, Barber J, et al. Monitoring flow in extracranialintracranial bypass grafts using duplex ultrasonography: a single-center experience in 80 grafts over 8 years. Neurosurgery 2014;74:62–70. [5] Ishishita Y, Tanikawa R, Noda K, et al. Universal extracranial-intracranial graft bypass for large or giant internal carotid aneurysms: techniques and results in 38 consecutive patients. World Neurosurg 2014;82:130–9. [6] Ishikawa T, Kamiyama H, Houkin K, et al. Postsurgical observations of mean hemispheric cerebral blood flow with patients receiving high-flow EC-IC bypass using a radial artery graft (preliminary report, one-year observation of 10 hemispheres). Surg Neurol 1995;43:500–6 [discussion 506–9]. [7] Sundt Jr TM, Piepgras DG, Marsh WR, et al. Saphenous vein bypass grafts for giant aneurysms and intracranial occlusive disease. J Neurosurg 1986;65: 439–50. [8] Diaz FG, Pearce J, Ausman JI. Complications of cerebral revascularization with autogenous vein grafts. Neurosurgery 1985;17:271–6. [9] Schwartz RB. Hyperperfusion encephalopathies: hypertensive encephalopathy and related conditions. Neurologist 2002;8:22–34. [10] Stiver SI, Ogilvy CS. Acute hyperperfusion syndrome complicating EC-IC bypass. J Neurol Neurosurg Psychiatry 2002;73:88–9.
http://dx.doi.org/10.1016/j.jocn.2015.03.022
Cauda equina syndrome caused by retained hemostatic agents Matthew C. Bessette, Addisu Mesfin ⇑ Department of Orthopaedics and Rehabilitation, University of Rochester Medical Center, 601 Elmwood Ave, Box 665, Rochester, NY 14642, USA
a r t i c l e
i n f o
Article history: Received 18 November 2014 Accepted 3 March 2015
Keywords: Cauda equina syndrome Complications Gelfoam Hemostatic agents Surgicel
a b s t r a c t We report a patient with cauda equina syndrome secondary to retained Surgicel (Ethicon: Johnson & Johnson Medical, Piscataway, NJ, USA) and Gelfoam (Pfizer, New York, NY, USA). Retained hemostatic agents such as these used in spinal surgery can create space occupying lesions that lead to neurological compromise. There are few reports of neurologic symptoms caused by hemostatic devices used in surgery despite their widespread use. Our patient had routine lumbar microdiscectomy with preexisting motor deficits, and the acute postoperative course was complicated by a decline in neurologic function. The findings at the time of reoperation confirmed compression due to Gelfoam and Surgicel in the spinal canal, and subjective and objective symptoms partially improved after reoperation. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction Difficult exposure, osteotomies, epidural venous plexi, and a low tolerance for hematoma formation are among the many variables that make hemostasis in spinal surgery both difficult and imperative. Various agents have been introduced over the years to help surgeons successfully control bleeding. While their use has been instrumental in many advances made in the field, this is not without unintended consequences. We report a routine lumbar microdiscectomy and foraminal decompression complicated by postoperative neurologic changes. Reoperation and wound exploration revealed retained Surgicel (Ethicon: Johnson & Johnson Medical, Piscataway, NJ, USA) and Gelfoam (Pfizer, New York, NY, USA) material around the thecal sac with no evidence of hematoma formation. 2. Case report A 62-year-old, otherwise healthy man presented with a 4 month history of gradually worsening radicular pain and ⇑ Corresponding author. Tel.: +1 585 275 5196; fax: +1 585 756 4726. E-mail address: addisu_mesfi[email protected] (A. Mesfin).
subjective numbness in his right leg. On examination, he had 4/5 motor strength on knee extension, 3/5 ankle dorsiflexion, 1/5 great toe extension and 4/5 ankle plantarflexion. Treatment with physical therapy, anti-inflammatory medications, gabapentin (Neurontin; Pfizer) and transforaminal nerve root injections was unsuccessful. MRI revealed a central L5–S1 herniated disc abutting the S1 nerve root and an extruded right central herniated disc at L4–L5 abutting the L5 nerve root (Fig. 1). The patient underwent a right L4–L5 microdiscectomy and a right L5–S1 foraminal decompression. This was complicated by a durotomy which was repaired with absorbable sutures. Hemostasis, which was noted to be particularly difficult during the surgery, was aided with the use of Surgicel and Gelfoam. At the time of his first postoperative evaluation, the man reported worsening numbness in the right L5 and S1 distributions as well as new numbness in his right buttock. He also reported diminished sensation during voiding which he was still able to control. His motor strength was unchanged. One day after surgery, significant changes included 1/5 ankle dorsiflexion and stress incontinence. An urgent MRI was initially interpreted by the neuroradiologist as concerning for epidural hematoma (Fig. 2). He was taken back to the operating room for an exploration of the wound. Findings included Gelfoam and
infiltration into the optic nerves. He died of CNS lymphoma before an optic nerve biopsy was obtained. Consent was obtained from his family to perform a post mortem optic nerve biopsy which showed infiltrative malignant lymphocytes causing vessel occlusion. It appears that although optic nerve biopsies are highly invasive and potentially blinding, they offer the best diagnostic results for LON. Many reports of elusive LON patients with negative MRI and LP in the literature have advocated for optic nerve sheath fenestration for diagnosis [5]. Diagnostic confirmation is also important as intrathecal chemotherapy and local radiotherapy can lead to toxicity and permanent damage to the brain. Our patient developed progressive diffuse white matter changes on his MRI secondary to chemotoxicity. Therefore, the radiation oncologists were initially reluctant to start radiotherapy. Moreover, the MRI raised the possibility of optic neuritis and all other investigations had been negative so far. When the ophthalmologists found similar progressive changes in the left eye to those that had occurred in the right eye which were causing no perception to light, local radiotherapy was administered to both eyes. Despite this treatment, our patient developed lymphomatous meningitis and died. Treatment of LON should not be delayed if typical fundus findings are present despite negative MRI and LP investigations. An
1515
optic nerve biopsy should be considered, especially in a blind eye, to confirm diagnosis if required.
Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.
References [1] Kim UR, Shah AD, Arora V, et al. Isolated optic nerve infiltration in systemic lymphoma – a case report and review of literature. Ophthal Plast Reconstr Surg 2010;26:291–3. [2] Shukla D, Arora A, Hadi KM, et al. Combined central retinal and vein occlusion secondary to systemic non-Hogkin’s lymphoma. Indian J Ophthalmol 2006;54:204–6. [3] Lee LC, Howes EL, Bhisitkul RB. Systemic non-Hodgkin’s lymphoma with optic nerve infiltration in a patient with AIDS. Retina 2002;22:75–9. [4] Fierz AB, Sartoretti S, Thoelen AM. Optic neuropathy and central retinal artery occlusion in non-Hodgkin lymphoma. J Neuroophthalmol 2001;21:103–5. [5] Behbehani RS, Vacerezza N, Sergott RC, et al. Isolated optic nerve lymphoma diagnosed by optic nerve biopsy. Am J Ophthalmol 2005;139:1128–30. [6] Tatsugawa M, Noma H, Mumura T, et al. Unusual orbital lymphoma undetectable by magnetic resonance imaging: a case report. J Med Case Rep 2009;3:104.
http://dx.doi.org/10.1016/j.jocn.2015.03.022
Posterior circulation cerebral hyperperfusion syndrome after high flow external carotid artery to middle cerebral artery bypass Eric T. Quach a,b,⇑, Andres A. Gonzalez c, Parastou Shilian c, Jonathan J. Russin b a b c
Temple University School of Medicine, 3500 North Broad Street, Philadelphia, PA 19140, USA Department of Neurological Surgery, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA Department of Neurology, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA
a r t i c l e
i n f o
Article history: Received 10 March 2015 Accepted 21 March 2015
Keywords: Cerebral hyperemia Cerebral hyperperfusion syndrome High flow extracranial-intracranial arterial bypass Revascularization
a b s t r a c t We present the first report, to our knowledge, in which revascularization of the middle cerebral artery (MCA) with a high flow extracranial-intracranial procedure resulted in symptomatic hyperemia of the posterior circulation. Cerebral hyperperfusion syndrome (CHS) is a poorly understood phenomenon that is classically seen in the distribution of a revascularized artery. A 37-year-old woman presented with a 3 month history of cognitive and speech difficulties, persistent headaches, weakness, numbness, and paresthesia which was worse in the right extremities and face. She was found to have bilateral watershed infarcts worse in the left cerebral hemisphere, severe bilateral stenosis of the supraclinoid internal carotid artery, and a small left superior hypophyseal aneurysm. The patient underwent left cerebral hemisphere revascularization with a high flow external carotid artery to MCA bypass with aneurysm trapping. During skin closure, significant changes were seen in her bilateral upper extremity motor-evoked potentials. The patient’s postoperative exam was noted for an intermittent inability to follow commands, bilateral upper extremity weakness, vertical nystagmus, and alogia that all dramatically improved with strict blood pressure control. Postoperative perfusion imaging revealed posterior circulation hyperemia. This patient highlights the potential for hyperemic complications outside the revascularized arterial territory. Strict blood pressure control is recommended in order to prevent and manage hyperemia-associated symptoms. Improving our understanding of CHS may assist in identifying at risk patients and at risk arterial territories in order to optimize CHS prevention and management strategies. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction
⇑ Corresponding author. Tel.: +1 94 92444780. E-mail address: [email protected] (E.T. Quach).
Cerebral hyperemia is a poorly understood phenomenon that is classically seen in the distribution of a revascularized artery. We present the first report, to our knowledge, in which revascularization of the middle cerebral artery (MCA) with a high flow
1516
Case Reports / Journal of Clinical Neuroscience 22 (2015) 1515–1518
extracranial-intracranial procedure resulted hyperemia of the posterior circulation.
in
symptomatic
2. Case report A 37-year-old woman presented with a 3 month history of cognitive and speech difficulties, persistent headaches, weakness, numbness, and paresthesia which was worse in the right extremities and face. Imaging revealed bilateral watershed infarcts with the left hemisphere more greatly affected than the right (Fig. 1A–C; Supp. Fig. 1A–D). A cerebral vessel angiogram demonstrated severe, bilateral stenosis of the supraclinoid internal carotid artery (ICA) consistent with a Moyamoya-like disease (Fig. 1D–E). Furthermore, a small, downward pointing left superior hypophyseal aneurysm was discovered (Fig. 1D). Due to the right-sided lateralization of the majority of the symptoms and the presence of a left superior hypophyseal aneurysm, a decision was made to revascularize the left cerebral hemisphere with an external carotid artery (ECA) to MCA bypass utilizing a radial artery graft (RAG) with proximal left ICA occlusion.
During the procedure, the patient’s motor evoked potentials (MEP) and somatosensory evoked potentials (SSEP) were monitored. After anastomosis of the RAG to the M3 branch of the MCA and the ECA, a significant reduction in amplitude was seen in bilateral intrinsic hand MEP. No changes were seen in SSEP. Typically, intraoperative monitoring changes during this procedure are related to hypoperfusion in a particular vascular territory resulting in neurophysiological changes contralateral to the injured area. In this woman, amplitude reduction was seen bilaterally. In the immediate postoperative period, the woman had a fluctuating neurological examination characterized by an intermittent inability to follow commands, significant bilateral upper extremity weakness, a coarse upbeat nystagmus, and alogia. A postoperative perfusion CT scan demonstrated increased cerebral blood flow (CBF), mean transit time (MTT), and cerebral blood volume (CBV) in the posterior circulation consistent with cerebral hyperemia (Supp. Fig. 1E–G). The revascularized left MCA territory showed increased CBF with normal MTT and CBV (Supp. Fig. 1E–G). The patient’s neurological exam returned to baseline after strict normotensive blood pressure control. Electroencephalography monitoring was negative for epileptiform activity. Follow-up imaging was negative for hemorrhage and
Fig. 1. Preoperative axial MRI demonstrating watershed infarcts and restricted diffusion on T2-weighted fluid-attenuated inversion recovery sequence (A), diffusionweighted imaging (B), and apparent diffusion coefficient map (C) affecting the left hemisphere greater than the right. Lateral views of left (D) and right (E) internal carotid artery preoperative angiogram revealing severe bilateral supraclinoid stenosis along with a small left superior hypophyseal aneurysm (arrow).
Case Reports / Journal of Clinical Neuroscience 22 (2015) 1515–1518
1517
Fig. 2. 3D CT scan reconstruction (A) and conventional angiography (B) demonstrating radial artery graft patency (arrow) along with occlusion of the left ICA with an aneurysm clip (arrowhead). Frontal (C) and lateral view (D) of preoperative vertebral angiogram demonstrating bilateral filling of the middle cerebral artery territories from the posterior communicating arteries. This figure is available in colour at www.sciencedirect.com.
confirmed graft patency (Fig. 2A–B). The symptoms improved by postoperative day 6 and she was discharged with plans to revascularize her right hemisphere. 3. Discussion Cerebral hyperperfusion syndrome (CHS), most often defined as a symptomatic increase in cerebral blood flow, has been reported in 0.4–38% of patients across all methods of carotid artery revascularization [1–3]. A recent patient series reported the incidence of CHS specifically after high flow extracranial-intracranial bypass to be 4%, while other patient series examining hyperemia reported no incidences [4,5]. In contrast, studies examining cerebral ischemia explicitly after high flow bypass reported that 60% of patients demonstrated transient elevations of mean CBF postoperatively with 17–23% found to have intracerebral hyperemia with hemorrhage [6–8]. However, to our knowledge, there are no reports of symptomatic cerebral hyperemia in the posterior circulation after high flow bypass into the MCA territory. Although CBF increased in the left MCA territory, the perfusion evidence of hyperemia was only seen in the posterior circulation. Our patient’s preoperative angiogram showed significant filling of the bilateral MCA territories via the posterior communicating arteries (Fig. 2C–D). Given the watershed infarcts, it is likely that the posterior circulation was maximally dilated for some time and likely had dysfunction
of the normal autoregulatory mechanisms. Additionally, the vertebrobasilar system has decreased sympathetic innervation in comparison to other vascular cerebral territories, further explaining the hyperemic pattern in our patient [1,9]. Regarding the timing of hyperemic complications, Stiver et al. described a patient with CHS which occurred intraoperatively during a high flow bypass for carotid stenosis [10]. In light of their experience, our patient’s intraoperative neurophysiological changes may have been a manifestation of cerebral hyperemia. Although patients with severe cases may progress to intracerebral hemorrhage, hyperemic symptoms often resolve once vigilant blood pressure control is instituted with care taken to avoid cerebral vasodilating medications such as hydralazine, nitrates, and calcium channel blockers which may worsen cerebral edema [1,2]. Cerebral edema and seizures should be treated in the usual fashion as no data exists for the treatment of these entities in the context of CHS [1,2]. 4. Conclusion A high index of suspicion for cerebral hyperemia should be maintained both during and after high flow extracranial-intracranial bypass, especially in patients with chronic cerebral ischemia. The woman reported here highlights the potential for hyperemic complications outside the revascularized arterial territory. Improving our understanding of CHS may assist in identifying at risk patients
1518
Case Reports / Journal of Clinical Neuroscience 22 (2015) 1518–1520
and at risk arterial territories in order to optimize CHS prevention and management strategies. Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. Appendix A. Supplementary material Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.jocn.2015.03.022. References [1] van Mook WN, Rennenberg RJ, Schurink GW, et al. Cerebral hyperperfusion syndrome. Lancet Neurol 2005;4:877–88. [2] Moulakakis KG, Mylonas SN, Sfyroeras GS, et al. Hyperperfusion syndrome after carotid revascularization. J Vasc Surg 2009;49:1060–8.
[3] Yamaguchi K, Kawamata T, Kawashima A, et al. Incidence and predictive factors of cerebral hyperperfusion after extracranial-intracranial bypass for occlusive cerebrovascular diseases. Neurosurgery 2010;67:1548–54 [discussion 1554]. [4] Morton RP, Moore AE, Barber J, et al. Monitoring flow in extracranialintracranial bypass grafts using duplex ultrasonography: a single-center experience in 80 grafts over 8 years. Neurosurgery 2014;74:62–70. [5] Ishishita Y, Tanikawa R, Noda K, et al. Universal extracranial-intracranial graft bypass for large or giant internal carotid aneurysms: techniques and results in 38 consecutive patients. World Neurosurg 2014;82:130–9. [6] Ishikawa T, Kamiyama H, Houkin K, et al. Postsurgical observations of mean hemispheric cerebral blood flow with patients receiving high-flow EC-IC bypass using a radial artery graft (preliminary report, one-year observation of 10 hemispheres). Surg Neurol 1995;43:500–6 [discussion 506–9]. [7] Sundt Jr TM, Piepgras DG, Marsh WR, et al. Saphenous vein bypass grafts for giant aneurysms and intracranial occlusive disease. J Neurosurg 1986;65: 439–50. [8] Diaz FG, Pearce J, Ausman JI. Complications of cerebral revascularization with autogenous vein grafts. Neurosurgery 1985;17:271–6. [9] Schwartz RB. Hyperperfusion encephalopathies: hypertensive encephalopathy and related conditions. Neurologist 2002;8:22–34. [10] Stiver SI, Ogilvy CS. Acute hyperperfusion syndrome complicating EC-IC bypass. J Neurol Neurosurg Psychiatry 2002;73:88–9.
http://dx.doi.org/10.1016/j.jocn.2015.03.022
Cauda equina syndrome caused by retained hemostatic agents Matthew C. Bessette, Addisu Mesfin ⇑ Department of Orthopaedics and Rehabilitation, University of Rochester Medical Center, 601 Elmwood Ave, Box 665, Rochester, NY 14642, USA
a r t i c l e
i n f o
Article history: Received 18 November 2014 Accepted 3 March 2015
Keywords: Cauda equina syndrome Complications Gelfoam Hemostatic agents Surgicel
a b s t r a c t We report a patient with cauda equina syndrome secondary to retained Surgicel (Ethicon: Johnson & Johnson Medical, Piscataway, NJ, USA) and Gelfoam (Pfizer, New York, NY, USA). Retained hemostatic agents such as these used in spinal surgery can create space occupying lesions that lead to neurological compromise. There are few reports of neurologic symptoms caused by hemostatic devices used in surgery despite their widespread use. Our patient had routine lumbar microdiscectomy with preexisting motor deficits, and the acute postoperative course was complicated by a decline in neurologic function. The findings at the time of reoperation confirmed compression due to Gelfoam and Surgicel in the spinal canal, and subjective and objective symptoms partially improved after reoperation. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction Difficult exposure, osteotomies, epidural venous plexi, and a low tolerance for hematoma formation are among the many variables that make hemostasis in spinal surgery both difficult and imperative. Various agents have been introduced over the years to help surgeons successfully control bleeding. While their use has been instrumental in many advances made in the field, this is not without unintended consequences. We report a routine lumbar microdiscectomy and foraminal decompression complicated by postoperative neurologic changes. Reoperation and wound exploration revealed retained Surgicel (Ethicon: Johnson & Johnson Medical, Piscataway, NJ, USA) and Gelfoam (Pfizer, New York, NY, USA) material around the thecal sac with no evidence of hematoma formation. 2. Case report A 62-year-old, otherwise healthy man presented with a 4 month history of gradually worsening radicular pain and ⇑ Corresponding author. Tel.: +1 585 275 5196; fax: +1 585 756 4726. E-mail address: addisu_mesfi[email protected] (A. Mesfin).
subjective numbness in his right leg. On examination, he had 4/5 motor strength on knee extension, 3/5 ankle dorsiflexion, 1/5 great toe extension and 4/5 ankle plantarflexion. Treatment with physical therapy, anti-inflammatory medications, gabapentin (Neurontin; Pfizer) and transforaminal nerve root injections was unsuccessful. MRI revealed a central L5–S1 herniated disc abutting the S1 nerve root and an extruded right central herniated disc at L4–L5 abutting the L5 nerve root (Fig. 1). The patient underwent a right L4–L5 microdiscectomy and a right L5–S1 foraminal decompression. This was complicated by a durotomy which was repaired with absorbable sutures. Hemostasis, which was noted to be particularly difficult during the surgery, was aided with the use of Surgicel and Gelfoam. At the time of his first postoperative evaluation, the man reported worsening numbness in the right L5 and S1 distributions as well as new numbness in his right buttock. He also reported diminished sensation during voiding which he was still able to control. His motor strength was unchanged. One day after surgery, significant changes included 1/5 ankle dorsiflexion and stress incontinence. An urgent MRI was initially interpreted by the neuroradiologist as concerning for epidural hematoma (Fig. 2). He was taken back to the operating room for an exploration of the wound. Findings included Gelfoam and
