Anesth Pain Med 2016; 11: 155-159 http://crossmark.crossref.org/dialog/?doi=10.17085/apm.2016.11.￭Case2.155&domain=pdf&date_stamp=2016-04-25 Report￭ http://dx.doi.org/10.17085/apm.2016.11.2.155 pISSN 1975-5171ㆍeISSN 2383-7977 Anesthetic experience of Benedikt syndrome complicating lumbar spine involved multiple myeloma -A case report- Department of Anesthesiology and Pain Medicine, Gachon University Gil Medical Center, Incheon, *Department of Anesthesiology and Pain Medicine, Ajou University College of Medicine, Suwon, Korea Ji Yeon Lee, Wol Seon Jung, Se Ryeon Lee*, and Youn Yi Jo Benedikt syndrome is characterized by ipsilateral ophthalmoplegia anesthetic management remains challenging because of the with contralateral hemichorea due to a midbrain lesion. A neurologic medications being taken, which might affect 67-year-old male with Benedikt syndrome underwent corpectomy hemodynamics and anesthesia depth. In particular, hypotension at L1 and anterolateral interbody fusion at T12-L2 due to pathologic bursting fracture at L1 involving multiple myeloma. He had a history and hypertension could aggravate the underlying brain lesion, of traumatic subarachnoid hemorrhage and subdural hemorrhage and thus, strict hemodynamic control is essential. In a previous 8 months before surgery. Magnetic resonance image of the brain report of hemiballism-hemichorea, only 3 minutes of hypo- revealed intracranial hemorrhage from thalamus to midbrain. Target tension led to focal cerebral ischemia involving thalamus, pons, controlled infusion with propofol and remifentanil were administered for anesthetic induction and maintenance and close hemodynamic and midbrain [2]. and neurologic monitoring led to successful anesthetic management. No previous report has been issued on the anesthetic (Anesth Pain Med 2016; 11: 155-159) management of Benedikt syndrome in the English literature. Key Words: Anesthesia, Midbrain hemorrhage, Trauma. Here, we describe general anesthetic management for spine fusion in a patient with mild hemiparesis with involuntary choreic movement and ptosis with diplopia after traumatic Benedikt syndrome is characterized by ipsilateral oculomotor brain injury. nerve palsy and contralateral hemiparesis with involuntary movement caused by a midbrain lesion [1]. CASE REPORT In 1889, Moritz Benedikt first described a patient with oculomotor paralysis and crossed hemiparesis with tremor. This A 67-year-old male (height 163 cm, weight 56 kg) was rare syndrome is caused by infarction, hemorrhage, and a scheduled for corpectomy at L1 and anterolateral interbody midbrain tumor, but a trauma origin is rare. Only one case of fusion at T12-L2 due to a pathologic bursting fracture at L1 posttraumatic Benedikt syndrome has been reported in the attributed to multiple myeloma. He suffered from back pain English literature [1]. Due to the rarity of this condition, and was diagnosed with lumbar spine involvement by multiple myeloma. Eight months before surgery, he experienced traumatic Received: September 30, 2015. subarachnoid hemorrhage, subdural hemorrhage, and intracranial Revised: 1st, November 9, 2015; 2nd, December 4, 2015. hemorrhage (Glasgow coma scale, 14) due to a fall down. Accepted: December 4, 2015. During admission for traumatic brain injury, he complained of Corresponding author: Youn Yi Jo, M.D., Ph.D., Department of dysphagia, dysarthria, involuntary movement and tremor in the Anesthesiology and Pain Medicine, Gachon University, Gil Medical Center, left arm and hand with grade 3 motor weakness. In addition, 21, Namdong-daero 774beon-gil, Namdong-gu, Incheon 21565, Korea. Tel: 82-32-460-3636, Fax: 82-32-469-6319, E-mail: [email protected] he suffered from right sided diplopia and ptosis. Brain magnetic resonance imaging showed right frontal base occipital This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) hemorrhage with bilateral temporal hemorrhage, and intracranial which permits unrestricted non-commercial use, distribution, and reproduction in any hemorrhage on thalamus to midbrain (Fig. 1). His symptoms medium, provided the original work is properly cited. of involuntary upper limb movement and diplopia with ptosis 155 156 Anesth Pain Med Vol. 11, No. 2, 2016 Fig. 1. Precontrast T1 (A) and T2 (B) magnetic resonance images showing hemorrhage in right mid brain (arrow). persisted until admission for spine surgery. He was taking 10 mmHg using intermittent nicardipine, phenylephrine, and haloperidol (23 mg twice daily) and galantamine hydrobromide ephedrine administration (Fig. 2). At 4 hours 45 minutes after (16 mg twice daily). Preoperative laboratory findings revealed starting operation, blood pressure and central venous pressure blood urea nitrogen (BUN)/creatinine 86/8.4 mg/dl, potassium dropped to 80/40 mmHg and 2 mmHg and restored within 1 6.5 mEq/L, calcium 11.1 mg/dl, hematocrit 16%, and a platelet min after injection of phenylephrine and ephedring. At 5 hours count of 138 × 103/mm. after starting operation, blood pressure and central venous He was diagnosed as having multiple myeloma involving pressure re-dropped to 40/34 mmHg and 0 mmHg after 2000 kidney and spine, and received hemodialysis and 4 units of ml of abrupt surgical bleeding and ephedrine, phenylephrine packed red blood cells to correct a hematocrit level of 28% and epinephrine were injected. And packed red blood cell were immediately before surgery. infused via rapid infusion system and restored the hemo- The patient was not premedicated. In the operating room, dynamic variables gradually and reached to 129/70 mmHg noninvasive blood pressure, bispectral index, pulse oximetry, about 10 min after injecting vasopressors. End tidal CO2 was and electrocardiogram monitors were applied. Initial vital signs maintained between 33 and 40 mmHg and arterial blood were BP 149/81 mmHg and HR 73 beats/min. After local gasses were analyzed (Table 1). Motor evoked potential and infiltration, a 20 G left radial arterial line was inserted and neuromuscular monitoring using train of four ratio were blood pressure was monitored continuously. Propofol 5 ug/ml monitored during the 9-hour operation. Estimated blood loss and remifentanil 5 ng/ml (dosages were determined using the was 3000 ml and 9 units of packed red blood cell and 4300 Schnider model and the Minto model, respectively) were ml of infused fluid were transfused. Postoperatively, pa- administered using a target-controlled infusion device tient-controlled analgesia using fentanyl 800 ug in a total (Orchestra, Fresenius Vial, Brezins, France) for anesthesia volume of 100 ml connected to an intravenous line was used induction. Propofol was adjusted to maintain an effect-site for 48 hours, and palonosetron was administered to prevent concentration of 2.0–3.5 ug/ml and bispectral index (BIS) postoperative nausea and vomiting. values between 40 and 50, and remifentanil was adjusted to After arrival in the surgical intensive care unit, his mean maintain an effect-site concentration of 0.5–3.0 ng/ml to blood pressure was controlled at 75–85 mmHg. There were no maintain a mean arterial pressure within 20% of baseline. adverse neurologic signs and no changes were detected by Rocuronium 50 mg was administered to facilitate tracheal postoperative brain computed tomography. On postoperative day intubation and no more rocuronium was used during the 3, the patient was transferred to a general ward. On operation. postoperative day 37, velcade, melphalan, and prednisolone Mean arterial and central venous pressure via the right chemotherapy was started, and on postoperative day 46, sudden internal jugular vein were maintained at 75–85 mmHg and 7– dyspnea and desaturation occurred due to aspiration ascribed to Ji Yeon Lee, et al：Anesthesia for Benedikt syndrome 157 Fig. 2. Hemodynamic drug and transfusion requirements. RBC: packed red blood cell, FFP: fresh frozen plasma. Table 1. Arterial Blood Gas Analysis during Anesthesia Hours after anesthetic induction Postanesthetic care unit 1246 pH 7.42 7.49 7.36 7.35 7.31 PaCO2 (mmHg) 40 33 37 37 40 PaO2 (mmHg) 279 300 347 324 175 Hematocrit (%)2627232231 a pre-existing swallowing difficulty. He expired on postope- midbrain injury. rative day 56. Benedikt syndrome has been called paramedian midbrain syndrome or red nucleus syndrome. The midbrain is near the DISCUSSION cerebral cortex, cerebellum, and brain stem, and plays a significant role in visual and auditory processing and motor Our patient suffered from involuntary limb movement with control [4]. Furthermore, a lesion involving tegmentum of the mild hemiparesis and diplopia with ptosis after traumatic brain midbrain produces oculomotor nerve palsy, contralateral injury. For general anesthesia, we used a propofol and hemiparesis, and involuntary movement and is usually induced remifentanil target controlled infusion system. After considering by infarction of posterior cerebral artery branches [1,4]. the patient’s underlying neurologic condition and the drugs Bendikt syndrome is diagnosed based on clinical symptoms of being taken, we considered close hemodynamic and neuro- oculomotor palsy and contralateral hemiparesis with involuntary muscular monitoring could lead to successful anesthetic movement and a midbrain involving lesion by imaging. Our management. Furthermore, because the midbrain is critical for patient experienced typical symptoms of Benedikt syndrome controlling hemodynamic response to blood loss,