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Show Photo Essay Remote Pontine Hemorrhage After Left Frontal Lobe Meningioma Resection Presenting as Bilateral Internuclear Ophthalmoplegia Maxwell Lee, BA, Bayan A. Al Othman, MD, Tonse A. Kini, MD, Andrew G. Lee, MD FIG. 1. Axial T1 postcontrast MRI scan of the brain shows the left frontal extra-axial mass. Abstract: Postoperative hemorrhages are relatively common complications of surgical procedures including craniotomies, and these typically occur at or near the operative site. Bleeding in remote areas (e.g., posterior fossa) after supratentorial craniotomy can occur and may be associated with a high morbidity and mortality. Although remote cerebellar hemorrhage after craniotomy is well described, Baylor College of Medicine (ML), Houston, Texas; Department of Ophthalmology (BAAO, TAK, AGL), Blanton Eye Institute, Houston Methodist Hospital, Houston, Texas; Departments of Ophthalmology (AGL), Neurology, and Neurosurgery, Weill Cornell Medicine, New York, New York; Department of Ophthalmology (AGL), University of Texas Medical Branch, Galveston, Texas; Department of Ophthalmology (AGL), University of Texas MD Anderson Cancer Center, Houston, Texas; Department of Ophthalmology (AGL), Texas A and M College of Medicine, Bryan, Texas; and Department of Ophthalmology (AGL), The University of Iowa Hospitals and Clinics, Iowa City, Iowa. The authors report no conflicts of interest. Address correspondence to Andrew G. Lee, MD, Blanton Eye Institute, Houston Methodist Hospital, 6560 Fannin Street Suite 450, Houston, TX 77030; E-mail: aglee@houstonmethodist.org Lee et al: J Neuro-Ophthalmol 2020; 40: 417-419 remote pontine hemorrhage is less common. We describe a bilateral internuclear ophthalmoplegia due to an RPH after otherwise uncomplicated resection of a frontal meningioma. Clinicians should be aware that neuro-ophthalmic findings can occur from hemorrhages remote from the operative site. Journal of Neuro-Ophthalmology 2020;40:417-419 doi: 10.1097/WNO.0000000000000895 © 2020 by North American Neuro-Ophthalmology Society A 62-year-old previously healthy woman presented with numbness and tingling in her right hand and lips. Magnetic resonance (MR) and computed tomography (CT) scans of the brain showed a 4.5 cm · 2.9 cm, wellcircumscribed, and partially calcified extra-axial mass in the posterior left frontal lobe (Fig. 1). A left frontal craniotomy with gross total resection of the World Health Organization (WHO) grade I meningioma was performed. 417 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Photo Essay FIG. 2. Computerized tomography scan of the brain shows paramedian left dorsal midbrain hemorrhage. Two days after surgery, however, her husband found her unresponsive at home with a fixed and dilated pupil in the left eye. She was transferred to the Houston Methodist Hospital, and repeat CT scan showed interval hemorrhage into the left frontal surgical cavity and adjacent subdural space, adjacent vasogenic edema, rightward midline shift, left uncal herniation, hydrocephalus, and hemorrhage in the dorsal pons (Fig. 2). The hemorrhage occurred within 48 hours of the initial surgery. Subsequently, she underwent an emergent left decompressive hemicraniectomy and placement of a right fron- tal external ventricular drain (EVD). One month later, she underwent reconstructive cranioplasty. Her stay in the intensive care unit was complicated; that is, she was comatose and had to have a percutaneous endoscopic gastrostomy tube placement and tracheostomy and later amnesia of most of the events. Once she was in rehabilitation after her stay in the intensive care unit, she became aware of binocular horizontal diplopia, dizziness, and difficulty with balance. Neuro-ophthalmic examination showed visual acuity of 20/30 in both eyes (OU). The pupils were normal with no relative afferent pupillary defect. Automated perimetry (Humphrey Visual Field 24-2) showed right, denser superiorly, incongruous homonymous quadrantanopia consistent with the left temporal lobe resection. Motility examination showed a 22 underaction of adduction bilaterally (adduction lag OU) with a dissociated, horizontal, abducting nystagmus in right and left gaze consistent with a bilateral internuclear ophthalmoplegia (INO). The upgaze and downgaze were normal with no vertical nystagmus (Fig. 3). There was 10 prism diopter exotropia in primary gaze consistent with a "wall eyed" bilateral INO (WEBINO). Slit-lamp and dilated fundus examinations were unremarkable. No underlying vascular malformation (e.g., cavernous malformation) was seen in the patient. Serial CT and MR imaging studies showed the evolving hemorrhage in the dorsal pons that evolved to brainstem encephalomalacia over time. Resolution of the hemorrhage occurred over time leading to spontaneous recovery of the INO. Prism therapy alleviated the diplopia symptoms during the time of resorption of the hemorrhage, and at the last follow-up, the patient was straight in the primary position without residual diplopia. The remainder of her examination was normal. Remote cerebellar hemorrhage (RCH) and remote pontine hemorrhage (RPH) after supratentorial craniotomy are FIG. 3. External photographs demonstrating ocular motility. 418 Lee et al: J Neuro-Ophthalmol 2020; 40: 417-419 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Photo Essay rare with an incidence ranging from 0.08% to 0.6% (1). Preoperatively, there are a few discernible risk factors for RCH/RPH. One study found that the male gender may be a factor, but the mechanism remains ill defined (2). A history of cerebrovascular accident with cerebral atrophy has also been associated with a higher risk of developing RCH (3). Age however was found to not be a significant risk factor in RCH, with a range from 10 to 83 years old (1,2). Preexisting arterial hypertension, a common cause of spontaneous cerebellar hemorrhage, was not shown to predispose patients to RCH (2,4). The type of underlying lesion was also found to not be an important risk factor (2,5). One proposed mechanism for RCH/RPH is that rapid intraoperative drainage of cerebrospinal fluid (CSF) leads to cerebellar displacement caudally and results in stretching and shearing of cerebellar veins, causing the hemorrhage (1,2,5,6). Alternatively, continued postoperative lowering of CSF volume can decrease intracranial pressure, which increases risk of cerebellar herniation (3). Underlying coagulopathy has been proposed as a possible mechanism for RCH, but several studies have shown no conclusive evidence for coagulation defects in these patients (2,4,5). In the case of RPH, one study proposed that tumor resection led to traction on the perforating arteries from the basilar artery, leading to the shearing effect (7). Another proposed explanation is that intraoperative positioning, rotation, and stretching of the head can disturb venous drainage and cause brain displacement (8). Duret hemorrhages are linear areas of bleeding that can occur in the midbrain and upper pons due to traumatic downward displacement of the brainstem. In our case, rapid CSF decompression was performed using an EVD for obstructive hydrocephalus in addition to the emergent hemicraniectomy performed in the setting of raised ICP, significant mass effect, uncal herniation, and brain shift. It may be that the pontine hemorrhage in this case was multifactorial, however (1,4-6). Although demyelination, infarcts, hydrocephalus, and hypertensive hemorrhage have all been reported to produce WEBINO, to the best of our knowledge, this is the first case of bilateral INO due to RPH after supratentorial craniotomy in the English language ophthalmic literature. Given the close temporal relationship of the RPH and Lee et al: J Neuro-Ophthalmol 2020; 40: 417-419 surgery, we believe the proposed mechanisms are relevant in this discussion. Clinicians should be aware of the possibility of unilateral or bilateral INO as the presenting or only sign of RPH and the possible relationship to previous remote location supratentorial surgery, rapid ICP decompression, or a Duret hemorrhage (9,10). Although there are not necessarily proven preventative measures to be taken, clinicians should be aware of the clinicoradiographic presentations of RPH. STATEMENT OF AUTHORSHIP Category 1: a. Conception and design: B. A. Al Othman, T. A. Kini, and A. G. Lee; b. Acquisition of data: B. A. Al Othman, T. A. Kini, and A. G. Lee; c. Analysis and interpretation of data: B. A. Al Othman, T. A. Kini, and A. G. Lee. Category 2: a. Drafting the manuscript: B. A. Al Othman, T. A. Kini, and A. G. Lee; b. Revising it for intellectual content: B. A. Al Othman, T. A. Kini, and A. G. Lee. Category 3: a. Final approval of the completed manuscript: B. A. Al Othman, T. A. Kini, and A. G. Lee. REFERENCES 1. Smith R, Kebriaei M, Gard A, Thorell W, Surdell D. Remote cerebellar hemorrhage following supratentorial cerebrovascular surgery. J Clin Neurosci. 2014;21:673-676. 2. Marquardt G, Setzer M, Schick U, Seifert V. Cerebellar hemorrhage after supratentorial craniotomy. Surg Neurol. 2002;57:241-252. 3. Huang CY, Lee PH, Lin SH, Chuang MT, Sun YT, Hung YC, Lee EJ. Remote cerebellar hemorrhage following supratentorial craniotomy. Neurol Res. 2012;34:422-429. 4. Sturiale CL, Rossetto M, Ermanil M, Volpin F, Baro V, Milanese L, Denaro L, d'Avella D. Remote cerebellar hemorrhage after supratentorial procedures (part 1): a systematic review. Neurosurg Rev. 2016;39:565-573. 5. Das KK, Nair P, Mehotra A, Sardhara J, Sahu RN, Jaiswal AK, Kumar R. Remote cerebellar hemorrhage: report of 2 cases and review of literature. Asian J Neurosurg. 2014;93:161-164. 6. Stuart MJ, Tsahtsarlis A, Amato D, Pattavilakom A. Remote cerebellar haemorrhage from site of craniotomy: a report of two cases. Br J Neurosurg. 2016;30:101-103. 7. 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