Case Report

Cerebral Amyloid Angiopathy Causing Large Contralateral Hemorrhage During Surgery for Lobar Hemorrhage: A Case Report Hidetaka Arishima, MD, Hiroyuki Neishi, MD, Toshiaki Kodera, MD, Ryuhei Kitai, MD, and Ken-ichiro Kikuta, MD

We report a rare case of cerebral amyloid angiopathy (CAA) causing large contralateral hemorrhage during surgery for lobar hemorrhage. A 62-year-old woman presented with lobar hemorrhage in the left frontal and parietal lobes recurring over the previous 1 month. Because we could not detect the origin of the lobar hemorrhage, we performed a biopsy around the lobar hemorrhage site with the removal of a hematoma. During the surgery, we identified acute brain swelling without bleeding from the operative field. Intraoperative computed tomography demonstrated new large lobar hemorrhage of the right parietal lobe, which we could promptly remove. Specimens around hematomas on both sides were pathologically diagnosed as CAA on immunohistochemical examination. After the surgery, she suffered from lobar hemorrhage three times in the space of only 3 months. To the best of our knowledge, there has been no reported case of CAA causing intracranial hemorrhage of another lesion during surgery. Neurosurgeons should know a possibility of intraoperative hemorrhage in surgeries for lobar hemorrhage caused by CAA. Key Words: Cerebral amyloid angiopathy—intraoperative computed tomography—intraoperative hemorrhage—lobar hemorrhage— recurrent hemorrhage. Ó 2015 by National Stroke Association

Cerebral amyloid angiopathy (CAA) is a cerebrovascular disorder characterized by the deposition of b-amyloid protein in the cortical and leptomeningeal vessels, increasing with advancing age.1-4 CAA is thought to be

From the Department of Neurosurgery, University of Fukui, Fukui, Japan. Received October 2, 2014; revision received November 6, 2014; accepted November 18, 2014. Address correspondence to Hidetaka Arishima, MD, Department of Neurosurgery, University of Fukui 23-3, Matsuokashimoaizuki, Eiheiji-cho, Yoshida-gun, Fukui 910-1193, Japan. E-mail: [email protected] 1052-3057/$ - see front matter Ó 2015 by National Stroke Association http://dx.doi.org/10.1016/j.jstrokecerebrovasdis.2014.11.015

a major cause of lobar hemorrhage in the elderly.3,5,6 Some cases have been reported as recurrent intracranial hemorrhage.7-9 Here, we report a case of definite CAA causing intracranial hemorrhage on the opposite side during surgery for biopsy, with removal of the lobar hemorrhage.

Case Report A healthy 62-year-old woman presented with headache and right mild hemiparesis. Her consciousness was almost clear, and she did not have dementia. She had neither hypertension nor family history of cerebral apoplexy. She took no medicine and did not have a habit of smoking and drinking. One month earlier, she had suffered from lobar hemorrhage of the left frontal lobe

Journal of Stroke and Cerebrovascular Diseases, Vol. -, No. - (---), 2015: pp e1-e3

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Figure 1. (A) CTshowing the first lobar hemorrhage before admission. (B) CTshowing the second lobar hemorrhage on admission. (C) T2* image demonstrating only one siderosis (arrow) of the left occipital lobe. (D, E) Intraoperative CTshowing new large hemorrhage on the contralateral side of the surgical lesion. (F, G, E) CT after the operation demonstrating at least 3 signs of intracranial hemorrhage at different sites within 3 months. Abbreviation: CT, computed tomography.

(Fig 1, A), which caused no neurologic deficit. Computed tomography (CT) showed another lobar hemorrhage of the left parietal lobe (Fig 1, B). Hematologic and cardiac examination revealed no causes of the cerebral hemorrhage. Digital subtraction angiography also identified no abnormal lesions. Magnetic resonance imaging showed known hematomas in the left frontal and parietal lobes. However, only 1 T2* image showed siderosis of the left occipital lobe (Fig 1, C). We decided to perform open biopsy with removal of a hematoma in the left parietal lobe to confirm the pathologic diagnosis. Under general anesthesia in a prone position, we performed surgery with a small craniotomy. At the beginning of the operation, the systolic blood pressure transiently rose to 180 mm Hg when we fixed her head with Mayfield Head Frame (Integra Lifesciences, Plainsboro, NJ) and we made the skin incision. Directly after taking some specimens, we identified acute brain swelling without bleeding from the operative field. Fortunately, we conducted surgery in the intraoperative CT room; therefore, we could promptly perform intraoperative CT, which demonstrated new large lobar hemorrhage of the right parietal lobe (Fig 1, D,E). We removed the right lobar hemorrhage with large decompressive craniectomy and took some cortical and arterial tissues from around the hematoma. Histopathologic examination of the surgical specimens from both lesions showed hyalinization of the adventitia

and outer media in cortical and leptomeningeal arteries, which were strongly positive for anti–b-amyloid protein antibody. These pathologic findings corresponded with CAA (Fig 2, A,B,C,D). Her postoperative state was comatose, and subsequent CT demonstrated at least 3 signs of intracranial hemorrhage (Fig 1, F,G,H) at different sites within 3 months. She died 16 months after operation without recovery of her consciousness.

Discussion Although some cases of probable or definite CAA have been reported as recurrent intracranial hemorrhage,7-9 to our knowledge, this is the first reported case of CAA causing large contralateral hemorrhage during surgery. Fortunately, we could identify this unexpected hemorrhage by intraoperative CT. Neurosurgeons should know a possibility of intraoperative hemorrhage in surgeries for lobar hemorrhage probably caused by CAA. We speculate that the acute elevation of blood pressure at the beginning of the operation may cause new lobar hemorrhage at another site during surgery. Intraoperative CT is useful for not only tumor surgery but also cerebrovascular surgery.10 Usually, we perform intraoperative CT to check the evacuation of intracranial hematoma; however, intraoperative CT immediately

CAA CAUSING LARGE HEMORRHAGE DURING SURGERY

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Figure 2. (A, B) Histopathologic examination of surgical specimens around the left lobar hematoma. Hematoxylin and eosin staining showing hyalinization of the adventitia and outer media in cortical and leptomeningeal arteries (A), which were strongly positive for anti–b-amyloid protein antibody (B). Magnification 3 100. (C, D) Histopathologic examination of surgical specimens around the right lobar hematoma. Hematoxylin and eosin staining showing hyalinization of the adventitia and outer media in a large leptomeningeal artery (C), which were strongly positive for anti–b-amyloid protein antibody (D). Magnification 3 100.

showed a new lobar hemorrhage at another site during the surgery in this case. Therefore, we think intraoperative CT also has a possibility to detect an unexpected occurrence during the surgery and help neurosurgeons. This case may be an instructive case to show the usefulness of intraoperative CT.

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References 1. Biffi A, Greenberg S. Cerebral amyloid angiopathy: a systematic review. J Clin Neurol 2011;7:1-9. 2. Charidimou A, Gang Q, Werring DJ. Sporadic cerebral amyloid angiopathy revisited: recent insights into pathophysiology and clinical spectrum. J Neurol Neurosurg Psychiatry 2012;83:124-137. 3. Revesz T, Ghiso J, Lashley T, et al. Cerebral amyloid angiopathies: a pathologic, biochemical, and genetic view. J Neuropathol Exp Neurol 2003;62:885-898. 4. Viswanathan A, Greenberg SM. Cerebral amyloid angiopathy in the elderly. Ann Neurol 2011;70:871-980. 5. Hirohata M, Yoshita M, Ishida C, et al. Clinical features of non-hypertensive lobar intracerebral hemorrhage related

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to cerebral amyloid angiopathy. Eur J Neurol 2010; 17:823-829. Pezzini A, Del Zotto E, Volonghi I, et al. Cerebral amyloid angiopathy: a common cause of cerebral hemorrhage. Curr Med Chem 2009;16:2498-2513. See comment in PubMed Commons below. Apoil M, Cogez J, Dubuc L, et al. Focal cortical subarachnoid hemorrhage revealed by recurrent paresthesias: a clinico-radiological syndrome strongly associated with cerebral amyloid angiopathy. Cerebrovasc Dis 2013;36: 139-144. Izumihara A, Suzuki M, Ishihara T. Recurrence and extension of lobar hemorrhage related to cerebral amyloid angiopathy: multivariate analysis of clinical risk factors. Surg Neurol 2005;64:160-164. Raposo N, Viguier A, Cuvinciuc V, et al. Cortical subarachnoid haemorrhage in the elderly: a recurrent event probably related to cerebral amyloid angiopathy. Eur J Neurol 2011;18:597-603. Fujisawa M, Yamashita S, Katagi R. Usefulness of intraoperative computed tomography for the evacuation of lobar hemorrhage. Acta Neurochir Suppl 2013;118: 175-179.

Cerebral amyloid angiopathy causing large contralateral hemorrhage during surgery for lobar hemorrhage: a case report.

We report a rare case of cerebral amyloid angiopathy (CAA) causing large contralateral hemorrhage during surgery for lobar hemorrhage. A 62-year-old w...
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