Surgical Neurology 68 (2007) 335 – 337 www.surgicalneurology-online.com
Vascular
Intracerebral hemorrhage after prophylactic revascularization in a patient with adult moyamoya disease Yasunari Otawara, MDa,4, Kuniaki Ogasawara, MDa, Kaoru Seki, MDb, Michihiro Kibe, MDb, Yoshitaka Kubo, MDa, Akira Ogawa, MDa a
Department of Neurosurgery, Iwate Medical University, Morioka, Iwate, 020-8505 Japan b Department of Neurosurgery, Kamaishi Hospital, Kamaishi, Iwate, 026-8550 Japan Received 13 September 2006; accepted 19 October 2006
Abstract
Background: The effect of revascularization surgery for a patient with moyamoya disease remains controversial. Case Description: A 60-year-old man presented with bleeding from asymptomatic moyamoya vessels 10 years after prophylactic revascularization surgery. Cerebral angiography 10 years after the surgery demonstrated that the bilateral direct anastomoses remained effective and a small aneurysm persisted in the anterior choroidal artery. The territories of perfusion through the anastomoses and the size of the aneurysm remained unchanged compared with the angiograms performed 10 years ago. Conclusions: Direct revascularization surgery may not always resolve microaneurysms in the moyamoya vessels and prevent rebleeding in patients with hemorrhagic moyamoya disease or bleeding in the nonaffected side. D 2007 Elsevier Inc. All rights reserved.
Keywords:
Moyamoya disease; Revascularization; Microaneurysm
1. Introduction Moyamoya disease is a chronic occlusive cerebrovascular disease characterized by specific angiographical findings of a net-like cluster of vessels at the base of the brain [5]. The initial symptom is generally cerebral ischemia in children and intracerebral hemorrhage in adults. Revascularization surgery for patients with hemorrhagic moyamoya disease is intended to reduce the hemodynamic stress on the moyamoya vessels and may also provide secondary prevention of stroke in the symptomatic moyamoya vessels ipsilateral to the hemorrhagic hemisphere [1,2,6]. In contrast, the effect of the surgery as a primary prevention of stroke for asymptomatic moyamoya vessels contralateral to the hemorrhagic hemisphere remains controversial.
Abbreviations: CT, computed tomography; MCA, middle cerebral artery; STA, superficial temporal artery. 4 Corresponding author. Tel.: +81 19 651 5111; fax: +81 19 625 8799. E-mail address:
[email protected] (Y. Otawara). 0090-3019/$ – see front matter D 2007 Elsevier Inc. All rights reserved. doi:10.1016/j.surneu.2006.10.061
We experienced a case of an adult patient with hemorrhage from asymptomatic moyamoya vessels 10 years after prophylactic revascularization surgery. 2. Case report A 60-year-old man without a history of hypertension had a sudden onset of severe headache. The CT on admission demonstrated hemorrhage in the left basal ganglia with ventricular hemorrhage. Cerebral angiography revealed severe occlusive changes in the bilateral carotid forks associated with moyamoya vessels, and a small aneurysm in the plexal segment of the right anterior choroidal artery. No aneurysms were detected in the left cerebral hemisphere ipsilateral to the hemorrhage. The patient underwent bilateral STA-MCA anastomosis and encephaloduroarteriomyosynangiosis. The postoperative course was uneventful. Cerebral angiography performed 3 months after the surgery revealed perfusion in the bilateral whole MCA territories through the anastomoses from the STAs (Fig. 1).
336
Y. Otawara et al. / Surgical Neurology 68 (2007) 335 – 337
Fig. 1. A: Right anteroposterior carotid angiogram obtained 3 months after surgery showing a small aneurysm in the plexal segment of the anterior choroidal artery (arrow). B: Right lateral external carotid angiogram obtained 3 months after surgery showing perfusion in the whole MCA territory through the anastomosis from the STA.
Ten years later, he again had a sudden onset of severe headache. The CT on admission revealed hemorrhage in the right basal ganglia with ventricular hemorrhage (Fig. 2). Cerebral angiography demonstrated that the bilateral direct anastomoses remained effective, and the small aneurysm persisted in the plexal segment of the right anterior choroidal artery (Fig. 3). The territories of perfusion through the anastomoses and the size of the aneurysm remained unchanged. He was medically treated and returned to his previous life without surgical intervention.
anastomoses. Therefore, direct revascularization surgery may not always resolve microaneurysms in the moyamoya vessels and prevent rebleeding in patients with hemorrhagic moyamoya disease or bleeding in the nonaffected side.
3. Discussion The present patient had undergone revascularization surgery for secondary prevention of stroke on the affected side and primary prevention of stroke on the nonaffected side. Rebleeding after direct revascularization surgery including STA-MCA anastomosis was reported in 3 patients [1] who experienced intracerebral hemorrhage, but other patients had no subsequent hemorrhage [2,6]. Of these 3 patients, 1 had hemorrhage on the nonaffected side after direct revascularization surgery, as in our patient. Revascularization surgery for moyamoya disease may reduce hemodynamic stress and may result in the resolution of microaneurysms located in the moyamoya vessels [3]. In the present case, the small aneurysm in the plexal segment of the anterior choroidal artery had not resolved 10 years after revascularization surgery and caused bleeding despite perfusion in the entire MCA territories through the
Fig. 2. Computed tomography scan on the second admission revealing hemorrhage in the right basal ganglia with ventricular hemorrhage.
Y. Otawara et al. / Surgical Neurology 68 (2007) 335 – 337
337
Fig. 3. A: Right anteroposterior carotid angiogram obtained 10 years after surgery showing a small aneurysm in the plexal segment of the anterior choroidal artery (arrow). The size of the aneurysm was unchanged. B: Right lateral carotid angiogram obtained 10 years after surgery showing perfusion in the whole MCA territory through the anastomosis from the STA.
A prospective randomized controlled trial is now ongoing in Japan to determine the effect of bilateral direct revascularization surgery for adult hemorrhagic moyamoya disease [4].
References [1] Houkin K, Kamiyama H, Abe H, et al. Surgical therapy for adult moyamoya disease. Can surgical revascularization prevent the recurrence of intracerebral hemorrhage? Stroke 1996;27:1342 - 6. [2] Kawaguchi S, Okuno S, Sakaki T. Effect of direct arterial bypass on the prevention of future stroke in patients with the hemorrhagic variety of moyamoya disease. J Neurosurg 2000;93:397 - 401. [3] Kuroda S, Houkin K, Kamiyama H, et al. Effects of surgical revascularization on peripheral artery aneurysms in moyamoya disease: report of three cases. Neurosurgery 2001;49:463 - 8. [4] Miyamoto S. Study design for a prospective randomized trial of extracranial-intracranial bypass surgery for adults with moyamoya disease and hemorrhagic onset—the Japan Adult Moyamoya Trial Group. Neurol Med Chir (Tokyo) 2004;44:218 - 9. [5] Suzuki J, Takaku A. Cerebrovascular bmoyamoya disease.Q Disease showing abnormal net-like vessels in base of brain. Arch Neurol 1969; 20:288 - 99. [6] Yoshida Y, Yoshimoto T, Shirane R, et al. Clinical course, surgical management, and long-term outcome of moyamoya patients with
rebleeding after an episode of intracerebral hemorrhage: an extensive follow-up study. Stroke 1999;30:2272 - 6.
Commentary This article reports a representative case of a patient with adult moyamoya disease of hemorrhagic type in which bypass surgery after the first bleeding could not prevent the second bleeding. This is still an important unsolved problem concerning moyamoya disease. In Japan, the institutes with better bypass surgical technique seem to have better results in preventing rebleeding. Therefore, a prospective randomized controlled study in Japan has started with members with better surgical technique. Nevertheless, such a rebleeding case occurs after preventative bypass surgery. The authors’ intent appears to be not to deny the preventing effect of the bypass surgery as a whole but to point out that such rebleeding can occur. The report of the controlled study is awaited. Masashi Fukui, MD Sasebo Kyosai Hospital Sasebo 857-8575, Japan