Neurosurgical Review

, Volume 40, Issue 3, pp 507–512 | Cite as

Uneven cerebral hemodynamic change as a cause of neurological deterioration in the acute stage after direct revascularization for moyamoya disease: cerebral hyperperfusion and remote ischemia caused by the ‘watershed shift’

  • Xian-kun Tu
  • Miki FujimuraEmail author
  • Sherif Rashad
  • Shunji Mugikura
  • Hiroyuki Sakata
  • Kuniyasu Niizuma
  • Teiji Tominaga
Case Report


Superficial temporal artery-middle cerebral artery (STA-MCA) anastomosis is the standard surgical treatment for moyamoya disease (MMD). The main potential complications of this treatment are cerebral hyperperfusion (CHP) syndrome and ischemia, and their managements are contradictory to each other. We retrospectively investigated the incidence of the simultaneous manifestation of CHP and infarction after surgery for MMD. Of the 162 consecutive direct revascularization surgeries performed for MMD, we encountered two adult cases (1.2%) manifesting the simultaneous occurrence of symptomatic CHP and remote infarction in the acute stage. A 47-year-old man initially presenting with infarction developed CHP syndrome (aphasia) 2 days after left STA-MCA anastomosis, as assessed by quantitative single-photon emission computed tomography (SPECT). Although lowering blood pressure ameliorated his symptoms, he developed cerebral infarction at a remote area in the acute stage. Another 63-year-old man, who initially had progressing stroke, presented with aphasia due to focal CHP in the left temporal lobe associated with acute infarction at the tip of the left frontal lobe 1 day after left STA-MCA anastomosis, when SPECT showed a paradoxical decrease in cerebral blood flow (CBF) in the left frontal lobe despite a marked increase in CBF at the site of anastomosis. Symptoms were ameliorated in both patients with the normalization of CBF, and there were no further cerebrovascular events during the follow-up period. CHP and cerebral infarction may occur simultaneously not only due to blood pressure lowering against CHP, but also to the ‘watershed shift’ phenomenon, which needs to be elucidated in future studies.


Moyamoya disease Surgical complication Cerebral hyperperfusion Cerebral infarction 


Compliance with ethical standard

Conflict of interest

The authors declare that they have no conflict of interest.

Research involving human participants and/or animals

The 123I-IMP-SPECT, MRI and MRA are routine postoperative examinations, which are covered by medical insurance in the Japanese healthcare system. All procedures are performed in accordance with the Declaration of Helsinki.

Informed consent

Informed consent to publish their clinical courses and radiological data was obtained from two individual patients included in the study.


This study was supported by JSPS KAKENHI Grant Number 426462150 from Ministry of Health, Labour and Welfare, Japan.

Financial and material support

This study was supported by JSPS KAKENHI Grant Number 426462150.


  1. 1.
    Suzuki J, Takaku A (1969) Cerebrovascular “moyamoya” disease. Disease showing abnormal net-like vessels in base of brain. Arch Neurol 20:288–299CrossRefPubMedGoogle Scholar
  2. 2.
    Research Committee on the Pathology and Treatment of Spontaneous Occlusion of the Circle of Willis; Health Labour Sciences Research Grant for Research on Measures for Infractable Diseases (2012) Guidelines for diagnosis and treatment of moyamoya disease (spontaneous occlusion of the circle of Willis). Neurol Med Chir (Tokyo) 52:245–266CrossRefGoogle Scholar
  3. 3.
    Fujimura M, Tominaga T (2015) Current status of revascularization surgery for moyamoya disease: special consideration for its “internal carotid-external carotid (IC-EC) conversion” as the physiological reorganization system. Tohoku J Exp Med 236:45–53CrossRefPubMedGoogle Scholar
  4. 4.
    Fujimura M, Inoue T, Shimizu H, Saito A, Mugikura S, Tominaga T (2012) Efficacy of prophylactic blood pressure lowering according to a standardized postoperative management protocol to prevent symptomatic cerebral hyperperfusion after direct revascularization surgery for moyamoya disease. Cerebrovasc Dis 33:436–445CrossRefPubMedGoogle Scholar
  5. 5.
    Fujimura M, Niizuma K, Inoue T et al (2014a) Minocycline prevents focal neurological deterioration due to cerebral hyperperfusion after extracranial-intracranial bypass for moyamoya disease. Neurosurgery 74:163–170CrossRefPubMedGoogle Scholar
  6. 6.
    Hayashi T, Shirane R, Fujimura M, Tominaga T (2010) Postoperative neurological deterioration in pediatric moyamoya disease: watershed shift and hyperperfusion. J Neurosurg Pediatr 6:73–81CrossRefPubMedGoogle Scholar
  7. 7.
    Fujimura M, Shimizu H, Inoue T, Niizuma K, Tominaga T (2014b) Issues in revascularization surgery for elderly patients with moyamoya disease. Surg Cereb Stroke 42:37–41 (in Japanese)CrossRefGoogle Scholar
  8. 8.
    Fujimura M, Kaneta T, Mugikura S, Shimizu H, Tominaga T (2007) Temporary neurologic deterioration due to cerebral hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in patients with adult-onset moyamoya disease. Surg Neurol 67:273–282CrossRefPubMedGoogle Scholar
  9. 9.
    Fujimura M, Shimizu H, Mugikura S, Tominaga T (2009b) Delayed intracerebral hemorrhage after superficial temporal artery-middle cerebral artery anastomosis in a patient with moyamoya disease: possible involvement of cerebral hyperperfusion and increased vascular permeability. Surg Neurol 71:223–227CrossRefPubMedGoogle Scholar
  10. 10.
    Kim JE, Oh CW, Kwon OK, Park SQ, Kim SE, Kim YK (2008) Transient hyperperfusion after superficial temporal artery/middle cerebral artery bypass surgery as a possible cause of postoperative transient neurological deterioration. Cerebrovasc Dis 25:580–586CrossRefPubMedGoogle Scholar
  11. 11.
    Fujimura M, Mugikura S, Kaneta T, Shimizu H, Tominaga T (2009a) Incidence and risk factors for symptomatic cerebral hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in patients with moyamoya disease. Surg Neurol 71:442–447CrossRefPubMedGoogle Scholar
  12. 12.
    Uchino H, Kuroda S, Hirata K, Shiga T, Houkin K, Tamaki N (2012) Predictors and clinical features of postoperative hyperperfusion after surgical revascularization for moyamoya disease: a serial single photon emission CT/positron emission tomography study. Stroke 43:2610–2616CrossRefPubMedGoogle Scholar

Copyright information

© Springer-Verlag Berlin Heidelberg 2017

Authors and Affiliations

  • Xian-kun Tu
    • 1
    • 2
  • Miki Fujimura
    • 1
    Email author
  • Sherif Rashad
    • 1
  • Shunji Mugikura
    • 3
  • Hiroyuki Sakata
    • 1
  • Kuniyasu Niizuma
    • 1
  • Teiji Tominaga
    • 1
  1. 1.Department of NeurosurgeryTohoku University Graduate School of MedicineSendaiJapan
  2. 2.Department of NeurosurgeryFujian Medical University Union HospitalFuzhouChina
  3. 3.Department of RadiologyTohoku University Graduate School of MedicineSendaiJapan

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