{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T01:43:34Z","timestamp":1792806214158,"version":"4.5.0"},"reference-count":40,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"2","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,8]]},"abstract":"\n OBJECTIVE<\/jats:title>\n \n Superficial temporal artery\u2013middle cerebral artery (STA-MCA) anastomosis is the standard surgical management for moyamoya disease (MMD), whereas cerebral hyperperfusion (CHP) is one of the potential complications of this procedure that can result in delayed intracerebral hemorrhage and\/or neurological deterioration. Recent advances in perioperative management in the early postoperative period have significantly reduced the risk of CHP syndrome, but delayed intracerebral hemorrhage and prolonged\/delayed CHP are still major clinical issues. The clinical implication of\n RNF213<\/jats:italic>\n gene polymorphism c.14576G>A (rs112735431), a susceptibility variant for MMD, includes early disease onset and a more severe form of MMD, but its significance in perioperative pathology is unknown. Thus, the authors investigated the role of\n RNF213<\/jats:italic>\n polymorphism in perioperative hemodynamics after STA-MCA anastomosis for MMD.\n <\/jats:p>\n <\/jats:sec>\n \n METHODS<\/jats:title>\n \n Among 96 consecutive adult patients with MMD comprising 105 hemispheres who underwent serial quantitative cerebral blood flow (CBF) analysis by\n N<\/jats:italic>\n -isopropyl-\n p<\/jats:italic>\n -[\n 123<\/jats:sup>\n I]iodoamphetamine SPECT after STA-MCA anastomosis, 66 patients consented to genetic analysis of\n RNF213<\/jats:italic>\n . Patients were routinely maintained under strict blood pressure control during and after surgery. The local CBF values were quantified at the vascular territory supplied by the bypass on postoperative days (PODs) 1 and 7. The authors defined the radiological CHP phenomenon as a local CBF increase of more than 150% compared with the preoperative values, and then they investigated the correlation between\n RNF213<\/jats:italic>\n polymorphism and the development of CHP.\n <\/jats:p>\n <\/jats:sec>\n \n RESULTS<\/jats:title>\n \n CHP at POD 1 was observed in 23 hemispheres (23\/73 hemispheres [31.5%]), and its incidence was not statistically different between groups (15\/41 [36.6%] in\n RNF213<\/jats:italic>\n -mutant group vs 8\/32 [25.0%] in\n RNF213<\/jats:italic>\n \u2013wild type (WT) group; p = 0.321). CHP on POD 7, which is a relatively late period of the CHP phenomenon in MMD, was evident in 9 patients (9\/73 hemispheres [12.3%]) after STA-MCA anastomosis. This prolonged\/delayed CHP was exclusively observed in the\n RNF213<\/jats:italic>\n -mutant group (9\/41 [22.0%] in the\n RNF213<\/jats:italic>\n -mutant group vs 0\/32 [0.0%] in the\n RNF213<\/jats:italic>\n -WT group; p = 0.004). Multivariate analysis revealed that\n RNF213<\/jats:italic>\n polymorphism was significantly associated with CBF increase on POD 7 (OR 5.47, 95% CI 1.06\u201328.35; p = 0.043).\n <\/jats:p>\n <\/jats:sec>\n \n CONCLUSIONS<\/jats:title>\n \n Prolonged\/delayed CHP after revascularization surgery was exclusively found in the\n RNF213<\/jats:italic>\n -mutant group. Although the exact mechanism underlying the contribution of\n RNF213<\/jats:italic>\n polymorphism to the prolonged\/delayed CBF increase in patients with MMD is unclear, the current study suggests that genetic analysis of\n RNF213<\/jats:italic>\n is useful for predicting the perioperative pathology of patients with MMD.\n <\/jats:p>\n <\/jats:sec>","DOI":"10.3171\/2020.6.jns201037","type":"journal-article","created":{"date-parts":[[2020,10,23]],"date-time":"2020-10-23T03:50:52Z","timestamp":1603425052000},"page":"417-424","source":"Crossref","is-referenced-by-count":26,"title":["Prolonged\/delayed cerebral hyperperfusion in adult patients with moyamoya disease with RNF213 gene polymorphism c.14576G>A (rs112735431) after superficial temporal artery\u2013middle cerebral artery anastomosis"],"prefix":"10.3171","volume":"135","author":[{"given":"Ryosuke","family":"Tashiro","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, Kohnan Hospital, Sendai;"},{"name":"Department of Neurosurgery, Graduate School of Medicine, Tohoku University, Sendai; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miki","family":"Fujimura","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Kohnan Hospital, Sendai;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masahito","family":"Katsuki","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Kohnan Hospital, Sendai;"},{"name":"Department of Neurosurgery, Graduate School of Medicine, Tohoku University, Sendai; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Taketo","family":"Nishizawa","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Kohnan Hospital, Sendai;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasutake","family":"Tomata","sequence":"additional","affiliation":[{"name":"Division of Epidemiology, Department of Health Informatics and Public Health, Tohoku University School of Public Health, Graduate School of Medicine, Sendai, Miyagi, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kuniyasu","family":"Niizuma","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Graduate School of Medicine, Tohoku University, Sendai; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teiji","family":"Tominaga","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Graduate School of Medicine, Tohoku University, Sendai; and"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"5","key":"b1","series-title":"Neurol Med Chir (Tokyo)","first-page":"327","article-title":"Lessons learned from moyamoya disease: outcome of direct\/indirect revascularization surgery for 150 affected hemispheres","volume":"52","author":"Fujimura\n \nM","year":"2012","unstructured":"Fujimura\n \nM\n\n, \n\nTominaga\n \nT\n\n\n. \nLessons learned from moyamoya disease: outcome of direct\/indirect revascularization surgery for 150 affected hemispheres\n. \nNeurol Med Chir (Tokyo)\n. \n2012\n;\n52\n(\n5\n):\n327\n\u2013\n332\n."},{"issue":"10","key":"b2","series-title":"Stroke","first-page":"3025","article-title":"Long-term outcomes after combined revascularization surgery in adult moyamoya disease","volume":"45","author":"Cho\n \nWS","year":"2014","unstructured":"Cho\n \nWS\n\n, \n\nKim\n \nJE\n\n, \n\nKim\n \nCH\n\n, \n\n\n \nLong-term outcomes after combined revascularization surgery in adult moyamoya disease\n. \nStroke\n. \n2014\n;\n45\n(\n10\n):\n3025\n\u2013\n3031\n."},{"issue":"5","key":"b3","series-title":"Stroke","first-page":"1415","article-title":"Effects of extracranial-intracranial bypass for patients with hemorrhagic moyamoya disease: results of the Japan Adult Moyamoya Trial","volume":"45","author":"Miyamoto\n \nS","year":"2014","unstructured":"Miyamoto\n \nS\n\n, \n\nYoshimoto\n \nT\n\n, \n\nHashimoto\n \nN\n\n, \n\n\n \nEffects of extracranial-intracranial bypass for patients with hemorrhagic moyamoya disease: results of the Japan Adult Moyamoya Trial\n. \nStroke\n. \n2014\n;\n45\n(\n5\n):\n1415\n\u2013\n1421\n."},{"issue":"1","key":"b4","series-title":"Tohoku J Exp Med","first-page":"45","article-title":"Current status of revascularization surgery for Moyamoya disease: special consideration for its \u2018internal carotid-external carotid (IC-EC) conversion\u2019 as the physiological reorganization system","volume":"236","author":"Fujimura\n \nM","year":"2015","unstructured":"Fujimura\n \nM\n\n, \n\nTominaga\n \nT\n\n\n. \nCurrent status of revascularization surgery for Moyamoya disease: special consideration for its \u2018internal carotid-external carotid (IC-EC) conversion\u2019 as the physiological reorganization system\n. \nTohoku J Exp Med\n. \n2015\n;\n236\n(\n1\n):\n45\n\u2013\n53\n."},{"issue":"1","key":"b5","series-title":"Stroke","first-page":"37","article-title":"Significance of the hemorrhagic site for recurrent bleeding: prespecified analysis in the Japan Adult Moyamoya Trial","volume":"47","author":"Takahashi\n \nJC","year":"2016","unstructured":"Takahashi\n \nJC\n\n, \n\nFunaki\n \nT\n\n, \n\nHoukin\n \nK\n\n, \n\n\n \nSignificance of the hemorrhagic site for recurrent bleeding: prespecified analysis in the Japan Adult Moyamoya Trial\n. \nStroke\n. \n2016\n;\n47\n(\n1\n):\n37\n\u2013\n43\n."},{"issue":"3","key":"b6","series-title":"J Neurosurg","first-page":"793","article-title":"Meta-analysis of the surgical outcomes of symptomatic moyamoya disease in adults","volume":"128","author":"Jeon\n \nJP","year":"2018","unstructured":"Jeon\n \nJP\n\n, \n\nKim\n \nJE\n\n, \n\nCho\n \nWS\n\n, \n\n\n \nMeta-analysis of the surgical outcomes of symptomatic moyamoya disease in adults\n. \nJ Neurosurg\n. \n2018\n;\n128\n(\n3\n):\n793\n\u2013\n799\n."},{"issue":"5","key":"b7","series-title":"Neurol Med Chir (Tokyo)","first-page":"245","article-title":"Guidelines for diagnosis and treatment of moyamoya disease (spontaneous occlusion of the circle of Willis)","volume":"52","author":"Research Committee on the Pathology and Treatment of Spontaneous Occlusion of the Circle of Willis","year":"2012","unstructured":"Research Committee on the Pathology and Treatment of Spontaneous Occlusion of the Circle of Willis\n\n\n. \nGuidelines for diagnosis and treatment of moyamoya disease (spontaneous occlusion of the circle of Willis)\n. \nNeurol Med Chir (Tokyo)\n. \n2012\n;\n52\n(\n5\n):\n245\n\u2013\n266\n."},{"issue":"1","key":"b8","series-title":"Surg Cereb Stroke","first-page":"136","article-title":"Recommendations for the management of Moyamoya Disease: a statement from Research Committee on Spontaneous Occlusion of the Circle of Willis (Moyamoya Disease) [2nd Edition]. 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