{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,7]],"date-time":"2026-11-07T02:25:46Z","timestamp":1794018346672,"version":"4.8.0"},"reference-count":14,"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 There are limited reports on long-term morbidity in pediatric patients who have undergone surgical revascularization for moyamoya disease (MMD). Here, the authors report long-term morbidity and mortality in a population of pediatric patients who underwent pial synangiosis for MMD from 1988 through 2016.<\/jats:p>\n <\/jats:sec>\n \n METHODS<\/jats:title>\n A single-center retrospective review of the hospital and personal operative databases of the senior authors was carried out to identify all patients who were treated for MMD at Boston Children\u2019s Hospital between 1988 and 2016, and who experienced any episode of late morbidity or mortality, which the authors defined as an event resulting in significant neurological deficit or death occurring more than 1 year after revascularization surgery. Hospital records were reviewed to determine pertinent demographic data, the initial mode of patient presentation, and associated comorbidities. Radiographic studies, when available, were reviewed for documentation of the diagnosis and for confirmation of the late complication, and the literature on this topic was reviewed.<\/jats:p>\n <\/jats:sec>\n \n RESULTS<\/jats:title>\n In total, 460 patients with MMD underwent surgery between 1988 and 2016 using the pial synangiosis surgical technique; 15 (3.3%) of these patients (9 females and 6 males) experienced documented late death (n = 14) or severe morbidity (n = 1). The median age at revascularization surgery was 8.0 years (range 1\u201321 years). The causes of these late complications were grouped into three etiologies: intraventricular or intracerebral hemorrhage (n = 8), systemic complications related to associated comorbidities or preoperative disabilities (n = 5), and the development of malignant brain tumors (n = 2). Four patients whose MMD was associated with a history of cranial radiation therapy died. These events occurred from as early as 2 years to as late as 27 years postoperatively.<\/jats:p>\n <\/jats:sec>\n \n CONCLUSIONS<\/jats:title>\n The risk of late morbidities and mortality following pial synangiosis for MMD in the pediatric patient appeared to be low. Nevertheless, the occurrence of catastrophic cerebrovascular events, particularly intracerebral and intraventricular hemorrhage in the otherwise neurologically stable revascularized patient, was concerning. Although there is value in long-term surveillance of patients who have undergone surgery for MMD, from both a neurological and a general medical standpoint, particularly in patients with the risk factor of prior cranial radiation therapy, it is not clear from the data how the late deaths in this population could have been prevented.<\/jats:p>\n <\/jats:sec>","DOI":"10.3171\/2021.1.peds20944","type":"journal-article","created":{"date-parts":[[2021,7,2]],"date-time":"2021-07-02T03:10:17Z","timestamp":1625195417000},"page":"206-211","source":"Crossref","is-referenced-by-count":3,"title":["Late morbidity and mortality following revascularization surgery for moyamoya disease in the pediatric population"],"prefix":"10.3171","volume":"28","author":[{"given":"Madeline B.","family":"Karsten","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward R.","family":"Smith","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R. Michael","family":"Scott","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"12","key":"r1","series-title":"N Engl J Med","doi-asserted-by":"publisher","first-page":"1226","DOI":"10.1056\/NEJMra0804622","article-title":"Moyamoya disease and moyamoya syndrome","volume":"360","author":"Scott RM","year":"2009","unstructured":"Scott RM, Smith ER. Moyamoya disease and moyamoya syndrome. N Engl J Med. 2009;360(12):1226\u20131237.1929757510.1056\/NEJMra0804622"},{"issue":"2 Suppl Pediatrics","key":"r2","series-title":"J Neurosurg","first-page":"142","article-title":"Long-term outcome in children with moyamoya syndrome after cranial revascularization by pial synangiosis","volume":"100","author":"Scott RM","year":"2004","unstructured":"Scott RM, Smith JL, Robertson RL, Long-term outcome in children with moyamoya syndrome after cranial revascularization by pial synangiosis. J Neurosurg. 2004;100(2 Suppl Pediatrics):142\u2013149.14758941"},{"issue":"5","key":"r3","series-title":"J Neurosurg Pediatr","doi-asserted-by":"publisher","first-page":"451","DOI":"10.3171\/2012.8.PEDS11539","article-title":"Long-term follow-up of surgically treated juvenile patients with moyamoya disease","volume":"10","author":"Mukawa M","year":"2012","unstructured":"Mukawa M, Nariai T, Matsushima Y, Long-term follow-up of surgically treated juvenile patients with moyamoya disease. J Neurosurg Pediatr. 2012;10(5):451\u2013456.2295775610.3171\/2012.8.PEDS11539"},{"issue":"3","key":"r4","series-title":"Acta Neurochir (Wien)","doi-asserted-by":"publisher","first-page":"551","DOI":"10.1007\/s00701-013-1975-7","article-title":"Incidence of late cerebrovascular events after direct bypass among children with moyamoya disease: a descriptive longitudinal study at a single center","volume":"156","author":"Funaki T","year":"2014","unstructured":"Funaki T, Takahashi JC, Takagi Y, Incidence of late cerebrovascular events after direct bypass among children with moyamoya disease: a descriptive longitudinal study at a single center. Acta Neurochir (Wien). 2014;156(3):551\u2013559.10.1007\/s00701-013-1975-7"},{"issue":"2","key":"r5","series-title":"Cerebrovasc Dis","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1159\/000369524","article-title":"Clinical features, surgical treatment, and long-term outcome in pediatric patients with moyamoya disease in China","volume":"39","author":"Bao XY","year":"2015","unstructured":"Bao XY, Duan L, Yang WZ, Clinical features, surgical treatment, and long-term outcome in pediatric patients with moyamoya disease in China. Cerebrovasc Dis. 2015;39(2):75\u201381.2557376410.1159\/000369524"},{"issue":"6","key":"r6","series-title":"Neurosurgery","doi-asserted-by":"publisher","first-page":"1093","DOI":"10.1227\/01.NEU.0000369606.00861.91","article-title":"Novel bypass surgery for moyamoya disease using pericranial flap: its impacts on cerebral hemodynamics and long-term outcome","volume":"66","author":"Kuroda S","year":"2010","unstructured":"Kuroda S, Houkin K, Ishikawa T, Novel bypass surgery for moyamoya disease using pericranial flap: its impacts on cerebral hemodynamics and long-term outcome. Neurosurgery. 2010;66(6):1093\u20131101.10.1227\/01.NEU.0000369606.00861.9120495424"},{"issue":"4","key":"r7","series-title":"Neurosurg Rev","doi-asserted-by":"publisher","first-page":"615","DOI":"10.1007\/s10143-016-0734-7","article-title":"Long-term follow-up of pediatric moyamoya disease treated by combined direct-indirect revascularization surgery: single institute experience with surgical and perioperative management","volume":"39","author":"Rashad S","year":"2016","unstructured":"Rashad S, Fujimura M, Niizuma K, Long-term follow-up of pediatric moyamoya disease treated by combined direct-indirect revascularization surgery: single institute experience with surgical and perioperative management. Neurosurg Rev. 2016;39(4):615\u2013623.2718055910.1007\/s10143-016-0734-7"},{"issue":"1","key":"r8","series-title":"Pediatr Neurosurg","doi-asserted-by":"publisher","first-page":"14","DOI":"10.1159\/000048343","article-title":"Long-term outcomes of pediatric moyamoya disease treated by encephalo-duro-arterio-synangiosis","volume":"36","author":"Isono M","year":"2002","unstructured":"Isono M, Ishii K, Kamida T, Long-term outcomes of pediatric moyamoya disease treated by encephalo-duro-arterio-synangiosis. Pediatr Neurosurg. 2002;36(1):14\u201321.1181874110.1159\/000048343"},{"issue":"2 Suppl Pediatrics","key":"r9","series-title":"J Neurosurg","first-page":"156","article-title":"Long-term effects of indirect bypass surgery on collateral vessel formation in pediatric moyamoya disease","volume":"100","author":"Goda M","year":"2004","unstructured":"Goda M, Isono M, Ishii K, Long-term effects of indirect bypass surgery on collateral vessel formation in pediatric moyamoya disease. J Neurosurg. 2004;100(2 Suppl Pediatrics):156\u2013162.14758943"},{"issue":"8","key":"r10","series-title":"J Clin Neurosci","doi-asserted-by":"publisher","first-page":"873","DOI":"10.1016\/j.jocn.2004.11.008","article-title":"Long term outcome in children with moyamoya disease: experience with 16 patients","volume":"12","author":"Darwish B","year":"2005","unstructured":"Darwish B, Besser M. Long term outcome in children with moyamoya disease: experience with 16 patients. J Clin Neurosci. 2005;12(8):873\u2013877.10.1016\/j.jocn.2004.11.00816257213"},{"issue":"11","key":"r11","series-title":"Stroke","doi-asserted-by":"publisher","first-page":"3177","DOI":"10.1161\/STROKEAHA.119.025609","article-title":"Long-term outcomes of indirect bypass for 629 children with moyamoya disease: longitudinal and cross-sectional analysis","volume":"50","author":"Ha EJ","year":"2019","unstructured":"Ha EJ, Kim KH, Wang KC, Long-term outcomes of indirect bypass for 629 children with moyamoya disease: longitudinal and cross-sectional analysis. Stroke. 2019;50(11):3177\u20133183.3155103710.1161\/STROKEAHA.119.025609"},{"issue":"6","key":"r12","series-title":"Neurol Med Chir (Tokyo)","doi-asserted-by":"publisher","first-page":"240","DOI":"10.2176\/nmc.ra.2018-0026","article-title":"Late cerebrovascular events and social outcome after adolescence: long-term outcome of pediatric moyamoya disease","volume":"58","author":"Funaki T","year":"2018","unstructured":"Funaki T, Takahashi JC, Miyamoto S. Late cerebrovascular events and social outcome after adolescence: long-term outcome of pediatric moyamoya disease. Neurol Med Chir (Tokyo). 2018;58(6):240\u2013246.10.2176\/nmc.ra.2018-0026"},{"issue":"8","key":"r13","series-title":"J Stroke Cerebrovasc Dis","doi-asserted-by":"publisher","first-page":"104786","DOI":"10.1016\/j.jstrokecerebrovasdis.2020.104786","article-title":"De novo renal artery stenosis developed in initially normal renal arteries during the long-term follow-up of patients with moyamoya disease","volume":"29","author":"Hara S","year":"2020","unstructured":"Hara S, Shimizu K, Nariai T, De novo renal artery stenosis developed in initially normal renal arteries during the long-term follow-up of patients with moyamoya disease. J Stroke Cerebrovasc Dis. 2020;29(8):104786.10.1016\/j.jstrokecerebrovasdis.2020.10478632229075"},{"issue":"10","key":"r14","series-title":"Acta Neurochir (Wien)","doi-asserted-by":"publisher","first-page":"2589","DOI":"10.1007\/s00701-020-04501-8","article-title":"Central retinal artery occlusion occurring 30 years after successful revascularization surgery for moyamoya disease: case report","volume":"162","author":"Karsten MB","year":"2020","unstructured":"Karsten MB, Oliveira C, Segal AZ, Scott RM. Central retinal artery occlusion occurring 30 years after successful revascularization surgery for moyamoya disease: case report. Acta Neurochir (Wien). 2020;162(10):2589\u20132592.10.1007\/s00701-020-04501-8"}],"container-title":["Journal of Neurosurgery: Pediatrics"],"original-title":[],"link":[{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-pediatr\/28\/2\/article-p206.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-pediatr\/28\/2\/article-p206.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-pediatr\/28\/2\/article-p206.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,11,7]],"date-time":"2026-11-07T01:56:56Z","timestamp":1794016616000},"score":1,"resource":{"primary":{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-pediatr\/28\/2\/article-p206.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,8]]},"references-count":14,"journal-issue":{"issue":"2"},"URL":"https:\/\/doi.org\/10.3171\/2021.1.peds20944","relation":{},"ISSN":["1933-0707","1933-0715"],"issn-type":[{"value":"1933-0707","type":"print"},{"value":"1933-0715","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,8]]}}}