{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,5]],"date-time":"2026-05-05T01:36:16Z","timestamp":1777944976182,"version":"3.51.4"},"reference-count":80,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["FOC"],"published-print":{"date-parts":[[2014,1]]},"abstract":"<jats:sec>\n                    <jats:title>Object<\/jats:title>\n                    <jats:p>Pediatric patients with sickle cell anemia (SCA) carry a significant risk of developing moyamoya syndrome (MMS) and brain ischemia. The authors sought to review the safety and efficacy of pial synangiosis in the treatment of MMS in children with SCA by performing a comprehensive review of all previously reported cases in the literature.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The authors retrospectively reviewed the clinical and radiographic records in 17 pediatric patients with SCA treated at the Morgan Stanley Children's Hospital of New York (MSCHONY) who developed radiological evidence of MMS and underwent pial synangiosis between 1996 and 2012. The authors then added any additional reported cases of pial synangiosis for this population in the literature for a combined analysis of clinical and radiographic outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The combined data consisted of 48 pial synangiosis procedures performed in 30 patients. Of these, 27 patients (90%) presented with seizure, stroke, or transient ischemic attack, whereas 3 (10%) were referred after transcranial Doppler screening. At the time of surgery, the median age was 12 years. Thirteen patients (43%) suffered an ischemic stroke while on chronic transfusion therapy. Long-term follow-up imaging (MR angiography or catheter angiography) at a mean of 25 months postoperatively was available in 39 (81%) treated hemispheres. In 34 (87%) of those hemispheres there were demonstrable collateral vessels on imaging. There were 4 neurological events in 1590 cumulative months of follow-up, or 1 event per 33 patient-years. In the patients in whom complete data were available (MSCHONY series, n = 17), the postoperative stroke rate was reduced more than 6-fold from the preoperative rate (p = 0.0003).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Pial synangiosis in patients with SCA, MMS, and brain ischemia appears to be a safe and effective treatment option. Transcranial Doppler and\/or MRI screening in asymptomatic patients with SCA is recommended for the diagnosis of MMS.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/2013.10.focus13405","type":"journal-article","created":{"date-parts":[[2014,1,1]],"date-time":"2014-01-01T03:35:28Z","timestamp":1388547328000},"page":"E12","source":"Crossref","is-referenced-by-count":39,"title":["Pial synangiosis for moyamoya syndrome in children with sickle cell anemia: a comprehensive review of reported cases"],"prefix":"10.3171","volume":"36","author":[{"given":"Benjamin C.","family":"Kennedy","sequence":"first","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael M.","family":"McDowell","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peter H.","family":"Yang","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Caroline M.","family":"Wilson","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sida","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Todd C.","family":"Hankinson","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Children's Hospital Colorado, Aurora, Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Neil A.","family":"Feldstein","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Richard C. E.","family":"Anderson","sequence":"additional","affiliation":[{"name":"Department of Neurological Surgery, Columbia University Medical Center, New York, New York; and"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"key":"b1-00360012","series-title":"Ann Neurol","first-page":"699","article-title":"Long-term stroke risk in children with sickle cell disease screened with transcranial Doppler","volume":"42","author":"Adams","year":"1997"},{"key":"b2-00360012","series-title":"N Engl J Med","first-page":"5","article-title":"Prevention of a first stroke by transfusions in children with sickle cell anemia and abnormal results on transcranial Doppler ultrasonography","volume":"339","author":"Adams","year":"1998"},{"key":"b3-00360012","series-title":"Ann Neurol","first-page":"559","article-title":"Sickle cell disease and stroke: primary prevention and transcranial Doppler","volume":"54","author":"Adams","year":"2003"},{"key":"b4-00360012","series-title":"Pediatr Radiol","first-page":"242","article-title":"Long-term follow-up of pediatric sickle cell disease patients with abnormal high velocities on transcranial Doppler","volume":"35","author":"Bernaudin","year":"2005"},{"key":"b5-00360012","series-title":"Pediatr Radiol","first-page":"733","article-title":"Transcranial Doppler (TCD) screening for stroke prevention in sickle cell anemia: pitfalls in technique variation","volume":"30","author":"Bulas","year":"2000"},{"key":"b6-00360012","series-title":"Blood","first-page":"3144","article-title":"Moyamoya syndrome in childhood sickle cell disease: a predictive factor for recurrent cerebrovascular events","volume":"99","author":"Dobson","year":"2002"},{"key":"b7-00360012","series-title":"Neurosurgery","first-page":"34","article-title":"Clinical and angiographic outcomes from indirect revascularization surgery for Moyamoya disease in adults and children: a review of 63 procedures","volume":"68","author":"Dusick","year":"2011"},{"key":"b8-00360012","series-title":"J Neurosurg","first-page":"521","article-title":"Results of extracranialintracranial arterial bypass for bilateral carotid occlusion","volume":"63","author":"el-Fiki","year":"1985"},{"key":"b9-00360012","series-title":"Pediatr Neurol","first-page":"124","article-title":"Sickle cell anemia with moyamoya disease: outcomes after EDAS procedure","volume":"29","author":"Fryer","year":"2003"},{"key":"b10-00360012","series-title":"Surg Neurol","first-page":"273","article-title":"Temporary neurologic deterioration due to cerebral hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in patients with adult-onset moyamoya disease","volume":"67","author":"Fujimura","year":"2007"},{"key":"b11-00360012","series-title":"Surg Neurol","first-page":"442","article-title":"Incidence and risk factors for symptomatic cerebral hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in patients with moyamoya disease","volume":"71","author":"Fujimura","year":"2009"},{"key":"b12-00360012","series-title":"Neurosurgery","first-page":"957","article-title":"Significance of focal cerebral hyperperfusion as a cause of transient neurologic deterioration after extracranialintracranial bypass for moyamoya disease: comparative study with non-moyamoya patients using N-isopropyl-p-[(123)I] iodoamphetamine single-photon emission computed tomography","volume":"68","author":"Fujimura","year":"2011"},{"key":"b13-00360012","series-title":"Childs Nerv Syst","first-page":"134","article-title":"Surgical treatment of moyamoya disease in children: which is more effective procedure, EDAS or EMS?","volume":"2","author":"Fujita","year":"1986"},{"key":"b14-00360012","series-title":"Surg Neurol","first-page":"138","article-title":"Current state of study on moyamoya disease in Japan","volume":"47","author":"Fukui","year":"1997"},{"key":"b15-00360012","series-title":"Surg Neurol","first-page":"425","article-title":"Pseudomoyamoya in sickle cell anemia","volume":"18","author":"Garza-Mercado","year":"1982"},{"key":"b16-00360012","series-title":"J Neurosurg Pediatr","first-page":"211","article-title":"Surgical treatment of moyamoya syndrome in patients with sickle cell anemia: outcome following encephaloduroarteriosynangiosis","volume":"1","author":"Hankinson","year":"2008"},{"key":"b17-00360012","series-title":"Childs Nerv Syst","first-page":"734","article-title":"How does angiogenesis develop in pediatric moyamoya disease after surgery? A prospective study with MR angiography","volume":"20","author":"Houkin","year":"2004"},{"key":"b18-00360012","series-title":"Blood","first-page":"1197","article-title":"Allogeneic hematopoietic stem cell transplantation for sickle cell disease: the time is now","volume":"118","author":"Hsieh","year":"2011"},{"key":"b19-00360012","series-title":"Br J Haematol","first-page":"14","article-title":"Prospects for primary stroke prevention in children with sickle cell anaemia","volume":"157","author":"Jordan","year":"2012"},{"issue":"Suppl 2","key":"b20-00360012","series-title":"Clin Neurol Neurosurg","first-page":"S123","article-title":"Surgical techniques and the results of a fronto-temporo-parietal combined indirect bypass procedure for children with moyamoya disease: a comparison with the results of encephalo-duro-arterio-synangiosis alone","volume":"99","author":"Matsushima","year":"1997"},{"key":"b21-00360012","series-title":"Bull Tokyo Med Dent Univ","first-page":"7","article-title":"Angiographic demonstration of cerebral revascularization after encephalo-duro-arterio-synangiosis (EDAS) performed on pediatric moyamoya patients","volume":"29","author":"Matsushima","year":"1982"},{"key":"b22-00360012","series-title":"Pediatr Hematol Oncol","first-page":"368","article-title":"Moyamoya syndrome associated with severe iron deficiency anemia in a young child","volume":"29","author":"Meena","year":"2012"},{"key":"b23-00360012","series-title":"Stroke","first-page":"45","article-title":"Cerebrovascular disease in sickle cell anemia: a clinical, pathological and radiological correlation","volume":"9","author":"Merkel","year":"1978"},{"issue":"2","key":"b24-00360012","series-title":"Neurosurg Focus","first-page":"E15","article-title":"Long-term outcome of superficial temporal artery-middle cerebral artery bypass for patients with moyamoya disease in the US","volume":"24","author":"Mesiwala","year":"2008"},{"issue":"5","key":"b25-00360012","series-title":"Neurosurg Focus","first-page":"E5","article-title":"Long-term outcome after STA-MCA anastomosis for moyamoya disease","volume":"5","author":"Miyamoto","year":"1998"},{"key":"b26-00360012","series-title":"Pediatr Hematol Oncol","first-page":"371","article-title":"Hodgkin's disease in a child with sickle cell disease treated with hydroxyurea","volume":"18","author":"Moschovi","year":"2001"},{"key":"b27-00360012","series-title":"Stroke","first-page":"2644","article-title":"Management of stroke in infants and children: a scientific statement from a Special Writing Group of the American Heart Association Stroke Council and the Council on Cardiovascular Disease in the Young","volume":"39","author":"Roach","year":"2008"},{"key":"b28-00360012","series-title":"Pediatr Neurol","first-page":"199","article-title":"Encephaloduroarteriosynangiosis (EDAS) for the treatment of childhood moyamoya disease","volume":"10","author":"Ross","year":"1994"},{"issue":"2 Suppl Pediatrics","key":"b29-00360012","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","year":"2004"},{"key":"b30-00360012","series-title":"Curr Opin Pediatr","first-page":"657","article-title":"Pediatric stroke: current developments","volume":"19","author":"Seidman","year":"2007"},{"issue":"4","key":"b31-00360012","series-title":"Neurosurg Focus","first-page":"E10","article-title":"Pial synangiosis in patients with moyamoya syndrome and sickle cell anemia: perioperative management and surgical outcome","volume":"26","author":"Smith","year":"2009"},{"issue":"2","key":"b32-00360012","series-title":"Neurosurg Focus","first-page":"E17","article-title":"Progression of disease in unilateral moyamoya syndrome","volume":"24","author":"Smith","year":"2008"},{"key":"b33-00360012","series-title":"Skull Base","first-page":"15","article-title":"Surgical management of moyamoya syndrome","volume":"15","author":"Smith","year":"2005"},{"key":"b34-00360012","series-title":"Blood","first-page":"1078","article-title":"Fetal hemoglobin in sickle cell anemia: determinants of response to hydroxyurea","volume":"89","author":"Steinberg","year":"1997"},{"key":"b35-00360012","series-title":"Am J Hematol","first-page":"223","article-title":"Hematologic malignancy in sickle cell disease: report of four cases and review of the literature","volume":"21","author":"Stricker","year":"1986"},{"key":"b36-00360012","series-title":"Surg Neurol","first-page":"333","article-title":"Cerebral hemodynamics in patients with moyamoya disease. A study of the epicerebral microcirculation by fluorescein angiography","volume":"21","author":"Takeuchi","year":"1984"},{"key":"b37-00360012","series-title":"Indian Pediatr","first-page":"123","article-title":"Moya Moya cases treated with encephaloduroarteriosynangiosis","volume":"44","author":"Tripathi","year":"2007"},{"key":"b38-00360012","series-title":"Pediatr Neurol","first-page":"226","article-title":"Encephaloduroarterio-synangiosis in a child with sickle cell anemia and moyamoya disease","volume":"14","author":"Vernet","year":"1996"},{"key":"b39-00360012","series-title":"Expert Rev Hematol","first-page":"301","article-title":"Stroke in patients with sickle cell disease","volume":"6","author":"Webb","year":"2013"},{"key":"b40-00360012","series-title":"Neurosurgery","first-page":"787","article-title":"Results of extracranial-intracranial arterial bypass for intracranial internal carotid artery stenosis: review of 105 cases","volume":"15","author":"Weinstein","year":"1984"},{"key":"b1-00360012","series-title":"Ann Neurol","first-page":"699","article-title":"Long-term stroke risk in children with sickle cell disease screened with transcranial Doppler","volume":"42","author":"Adams","year":"1997"},{"key":"b2-00360012","series-title":"N Engl J Med","first-page":"5","article-title":"Prevention of a first stroke by transfusions in children with sickle cell anemia and abnormal results on transcranial Doppler ultrasonography","volume":"339","author":"Adams","year":"1998"},{"key":"b3-00360012","series-title":"Ann Neurol","first-page":"559","article-title":"Sickle cell disease and stroke: primary prevention and transcranial Doppler","volume":"54","author":"Adams","year":"2003"},{"key":"b4-00360012","series-title":"Pediatr Radiol","first-page":"242","article-title":"Long-term follow-up of pediatric sickle cell disease patients with abnormal high velocities on transcranial Doppler","volume":"35","author":"Bernaudin","year":"2005"},{"key":"b5-00360012","series-title":"Pediatr Radiol","first-page":"733","article-title":"Transcranial Doppler (TCD) screening for stroke prevention in sickle cell anemia: pitfalls in technique variation","volume":"30","author":"Bulas","year":"2000"},{"key":"b6-00360012","series-title":"Blood","first-page":"3144","article-title":"Moyamoya syndrome in childhood sickle cell disease: a predictive factor for recurrent cerebrovascular events","volume":"99","author":"Dobson","year":"2002"},{"key":"b7-00360012","series-title":"Neurosurgery","first-page":"34","article-title":"Clinical and angiographic outcomes from indirect revascularization surgery for Moyamoya disease in adults and children: a review of 63 procedures","volume":"68","author":"Dusick","year":"2011"},{"key":"b8-00360012","series-title":"J Neurosurg","first-page":"521","article-title":"Results of extracranialintracranial arterial bypass for bilateral carotid occlusion","volume":"63","author":"el-Fiki","year":"1985"},{"key":"b9-00360012","series-title":"Pediatr Neurol","first-page":"124","article-title":"Sickle cell anemia with moyamoya disease: outcomes after EDAS procedure","volume":"29","author":"Fryer","year":"2003"},{"key":"b10-00360012","series-title":"Surg Neurol","first-page":"273","article-title":"Temporary neurologic deterioration due to cerebral hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in patients with adult-onset moyamoya disease","volume":"67","author":"Fujimura","year":"2007"},{"key":"b11-00360012","series-title":"Surg Neurol","first-page":"442","article-title":"Incidence and risk factors for symptomatic cerebral hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in patients with moyamoya disease","volume":"71","author":"Fujimura","year":"2009"},{"key":"b12-00360012","series-title":"Neurosurgery","first-page":"957","article-title":"Significance of focal cerebral hyperperfusion as a cause of transient neurologic deterioration after extracranialintracranial bypass for moyamoya disease: comparative study with non-moyamoya patients using N-isopropyl-p-[(123)I] iodoamphetamine single-photon emission computed tomography","volume":"68","author":"Fujimura","year":"2011"},{"key":"b13-00360012","series-title":"Childs Nerv Syst","first-page":"134","article-title":"Surgical treatment of moyamoya disease in children: which is more effective procedure, EDAS or EMS?","volume":"2","author":"Fujita","year":"1986"},{"key":"b14-00360012","series-title":"Surg Neurol","first-page":"138","article-title":"Current state of study on moyamoya disease in Japan","volume":"47","author":"Fukui","year":"1997"},{"key":"b15-00360012","series-title":"Surg Neurol","first-page":"425","article-title":"Pseudomoyamoya in sickle cell anemia","volume":"18","author":"Garza-Mercado","year":"1982"},{"key":"b16-00360012","series-title":"J Neurosurg Pediatr","first-page":"211","article-title":"Surgical treatment of moyamoya syndrome in patients with sickle cell anemia: outcome following encephaloduroarteriosynangiosis","volume":"1","author":"Hankinson","year":"2008"},{"key":"b17-00360012","series-title":"Childs Nerv Syst","first-page":"734","article-title":"How does angiogenesis develop in pediatric moyamoya disease after surgery? A prospective study with MR angiography","volume":"20","author":"Houkin","year":"2004"},{"key":"b18-00360012","series-title":"Blood","first-page":"1197","article-title":"Allogeneic hematopoietic stem cell transplantation for sickle cell disease: the time is now","volume":"118","author":"Hsieh","year":"2011"},{"key":"b19-00360012","series-title":"Br J Haematol","first-page":"14","article-title":"Prospects for primary stroke prevention in children with sickle cell anaemia","volume":"157","author":"Jordan","year":"2012"},{"issue":"Suppl 2","key":"b20-00360012","series-title":"Clin Neurol Neurosurg","first-page":"S123","article-title":"Surgical techniques and the results of a fronto-temporo-parietal combined indirect bypass procedure for children with moyamoya disease: a comparison with the results of encephalo-duro-arterio-synangiosis alone","volume":"99","author":"Matsushima","year":"1997"},{"key":"b21-00360012","series-title":"Bull Tokyo Med Dent Univ","first-page":"7","article-title":"Angiographic demonstration of cerebral revascularization after encephalo-duro-arterio-synangiosis (EDAS) performed on pediatric moyamoya patients","volume":"29","author":"Matsushima","year":"1982"},{"key":"b22-00360012","series-title":"Pediatr Hematol Oncol","first-page":"368","article-title":"Moyamoya syndrome associated with severe iron deficiency anemia in a young child","volume":"29","author":"Meena","year":"2012"},{"key":"b23-00360012","series-title":"Stroke","first-page":"45","article-title":"Cerebrovascular disease in sickle cell anemia: a clinical, pathological and radiological correlation","volume":"9","author":"Merkel","year":"1978"},{"issue":"2","key":"b24-00360012","series-title":"Neurosurg Focus","first-page":"E15","article-title":"Long-term outcome of superficial temporal artery-middle cerebral artery bypass for patients with moyamoya disease in the US","volume":"24","author":"Mesiwala","year":"2008"},{"issue":"5","key":"b25-00360012","series-title":"Neurosurg Focus","first-page":"E5","article-title":"Long-term outcome after STA-MCA anastomosis for moyamoya disease","volume":"5","author":"Miyamoto","year":"1998"},{"key":"b26-00360012","series-title":"Pediatr Hematol Oncol","first-page":"371","article-title":"Hodgkin's disease in a child with sickle cell disease treated with hydroxyurea","volume":"18","author":"Moschovi","year":"2001"},{"key":"b27-00360012","series-title":"Stroke","first-page":"2644","article-title":"Management of stroke in infants and children: a scientific statement from a Special Writing Group of the American Heart Association Stroke Council and the Council on Cardiovascular Disease in the Young","volume":"39","author":"Roach","year":"2008"},{"key":"b28-00360012","series-title":"Pediatr Neurol","first-page":"199","article-title":"Encephaloduroarteriosynangiosis (EDAS) for the treatment of childhood moyamoya disease","volume":"10","author":"Ross","year":"1994"},{"issue":"2 Suppl Pediatrics","key":"b29-00360012","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","year":"2004"},{"key":"b30-00360012","series-title":"Curr Opin Pediatr","first-page":"657","article-title":"Pediatric stroke: current developments","volume":"19","author":"Seidman","year":"2007"},{"issue":"4","key":"b31-00360012","series-title":"Neurosurg Focus","first-page":"E10","article-title":"Pial synangiosis in patients with moyamoya syndrome and sickle cell anemia: perioperative management and surgical outcome","volume":"26","author":"Smith","year":"2009"},{"issue":"2","key":"b32-00360012","series-title":"Neurosurg Focus","first-page":"E17","article-title":"Progression of disease in unilateral moyamoya syndrome","volume":"24","author":"Smith","year":"2008"},{"key":"b33-00360012","series-title":"Skull Base","first-page":"15","article-title":"Surgical management of moyamoya syndrome","volume":"15","author":"Smith","year":"2005"},{"key":"b34-00360012","series-title":"Blood","first-page":"1078","article-title":"Fetal hemoglobin in sickle cell anemia: determinants of response to hydroxyurea","volume":"89","author":"Steinberg","year":"1997"},{"key":"b35-00360012","series-title":"Am J Hematol","first-page":"223","article-title":"Hematologic malignancy in sickle cell disease: report of four cases and review of the literature","volume":"21","author":"Stricker","year":"1986"},{"key":"b36-00360012","series-title":"Surg Neurol","first-page":"333","article-title":"Cerebral hemodynamics in patients with moyamoya disease. 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