Factors Associated with the Presentation of Moyamoya in Childhood

Amlie‐Lefond, Catherine; Ellenbogen, Richard G.

doi:10.1016/j.jstrokecerebrovasdis.2015.01.018

Cited by 26 publications

(20 citation statements)

References 39 publications

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“…All patients that were surgically treated received indirect revascularization, consistent with the literature. A history of headaches was present in six patients (36%), consistent with previous observations, which ranged between 22% and 52% in childhood (Amlie‐Lefond & Ellenbogen, ).…”

Section: Discussionsupporting

confidence: 90%

Moyamoya syndrome in children with neurofibromatosis type 1: Italian–French experience

Santoro

Rocco

Kossorotoff

et al. 2017

American J of Med Genetics Pt A

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Moyamoya syndrome (MMS) is the most common cerebral vasculopathy among children with neurofibromatosis type 1 (NF1). In this study, we clinically, radiologically, and genetically examined a cohort that was not previously described, comprising European children with NF1 and MMS. The NF1 genotyping had been registered. This study included 18 children. The mean age was 2.93 ± 3.03 years at the NF1 diagnosis and 7.43 ± 4.27 years at the MMS diagnosis. In seven patients, MMS was diagnosed before or at the same time as NF1. Neuroimaging was performed in 10 patients due to clinical symptoms, including headache (n = 6), cerebral infarction (n = 2), and complex partial seizures (n = 2). The remaining eight children (47%) had MMS diagnosed incidentally. Sixteen children were characterized molecularly. The features of MMS were similar between patients with and without NF1. Additionally, the NF1 phenotype and genotype were similar between children with and without MMS. Interestingly, three children experienced tumors with malignant histology or behavior. The presence of two first cousins in our cohort suggested that there may be potential genetic factors, not linked to NF1, with an additional role respect of NF1 might play a role in MMS pathogenesis. The incidental diagnosis of MMS, and the observation that, among children with NF1, those with MMS were clinically indistinguishable from those without MMS, suggested that it might be worthwhile to add an angiographic sequence to brain MRIs requested for children with NF1. A MMS diagnosis may assist in properly addressing an NF1 diagnosis in very young children who do not fulfill diagnostic criteria.

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Section: Discussionsupporting

confidence: 90%

Moyamoya syndrome in children with neurofibromatosis type 1: Italian–French experience

Santoro

Rocco

Kossorotoff

et al. 2017

American J of Med Genetics Pt A

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“…We found a relatively higher incidence of Moyamoya disease than was found in previous series. This might be explained by the fact that this disease is more prevalent among East Asian populations [33,34]. The present study also indicates that patients with underlying vascular pathologies leading to PIVH are younger than those with normal angiographic results.…”

Section: Discussionsupporting

confidence: 51%

Factors Associated with Clinical Outcomes in Patients with Primary Intraventricular Hemorrhage

Lee

Park

et al. 2017

Med Sci Monit

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BackgroundPrimary intraventricular hemorrhage (PIVH) is an uncommon type of intracerebral hemorrhage. Owing to its rarity, the clinical and radiological factors affecting outcomes in patients with PIVH have not been widely studied.Material/MethodsWe retrospectively reviewed 112 patients (mean age 53 years) treated for PIVH at our institution from January 2004 to December 2014. Clinical and radiological parameters were analyzed 3 months after initial presentation to identify factors associated with clinical outcomes, as assessed by the Glasgow Outcome Scale (favorable ≥4, unfavorable <4).ResultsOf the 99 patients who underwent angiography, causative vascular abnormalities were found in 46%, and included Moyamoya disease, arteriovenous malformation, and cerebral aneurysm. At 3 months after initial presentation, 64% and 36% of patients were in the favorable and unfavorable outcome groups, respectively. The mortality rate was 19%. However, most survivors had no or mild deficits. Age, initial Glasgow Coma Scale (GCS) score, simplified acute physiology score (SAPS II), modified Graeb score, and various radiological parameters reflecting ventricular dilatation were significantly different between the groups. Specifically, a GCS score of less than 13 (p=0.015), a SAPS II score of less than 33 (p=0.039), and a dilated fourth ventricle (p=0.043) were demonstrated to be independent predictors of an unfavorable clinical outcome.ConclusionsIn this study we reveal independent predictors of poor outcome in primary intraventricular hemorrhage patients, and show that nearly half of the patients in our study had predisposing vascular abnormalities. Routine angiography is recommended in the evaluation of PIVH to identify potentially treatable etiologies, which may enhance long-term prognosis.

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“…However, MMD should be included in the differential for a stroke in a child. In fact, MMD represents one of the most severe arteriopathies and accounts for approximately one fifth of identified cerebral arteriopathies in children presenting with acute stroke and is diagnosed in up to 20% of cases of childhood acute ischemic stroke (AIS) [ 8 ]. The condition is common in Japan and East Asia, where familial cases are also clearly recognized.…”

Section: Discussionmentioning

confidence: 99%

Moyamoya disease in a Moroccan baby: a case report

et al. 2018

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BackgroundA stroke in a baby is uncommon, recent studies suggested that their incidence is rising. Moyamoya disease is one of the leading causes of stroke in babies. This condition is mostly described in Japan. In Morocco, moyamoya disease has rarely been reported and a few cases were published. We report a rare Moroccan case of a 23-month-old baby boy who presented with left-sided hemiparesis and was diagnosed as having moyamoya disease.Case presentationA 23-month-old full-term Moroccan baby boy born to a non-consanguineous couple was referred to our hospital with the complaint of sudden onset left-sided hemiparesis. On neurological examination, there were no signs of meningeal irritation, his gait was hemiplegic, tone was decreased over left side, power was 2/5 over left upper and lower limb, and deep tendon reflexes were exaggerated. Preliminary neuroimaging suggested an arterial ischemic process. Clinical and laboratory evaluation excluded hematologic, metabolic, and vasculitic causes. Cerebral angiography confirmed the diagnosis of moyamoya disease. Our patient was treated with acetylsalicylic acid 5 mg/kg per day and referred to follow-up with pediatric neurosurgeon. Cerebral revascularization surgery using encephaloduroarteriosynangiosis was performed. At 8-month follow-up, his hemiparesis had improved and no further ischemic events had occurred.ConclusionThis case highlights the importance of considering moyamoya disease to be one of the classic etiologies of acute ischemic strokes in children from North Africa. It also emphasizes the rare presentation among the African population and the use of neurovascular imaging techniques to facilitate diagnosis of moyamoya disease.

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Factors Associated with the Presentation of Moyamoya in Childhood

Cited by 26 publications

References 39 publications

Moyamoya syndrome in children with neurofibromatosis type 1: Italian–French experience

Moyamoya syndrome in children with neurofibromatosis type 1: Italian–French experience

Factors Associated with Clinical Outcomes in Patients with Primary Intraventricular Hemorrhage

Moyamoya disease in a Moroccan baby: a case report

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