RSS-Feed abonnieren
DOI: 10.1055/a-1993-3860
Characterization of Neuropsychological Outcomes in a Cohort of Pediatric Patients with Moyamoya Arteriopathy
Funding This work is supported by the Laney Jaymes Foundation for Pediatric Stroke, the American Heart Association (Career Development Award [850044]), the D.C. Women's Board, and the Johns Hopkins School of Medicine Office of Medical Student Affairs.![](https://www.thieme-connect.de/media/neuropediatrics/202302/lookinside/thumbnails/10-1055-a-1993-3860_0920223311sc-1.jpg)
Abstract
Introduction Moyamoya arteriopathy is a severe, progressive cerebral arteriopathy that places affected children at high risk for stroke. Moyamoya has been associated with a range of neuropsychological deficits in adults, but data on many cognitive domains remain limited in the pediatric population and little is known about the neuropsychological profile of children with syndromic moyamoya.
Methods This is a single-center, retrospective cohort study of children with moyamoya arteriopathy followed at our center who underwent neuropsychological testing between 2003 and 2021. Test scores were extracted from neuropsychological reports. Medical records were reviewed with attention to individual neuropsychological test results, medical comorbidities, presence of infarct(s) on neuroimaging, and history of clinical ischemic stroke.
Results Of the 83 children with moyamoya followed at our center between 2003 and 2021, 13 had completed neuropsychological testing across multiple cognitive domains. Compared to age-based normative data, children in this sample had lower scores in overall intelligence (p = 0.003), global executive functioning (p = 0.005), and overall adaptive functioning (p = 0.015). There was no significant difference in overall intelligence between children with (n = 6) versus without (n = 7) a history of clinical stroke (p = 0.368), though children with any radiographic infarct scored lower in this domain (p = 0.032).
Conclusion In our cohort, children with moyamoya demonstrated impaired intelligence and executive functioning, even in the absence of clinical stroke. Neuropsychological evaluation should be considered standard of care for all children with moyamoya, even those without a history of clinical stroke.
Publikationsverlauf
Eingereicht: 03. Oktober 2022
Angenommen: 01. Dezember 2022
Accepted Manuscript online:
06. Dezember 2022
Artikel online veröffentlicht:
19. Januar 2023
© 2023. Thieme. All rights reserved.
Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany
-
References
- 1 Suzuki J, Kodama N. Moyamoya disease—a review. Stroke 1983; 14 (01) 104-109
- 2 Ferriero DM, Fullerton HJ, Bernard TJ. et al. Management of stroke in neonates and children: a scientific statement from the American Heart Association/American Stroke Association. Stroke 2019; 50 (03) e51-e96
- 3 Fullerton HJ, Wintermark M, Hills NK. et al; VIPS Investigators. Risk of recurrent arterial ischemic stroke in childhood: a prospective international study. Stroke 2016; 47 (01) 53-59
- 4 Kaseka ML, Slim M, Muthusami P. et al. Distinct clinical and radiographic phenotypes in pediatric patients with moyamoya. Pediatr Neurol 2021; 120: 18-26
- 5 Gatti JR, Torriente AG, Sun LR. Clinical presentation and stroke incidence differ by moyamoya etiology. J Child Neurol 2021; 36 (04) 272-280
- 6 Ohtaki M, Uede T, Morimoto S, Nonaka T, Tanabe S, Hashi K. Intellectual functions and regional cerebral haemodynamics after extensive omental transplantation spread over both frontal lobes in childhood moyamoya disease. Acta Neurochir (Wien) 1998; 140 (10) 1043-1053 , discussion 1052–1053
- 7 Kronenburg A, van den Berg E, van Schooneveld MM. et al. Cognitive functions in children and adults with moyamoya vasculopathy: a systematic review and meta-analysis. J Stroke 2018; 20 (03) 332-341
- 8 Sato H, Sato N, Tamaki N, Matsumoto S. Chronic low-perfusion state in children with moyamoya disease following revascularization. Childs Nerv Syst 1990; 6 (03) 166-171
- 9 Choi EJ, Westmacott R, Kirkham FJ. et al. Fronto-parietal and white matter haemodynamics predict cognitive outcome in children with moyamoya independent of stroke. Transl Stroke Res 2022; 13 (05) 757-773
- 10 Gatti JR, Sun LR. Nonischemic presentations of pediatric moyamoya arteriopathy: a natural history study. Stroke 2022; 53 (06) e219-e220
- 11 Wechsler D. Wechsler Intelligence Scale for Children. 4th ed. San Antonio, TX: The Psychological Corporation; 2003
- 12 Wechsler D. WISC-V: Wechsler Intelligence Scale for Children. 5th ed. San Antonio, TX: Pearson Education Inc.; Psychological Corporation; 2014
- 13 Elliott CD. Differential Ability Scales. 2nd ed. San Antonio, TX: Harcourt Assessment; 2007
- 14 Gioia GA, Isquith PK, Guy SC, Kenworthy L. Behavior Rating Inventory of Executive Function®, Second Edition (BRIEF®2). Lutz, FL: Psychological Assessment Resources Inc.; 2015
- 15 Isquith PK, Gioia GA. Behavior Rating Inventory of Executive Function ® - Preschool Version. Lutz, FL: Psychological Assessment Resources Inc.; 2003
- 16 Harrison PL, Oakland T. Adaptive Behavior Assessment System. 3rd ed. Torrance, CA: Western Psychological Services; 2015
- 17 Reynolds CR, Kamphaus RW. BASC-2 Behavior Assessment System for Children. 2nd ed. Minnesota City, MN: American Guidance Service Publishing; 2004
- 18 Reynolds CR, Kamphaus RW. Behavior Assessment System for Children 3rd ed. Bloomington, MN: NCS Pearson Inc.; 2015
- 19 Conners CK. Conners Comprehensive Behavior Rating Scale. 3rd ed. Toronto, Ontario, Canada: Multi-Health Systems; 2008
- 20 Beery KE, Buktenica NA, Beery NA. The Beery-Buktenica Developmental Test of Visual-Motor Integration: Administration, Scoring, and Teaching Manual. 6th ed. Minneapolis, MN: NCS Pearson Inc.; 2010
- 21 Williams TS, Westmacott R, Dlamini N. et al. Intellectual ability and executive function in pediatric moyamoya vasculopathy. Dev Med Child Neurol 2012; 54 (01) 30-37