Files and links (1)
Accepted (AM)Open Access (License Unspecified), Open
Journal article
A systematic review and meta‐analysis of intellectual, neuropsychological, and psychoeducational functioning in neurofibromatosis type 1
American journal of medical genetics. Part A, v 188(8), pp 2277-2292
Aug 2022
PMID: 35546306
Featured in Collection : UN Sustainable Development Goals @ Drexel
Abstract
Neurofibromatosis Type 1 (NF1) is a common genetic disorder frequently associated with cognitive deficits. Despite cognitive deficits being a key feature of NF1, the profile of such impairments in NF1 has been shown to be heterogeneous. Thus, we sought to quantitatively synthesize the extant literature on cognitive functioning in NF1. A random‐effects meta‐analysis of cross‐sectional studies was carried out comparing cognitive functioning of patients with NF1 to typically developing or unaffected sibling comparison subjects of all ages. Analyses included 50 articles (Total NNF1 = 1,522; MAge = 15.70 years, range = 0.52–69.60), yielding 460 effect sizes. Overall moderate deficits were observed [g = −0.64, 95% CI = (−0.69, −0.60)] wherein impairments differed at the level of cognitive domain. Deficits ranged from large [general intelligence: g = −0.95, 95% CI = (−1.12, −0.79)] to small [emotion: g = −0.37, 95% CI = (−0.63, −0.11)]. Moderation analyses revealed nonsignificant contributions of age, sex, educational attainment, and parental level of education to outcomes. These results illustrate that cognitive impairments are diffuse and salient across the lifespan in NF1. Taken together, these results further demonstrate efforts should be made to evaluate and address cognitive morbidity in patients with NF1 in conjunction with existing best practices.
Metrics
Details
- Title
- A systematic review and meta‐analysis of intellectual, neuropsychological, and psychoeducational functioning in neurofibromatosis type 1
- Creators
- Andrew J. D. Crow - University of PennsylvaniaJennica M. Janssen - Drexel UniversityCarolina Marshall - University of PennsylvaniaAnne Moffit - University of PennsylvaniaLaura Brennan - Genomind, San Diego, California, USA.Christian G. Kohler - University of PennsylvaniaDavid R. Roalf - University of PennsylvaniaPaul J. Moberg - University of Pennsylvania
- Publication Details
- American journal of medical genetics. Part A, v 188(8), pp 2277-2292
- Publisher
- John Wiley & Sons, Inc
- Number of pages
- 16
- Grant note
- Children's Hospital of Philadelphia National Institute of Mental Health (MH119185)
- Resource Type
- Journal article
- Language
- English
- Academic Unit
- Psychological and Brain Sciences (Psychology)
- Web of Science ID
- WOS:000793707500001
- Scopus ID
- 2-s2.0-85129908262
- Other Identifier
- 991019169649104721
UN Sustainable Development Goals (SDGs)
This publication has contributed to the advancement of the following goals:
InCites Highlights
Data related to this publication, from InCites Benchmarking & Analytics tool:
- Collaboration types
- Domestic collaboration
- Web of Science research areas
- Genetics & Heredity