Poster #18 - ADHD Symptoms of Children with Neurofibromatosis Type 1: A Systematic Review and Meta-analysis

• In Event: Poster Session 03

Abstract

Neurofibromatosis type 1 (NF1) is a common genetic neurodevelopmental disorder affecting approximately 1:3500 individuals (McClatchey, 2007). About 50% of children with NF1 meet the diagnosis criteria of Attention Deficit Hyperactivity Disorder (ADHD) (Hyman et al., 2005). However, ADHD symptoms are largely heterogeneous in the ADHD population and the NF1 population (Mahone & Denckla, 2017; Payne et al., 2021). Neuropsychological theories of ADHD highlight the importance of understanding ADHD as multidimensional constructs to better understand the heterogeneity of ADHD, moving beyond categorical diagnoses (Insel et al., 2010). Although many studies of children with NF1 reported the percentage of children diagnosed with ADHD, there is a lack of systematic evaluation on the extent and predictors of ADHD symptoms in children with NF1, taking a multidimensional perspective of ADHD. The current study aims to provide a systematic review of studies on ADHD symptoms in children with NF1. Meta-analyses will be conducted to determine group differences in ADHD symptoms between the NF1 and the control or normative group and moderators of the group differences. Additionally, a qualitative review will be conducted to summarize prior findings on predictors of ADHD symptoms (e.g., executive function) in children with NF1.

This study will use data from a larger meta-analysis project on the neurobehavioral functions of individuals with NF1. Systematic literature searches were conducted in Scopus, PsycINFO, Web of Science, ProQuest, PubMed, and Medline. Search terms included a combination of NF1 terms (e.g., neurofibromatosis type 1, NF1) and neurobehavioral functioning terms (e.g., attention, hyperact*, impuls*, ADHD). See the appendix for complete search terms and inclusion criteria. All papers will be coded by at least two authors with an interrater agreement above .80. We estimate that there are 72 studies meeting the inclusion criteria for the current study based on coding already completed.

Hedges’ g will be calculated for group differences in ADHD symptoms between the NF1 group and the control or normative group for each study. A robust standard error estimation technique will be used to handle dependent effect sizes (Hedges et al., 2010). Multiple methods will be used to evaluate the publication bias effect. Meta-regression with mixed-effects models (Card, 2011) will be used to examine nine potential moderators of group differences in ADHD symptoms.

Based on prior literature, we expect that children with NF1 have more ADHD symptoms than control/normative groups. This group difference would be larger in inattention symptoms than hypoactivity/impulsivity symptoms, in performance-based and parent-reported (vs. self, teacher, or observer-reported) measures, and in NF1 samples with older (vs. younger) participants, a higher portion of boys (vs. girls), lower IQ, lower parental education, a higher percentage of familial NF1, more severe NF1 symptoms, or higher portion of participants diagnosed with ADHD.

This systematic review and meta-analytic study will provide important information on the variabilities of ADHD symptoms in children with NF1 and factors accounting for the variabilities. Such information is critical for patient management and intervention development. We will discuss gaps in the existing literature and make recommendations for future research.

Authors

• Yang Hou, Florida State University
  Presenting Author

• Sara Yamini

• Dan Liu, Florida State University

• Xian Wu

• Hossein Dabiriyan Tehrani