Poster #39 - Internalizing and Externalizing Symptoms In Children with Neurofibromatosis Type 1: A Systematic Review and Meta-analysis

• In Event: Poster Session 14

Abstract

Neurofibromatosis type 1 (NF1) is a genetic neurodevelopmental disorder diagnosed in approximately 1:3500 individuals (McClatchey, 2007). Children with NF1 are susceptible to various sociobehavioral problems including two major categories - internalizing and externalizing problems (Rietman et al., 2018; Vogel et al., 2017). Considerable variability also exists in the degree of impairment and experiences of the specific problems among the NF1 population (Johnson et al., 1999; van der Vaart et al., 2016), but a symtematic evaluation of this is still lacking. Informed by the hierarchical–dimensional model for psychopathology (Achenbach et al., 2016), the current study aims to provide a systematic review and meta-analysis of studies on internalizing and externalizing symptoms in children with NF1, focusing on both the two general categories as well as the specific symptoms in each category (e.g., anxiety and depression, aggression and delinquency). Meta-analyses will be conducted to determine the extent to which NF1 patients experience internalizing and externalizing symptoms compared to the control or normative group and moderators of group differences. A qualitative review will also be conducted to summarize prior findings on predictors of internalizing and externalizing symptoms in children with NF1.

Data will come from a larger meta-analysis project on neurobehavioral functions of individuals with NF1. Literature searches were conducted in Scopus, PsycINFO, Web of Science, ProQuest, PubMed, and Medline (see appendix for the search terms and the inclusion criteria). Selected papers will be coded by at least two researchers. Based on coding already completed, we estimate there to be 117 studies meeting the inclusion criteria.

Hedges’ g will be calculated to compare internalizing and externalizing symptoms between the NF1 group and the control/normative group, along with a robust standard error estimation technique for handling dependent effect sizes (Hedges et al., 2010). Publication bias will be evaluated using funnel plots with Egger’s tests (Sterne & Egger, 2001; Sterne et al., 2008), the trim-and-fill technique (Duval & Tweedie, 2000), and meta-regression analyses. Meta-regression with mixed-effects models (Card, 2011) will be used to examine nine potential moderators of group differences in outcomes.

We expect that children with NF1 will score higher on internalizing and externalizing as well as the subdomains than control/normative groups. Additionally, we expect the group differences to be greater for parent (vs. self, teacher) reported measures and in samples with older (vs. younger) participants, lower parental education, more cases of familial NF1, more severe NF1 symptoms, lower IQ, or more participants diagnosed of a psychiatric disorder. We also expect greater group differences in samples with more males for externalizing dimensions but more females for internalizing dimensions.

Study findings will illuminate whether children with NF1 consistently experience more internalizing and externalizing symptoms compared to control/normative groups, and factors related to the symptoms and the group differences. Such information is critical for intervention development as well as practices for healthier sociobehavioral development in children with NF1. Limitations and gaps in extant literature and directions for future research will also be discussed.

Authors

• Dan Liu, Florida State University
  Presenting Author

• Sara Yamini

• Xian Wu

• Hossein Dabiriyan Tehrani

• Yang Hou, Florida State University