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. Author manuscript; available in PMC: 2025 Oct 17.
Published in final edited form as: J Fam Psychol. 2024 Oct 17;39(1):99–106. doi: 10.1037/fam0001256

Parent-Child Discrepancies in Reports of Child Psychosocial Functioning in Neurofibromatosis Type 1

Nour Al Ghriwati 1, Paige Little 2, Staci Martin 2, Mary Anne Tamula 2, Brigitte C Widemann 2, Pamela L Wolters 2
PMCID: PMC12371602  NIHMSID: NIHMS2056776  PMID: 39418437

Abstract

Children with neurofibromatosis type 1 (NF1) are at an increased risk for social-emotional difficulties. These difficulties, including depression and anxiety, are typically measured through parental report of child functioning in research; rarely have children with NF1 rated their own well-being. Discrepancies between parent proxy and child self-report of psychosocial functioning in other populations have been shown to relate to socio-emotional problems and distress. This study examined the concordance of parent proxy and child self-report of child behavioral and social-emotional functioning on selected Behavior Assessment System for Children–Second Edition subscales in families of children with NF1 and plexiform neurofibroma tumors (pNFs). We also sought to explore possible child, family, and community factors relating to discrepancies in reporting for youth with NF1 and pNFs. Overall, parents reported higher symptoms across psychosocial domains (anxiety, depression, and atypicality) in comparison to their children. Furthermore, characteristics like child sex, attention-deficit hyperactivity disorder diagnosis, and family functioning significantly predicted differences in ratings of child functioning. These findings indicate that multi-informant studies are crucial to understanding multiple perspectives among family members in symptom-reporting and risk factors for these discrepancies.

Keywords: neurofibromatosis, psychosocial functioning, family functioning, reporting discrepancies, pediatric psychology

Neurofibromatosis type 1 (NF1) is a genetic condition caused by heterozygous mutations of the NF1 gene. It affects about one in 2,500 to 4,000 individuals (Evans et al., 2010; Tamura, 2021) and characteristic clinical manifestations include café-au-lait macules, optic gliomas, Lisch nodules, bony abnormalities, and neurofibroma tumors (Jett & Friedman, 2010; Tonsgard, 2006). Furthermore, approximately 50% of those with NF1 have plexiform neurofibromas (pNFs), which are histologically benign tumors that grow along nerve fascicles and can lead to pain, disfigurement, and morbidity (Jett & Friedman, 2010; Prada et al., 2012). Additionally, attention, executive functioning, and learning problems are common in youth with NF1 (Hou et al., 2020; Payne et al., 2021). These physical complications and neurocognitive difficulties put children with NF1 at risk for a variety of social-emotional problems (Hou et al., 2022; Martin et al., 2012).

Numerous studies have documented social-emotional impairments in children with NF1 compared to normative samples. For example, youth with NF1 have higher parent- and teacher-reported internalizing problems compared to normative means (Rietman et al., 2017). Another study found higher rates of parent-reported social withdrawal and depression in a sample of children with NF1 and pNFs in comparison to community means (Martin et al., 2012). In a longitudinal analysis using parent and child self-report, Hou et al. (2022) found that children with NF1 and pNFs had more issues with adaptive skills and internalizing problems than same-age peers. Several factors have been found to relate to child psychosocial functioning in this population; for example, poorer cognitive functioning, greater disease severity, and higher environmental stress all related to more social-emotional difficulties (Martin et al., 2012). In addition, research has found the potential for children with familial NF1 to have poorer social-emotional functioning (McNeill et al., 2019) than those with a spontaneous mutation, although results are mixed (Rietman et al., 2018).

Most studies that have examined social-emotional functioning among youth with NF1 have relied solely on observer (e.g., parent proxy) reports. We identified only three studies that obtained ratings from both parents and their children with NF1 using validated measures of social-emotional functioning. McNeill et al. (2019) found that scores on the Behavior Assessment System for Children– Second Edition (BASC-2) Internalizing and ADHD scales, among others, were higher than the normative means according to parent ratings, while only the Attention Problems scale yielded scores higher than normative means per child reports. Furthermore, in a longitudinal study looking at children with NF1 and pNFs, Hou et al. (2022) found that demographic factors differentially predicted child self-reported versus parent proxy-reported child social-emotional functioning on the BASC-2 (Hou et al., 2022). For example, based on parent report, children who were younger, White, and had more educated parents were more at risk for developing social-emotional difficulties over time. In contrast, there were inverse associations based on child report, whereby lower parental education, being an ethnic minority, or having less visible tumors related to greater risk of poor personal adjustment and higher school disconnectedness. Finally, research also has shown that parent ratings of internalizing and externalizing problems on the CBCL were more likely to be in the clinical range than self-report ratings from youth with NF1 (Rietman et al., 2018). Although these NF studies have obtained and compared group parent proxy-report and child-self report ratings, none have compared these ratings directly between individual child-parent dyads.

As evidenced by research in other pediatric populations, examining discrepancies among informants about the child’s functioning is useful for further understanding the factors that may contribute to the social-emotional difficulties of children with NF1. In non-medical community samples, greater discrepancies between parents and children are associated with child depression, maternal depression, and conflicts between mothers and children (Affrunti & Woodruff-Borden, 2015; De Los Reyes et al., 2008; De Los Reyes & Kazdin, 2006). These child and family factors are related to greater discrepancies in which the children generally rated more social-emotional difficulties in comparison to parent proxy-reports (Affrunti & Woodruff-Borden, 2015). In a clinical sample of children recovering from mild traumatic brain injury, significant parent-child discrepancies in reporting internalizing problems on the BASC-2 emerged during periods of greater family stress due to higher parent ratings of their child’s symptoms (Murphy & Dodd, 2021). In another sample of youth with a mild traumatic brain injury, greater parent-child discrepancies in scores on multiple measures of psychosocial functioning occurred among females and older youth (Johnson et al., 2021). A study of adolescents with asthma examined differences across five latent profiles or patterns of parent-adolescent discrepancies in rating adolescent social-emotional functioning (Al Ghriwati et al., 2018). Of those five profiles, two had worse pulmonary functioning and higher parent-rated family conflict: (a) adolescents who agreed with their parents on the presence of elevated social-emotional symptoms, and (b) those who had significant discrepancies in ratings, such that parents reported more symptoms compared to youth self-report.

At the societal level, race and ethnicity have been associated with rater reports of social-emotional functioning, such that parent/child dyads from minority groups had larger discrepancies in comparison to those in the majority (Kim et al., 2016; Lau et al., 2004). Also, parents with higher socioeconomic status (SES) have been found to report fewer problems in their adolescents, leading to increased discrepancies (Chen et al., 2017). In another study using community samples, discrepant dyads in which parents reported more problems tended to be less educated and have lower SES (Van Roy et al., 2010). Given conflicting research regarding the association between SES and discrepancies in ratings, it is important for further research to clarify these relationships.

The frequency of discrepant parent-child reports is understandable but concerning, since clinicians who rely on only one report, or value one person’s report over another, may be missing important information. Moreover, when family members disagree about the child’s problems, they may be less likely to agree on treatment goals and address the problems in a unified way. Thus, it is crucial to understand what patterns of discrepancies are common among families of children with NF1 to inform clinical decision making and to support the inclusion of patient and parent-reported outcome measures in research. To our knowledge, no studies have examined factors that may impact parent-child discrepancies among youth with NF1. The primary aim of the current study is to assess parent-child concordance of social-emotional functioning ratings among youth with NF1 and plexiform neurofibromas (pNFs). Based on previous literature, we hypothesized that statistically significant discrepancies would exist between parent proxy- and child self-reports ratings across four BASC scales (Anxiety, Depression, Atypicality, and Hyperactivity; Hypothesis 1). A secondary aim of the study is to examine child, family, and community factors associated with parent-child rating discrepancies. Next, we hypothesized that dyads with older children and female children would have greater discrepancies between parent and child ratings of social-emotional functioning (Hypothesis 2). Third, we hypothesized that dyads with poorer family functioning or greater parental stress would have greater discrepancies in reporting child social-emotional functioning (Hypothesis 3). Last, given conflicting research regarding SES and rating discrepancies, an exploratory aim was to identify how community prosperity/level of distress relates to parent-child discrepancies in ratings of social-emotional functioning.

Materials and Methods

Participants and Procedures

Individuals ages ≤35 years with a diagnosis of NF1 according to the National Institutes of Health Consensus Conference criteria (Stumpf et al., 1988) or a confirmed NF1 germline mutation with analysis performed in a Clinical Laboratory Improvement Amendments-certified laboratory were eligible to enroll on a natural history protocol at the National Institutes of Health. After being found eligible, participants provided written, informed consent to participate. Youth with NF1 and pNFs, ages 8 to 18 years, who were enrolled in the natural history protocol, and administered at least one comprehensive neuropsychological evaluation that included completed parent proxy- and child self-report questionnaires were identified for this sub-study. Although the natural history study is longitudinal with evaluations approximately every 3 years, the cross-sectional data included in this manuscript are from the first timepoint when participants completed the questionnaires. The natural history protocol was approved by the Institutional Review Board of the National Cancer Institute. The multidisciplinary research study from which these data were obtained was preregistered on https://ClinicalTrials.gov (NCT00924196). We report how we determined our sample size, all data exclusions, all manipulations, and all measures in the study.

Measures

Demographic Information Form.

Parents of participating children with NF1 completed a form at the first assessment to obtain demographic data about the child and family. Data used in this study included the child’s sex, age, race, psychiatric diagnosis (e.g., ADHD, anxiety, depression), whether the mother or father had NF1, as well as the family’s composition of having one or two parents in the household. In our analyses, given small numbers of participants that are racial minorities, race was summarized as participants who identified as White, those who identified as racial minorities (African American, Native Americans, or Hispanic), and those who identified as Asian or “Other” race.

Rating of Overall Stress Scale.

Caregivers rated their overall stress in the past month on a single item from 1–10 (1= not stressed, 10= very stressed), which was adapted from Leidy et al. (2005).

Behavior Assessment System for Children 2nd Edition.

The BASC-2 is a multidimensional parent proxy- and child self-report questionnaire that evaluates clinical and adaptive aspects of behavior and emotional functioning in the community and at home (Reynolds, 2004). This widely used outcome measure assesses 14 scales in the domains of Internalizing Problems, Externalizing Problems, Behavioral Symptoms, and Adaptive Skills. For this study, we used the scales that were similar across child and parent forms, which included Depression, Anxiety, Hyperactivity, and Atypicality (unusual thoughts or behaviors). The BASC-2 yields standardized T scores (M = 50, SD = 10); scores between 60 and 69 are in the “at-risk” range and 70 or higher are in the “clinically significant” range. Discrepancy scores were calculated as the absolute value of difference between parent and child ratings of symptoms. The BASC-2 is a reliable and valid measure for assessing behavioral and emotional functioning (Reynolds, 2004; Song et al., 2017).

Family Assessment Device.

This parent-report questionnaire assesses family functioning in six domains, such as communication, affective responsiveness, problem solving, and roles, as well as an overall General Functioning subscale obtained from 12 items. FAD items are rated on a Likert scale from 1 (strongly disagree) to 4 (strongly agree) with higher scores indicating poorer functioning. The Family Communication and General Functioning subscales were used in the current study (Epstein, Baldwin, & Bishop, 1983). The Family Assessment Device has been shown to be a reliable and valid measure for assessing family functioning in other studies (Byles et al., 1988; Mansfield, Keitner, & Dealy, 2015; Miller et al., 1985).

Distressed Community Index.

The Distressed Community Index (DCI) is a measure of economic well-being that is used as a proxy of SES. It is obtained by combining seven metrics, including education, poverty, employment, housing vacancy, income, change in employment, and change in establishments, into one index based on an individual’s zip code (https://eig.org/dci). The scores range from 0 to 100, and communities are grouped into quintiles, with the highest quintile called “prosperous” and the lowest quintile identified as “distressed.” The data used to calculate the DCI is obtained from the U.S. Census Bureau’s American Community Survey 5-Year Estimates and Business Patterns products. The DCI has been used in other studies assessing child and family functioning (Williamson et al., 2021; Williamson & Mindell, 2020).

Behavior Rating Inventory of Executive Function.

The Behavior Rating Inventory of Executive Function (BRIEF) is a parent proxy questionnaire assessing child executive functioning at home or at school (Gioia, 2000). The BRIEF contains 86 items (1= never, 2= sometimes, 3= always) that assesses the frequency of problems with specific behaviors. This measure gives eight subscales that combine to give Behavioral Regulation Index (BRI) and Metacognition Index (MI) scales that have been shown to reliably measure executive functioning (Gioia et al., 2002; Gioia, 2000).

Data Analysis

All data analyses were conducted using IBM SPSS version 28.0.0. To address the first aim and hypothesis, paired samples t-tests examined differences between parent and child ratings of child functioning on the following BASC-2 scales: Depression, Anxiety, Atypicality, and Hyperactivity. Then, difference scores between parent and child ratings of symptoms were calculated and the absolute value identified discrepancy scores between parent and child ratings on these scales. To address our second and third aims, linear regression, analyses of variance (ANOVAs), and independent samples t-tests were used to assess whether any community, family, or child characteristics were associated with parent-child discrepancies. Because the DCI is a newer metric, we looked at the association of both the DCI categories and overall scores with discrepancy ratings. The α was set at .05, and since this sub-study was exploratory in nature, we did not adjust for multiple comparisons. The data that support the findings of this study are available from the corresponding author upon reasonable request.

Results

Eighty-seven youth with NF1 and pNFs (Mage= 12.40, SD=2.89; range 8–18 years; 54% male; 78% White) and their caregivers (77% mothers, 18% fathers, 3% other) participated in this study. According to Distressed Communities Index (DCI) metrics, 26% of participants were living in prosperous communities and 16% were living in distressed communities. Seventy-two percent of youth had families consisting of two parent/guardian households. Detailed descriptive data are presented in Table 1.

Table 1.

Demographic Characteristics of Sample

Variable M(SD) Range
Age 12.40 (2.89) 7.71–18.96
DCI 41.79 (28.01) 0.7–99.0
General Family Functioning (FAD mean) 1.64 (0.51) 1–3
FAD Family Communication 2.76 (0.29) 2.17–3.5
N %
Child Sex
   Male 47 54.0
   Female 40 46.0
Racea
   White 68 78.2
   Black 5 5.7
   Hispanic 3 3.4
   Asian 2 2.3
   Other 9 10.3
Any Psychiatric Diagnosis 38 43.7
Learning Disability Diagnosis 32 36.8
ADHD Diagnosis 31 35.6
Homeschooled 6 6.9
Family Composition
   Two Parent/Guardians 63 72.4
   One Parent/Guardian 24 27.6
Familial or Sporadic NF1
   Familial NF1 34 39.1
   Sporadic NF1 44 50.6
DCI Categories
  Prosperous 23 26.4
  Comfortable 25 28.7
  Midtier 14 16.1
  At-risk 9 10.3
  Distressed 14 16.1
  Missing 2 2.3
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Note. ADHD = attention-deficit hyperactivity disorder; DCI = Distressed Community Index; FAD = Family Assessment Device; NF1 = neurofibromatosis type 1.

a

These categories of race/ethnicity were assessed in one item asking participants to select their race on the background form.

According to parent ratings, 24.1% of the sample fell within the at-risk or clinically significant range for anxiety, 21.8% for atypicality, 27.6% for depression, and 21.8% for hyperactivity/impulsivity. According to child self-report ratings, 18.4% of the sample fell within the at-risk or clinically significant range for anxiety, 14.9% for atypicality, 6.9% for depression, and 21.8% for hyperactivity/impulsivity.

Paired samples t tests comparing parent proxy and child self-report ratings yielded several significant discrepancies in responses. First, parents provided significantly higher ratings of anxiety in comparison to their child’s self-reports t(85) = 3.82, p < .001. Second, there were statistically significant differences in ratings between parent proxy- and child self-reports of depression, whereby parents’ responses resulted in higher scores in comparison to their child t(85) = 5.25, p < .001. Finally, there were significant parent-child discrepancies in ratings of youth symptoms within the Atypicality scale, with parents again reporting more severe symptoms than their children t(85) = 2.63, p = .01. Results did not indicate significant parent-child differences in the hyperactivity domain t(85) = 1.75, p = .08. The mean scores of the parents and children as well as the mean discrepancies are reported in Table 2.

Table 2.

Paired Samples T Tests Comparing Caregiver and Youth Ratings of Child Functioning

Variable Parent/Caregiver T-scores Child T-scores Difference t (df) p-value
M SD M SD M
Anxiety 54.72 11.21 49.86 10.00 4.86 3.82 (85) <.001
Depression 54.92 10.98 48.23 8.04 6.69 5.25 (85) <.001
Atypicality 52.94 10.47 49.53 8.32 3.41 2.63 (85) .01
Hyperactivity 53.34 12.10 50.93 10.81 2.41 1.75 (84) .08
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Child Factors Associated with Discrepancies

Several child factors were associated with parent-child discrepancies in ratings of youth functioning. First, dyads with female children had significantly greater discrepancies in ratings of depressive symptoms, with parents rating higher symptoms, in comparison to dyads with male children t(53.89) = 2.13, p = 0.038; Mdiff = 4.41. Results suggested no other sex differences in parent-child discrepancies of rating youth anxiety, atypicality, or hyperactivity. Youth age did not predict discrepancies in ratings across any domains (all ps > .05). Moreover, there were no statistically significant differences across dyads from different racial groups (White vs. racial minorities vs. individuals who identify as Asian or “Other” race) with regards to discrepancy scores across all domains (all ps > .05). No differences were apparent in parent-child discrepancies across any BASC-2 domains with regards to the child having a psychiatric diagnosis (all ps> .05) or learning disability diagnosis (all ps> .05) in comparison to dyads with no diagnosis. Finally, youth with ADHD had larger discrepancies with their parents in hyperactivity ratings than those without ADHD, t(48.30) = 2.18, p = .034; Mdiff = 4.40). More specifically, parents of children with ADHD rated higher hyperactivity symptoms compared to their child’s self-reports than parents of children without ADHD.

Associations between caregiver ratings of the child’s executive functioning on the BRIEF and parent-child discrepancies in BASC-2 ratings also were examined via linear regressions. Results suggested that higher Behavior Regulation Index scores were significantly associated with larger differences in parent-child ratings of the child’s symptoms of depression, atypicality, and hyperactivity, but not with anxiety. Finally, results suggest that higher Metacognition Index scores were associated with greater differences in discrepancies of rating child hyperactivity (b = 0.35, p = .001), but there were no associations with discrepancies in anxiety, depression, or atypical behavior.

Family Factors Associated with Discrepancies

With regards to family factors associated with parent-child discrepancies in rating youth symptoms, linear regressions suggested that poorer overall family functioning significantly predicted larger discrepancies in ratings of youth anxiety (b = 0.26, p = .025). However, differences in overall family functioning were not associated with discrepancies in ratings of depression, atypicality, or hyperactivity (all ps > .05). Differences in family communication did not predict discrepancies in ratings across any of the BASC-2 common domains (all ps > .05). Further, there were no statistically significant differences in discrepancies observed between families consisting of two parents in comparison to families with one parent (all ps > .05) across all domains. Finally, there were no differences in discrepancies between mother versus father raters across any domains (all ps > .05).

Linear regression suggested a significant association between parental stress and parent-youth discrepancies in ratings of atypicality. In particular, higher overall parental stress on the Rating of Overall Stress Scale significantly predicted greater discrepancies in ratings of atypicality (b = .24, p = .035). Differences in overall parental stress did not significantly predict discrepancies in ratings of depression, anxiety, or hyperactivity (all ps > .05). No significant differences emerged between youth with versus without a parent with NF1 with regards to discrepancies in ratings across all domains (all ps > .05). Finally, linear regression suggested no significant associations between maternal or paternal years of education and discrepancies across all domains (all ps > .05).

Community Factors Associated with Discrepancies

When examining associations between DCI categories and parent-child discrepancies in ratings, no differences emerged across DCI quintiles in any BASC-2 domain (all ps > .05). When condensing DCI categories to 3 groups (distressed/at-risk, midtier, comfortable/prosperous), there were also no significant differences between groups in discrepancy ratings (all ps > .05). Finally, linear regression suggested no significant associations between DCI scores and rating discrepancies (all ps > .05).

Results summarizing associations between child, family, and community factors with parent child discrepancies in ratings organized by BASC domains are presented in Table 3.

Table 3.

Analyses of Child, Family, and Community Factors and Discrepancies in Ratings by BASC Domains

Predictors Analysis Test statistic P-value
Predictors of Discrepancies in Anxiety Ratings
Sex T-test −0.08 .933
Age Regression 0.19 .128
Race ANOVA 0.022 .978
Psychiatric Diagnosis T test 0.474 .637
Learning Disability T test −0.695 .491
ADHD Diagnosis T test 0.170 .865
BRIEF BRI Regression 0.123 .262
BRIEF MCI Regression −0.022 .839
Familial or Sporadic NF T-test 1.113 .269
Family Communication Regression −0.027 .815
Family Functioning Regression 0.255 .025*
Parental Stress (ROSS) Regression −0.063 .575
DCI ANOVA 1.539 .221
Predictors of Discrepancies in Depression Ratings
Sex T-test 2.13 .038*
Age Regression 0.23 .059
Race ANOVA 0.233 .793
Psychiatric Diagnosis T test 0.591 .556
Learning Disability T test −0.892 .376
ADHD Diagnosis T test −0.638 .526
BRIEF BRI Regression 0.345 .001**
BRIEF MCI Regression 0.061 .578
Familial or Sporadic NF T-test 0.594 .554
Family Communication Regression 0.013 .912
Family Functioning Regression 0.018 .878
Parental Stress (ROSS) Regression 0.148 .188
DCI ANOVA 1.124 .330
Predictors of Discrepancies in Hyperactivity Ratings
Sex T-test −0.93 .355
Age Regression −0.089 .420
Race ANOVA 0.174 .841
Psychiatric Diagnosis T test −1.40 .167
Learning Disability T test 0.166 .869
ADHD Diagnosis T test −2.181 .034*
BRIEF BRI Regression 0.472 <.001**
BRIEF MCI Regression 0.348 .001**
Familial or Sporadic NF T-test 0.422 .674
Family Communication Regression 0.049 .675
Family Functioning Regression 0.116 .317
Parental Stress (ROSS) Regression 0.154 .174
DCI ANOVA 0.783 .461
Predictors of Discrepancies in Rating Atypicality
Sex T-test 0.046 .963
Age Regression 0.022 .841
Race ANOVA 1.025 .363
Psychiatric Diagnosis T test 0.016 .987
Learning Disability T test −0.309 .758
ADHD Diagnosis T test −1.814 .074
BRIEF BRI Regression 0.431 <.001**
BRIEF MCI Regression 0.152 .166
Familial or Sporadic NF T-test 1.05 .297
Family Communication Regression −0.075 .517
Family Functioning Regression 0.161 .163
Parental Stress (ROSS) Regression 0.235 .035*
DCI ANOVA 1.693 .190
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Note. ADHD = attention-deficit hyperactivity disorder; BASC = Behavior Assessment System for Children; ANOVA = analysis of variance; BRIEF = Behavior Rating Inventory of Executive Function; BRI = Behavioral Regulation Index; MCI = Metacognition Index; NF = neurofibromatosis; ROSS = Rating of Overall Stress Scale; DCI = Distressed Community Index.

*

p < .05.

**

p < .01.

Discussion

To our knowledge, this is the first study in the NF1 population to analyze discrepancies between parent-child dyads in their ratings of child functioning to shed light on the unique perspectives of respondents within a family. In support of our first hypothesis, our results confirmed the presence of discrepancies between parent proxy- and child self-report ratings of child functioning on BASC-2 domains of depression, anxiety, and atypicality. We determined that youth with NF1 and pNFs generally rated lower symptoms across domains of anxiety, depression, and atypicality when compared to their parent’s ratings on proxy reports. Our findings are consistent with another study of youth with NF1 showing that children endorsed fewer emotional and behavioral problems (e.g., internalizing and externalizing symptoms) compared to parent proxy-reports (Rietman et al., 2018). However, these results are in contrast to other research from community samples indicating that children generally provide higher ratings of psychosocial and anxiety symptoms compared to parent proxy-reports (Affrunti & Woodruff-Borden, 2015). These differences across the existing literature may be related to the population studied, in particular, whether the participants have a chronic medical illness or are from a community sample.

In partial support of our second hypothesis, certain child characteristics predicted differences in ratings of child functioning. There were greater parent-child discrepancies in depression symptoms within dyads of female children due to higher parental ratings in comparison to dyads with male children. However, outside of depression scores, child sex did not otherwise predict discrepancies in ratings. Other studies have similarly noted inconsistent findings when examining whether child sex is related to parent-child discrepancies in ratings across domains (Affrunti & Woodruff-Borden, 2015; De Los Reyes & Kazdin, 2006). In the current sample, it is unclear why child sex did not predict discrepancies in ratings of anxiety, atypicality, or hyperactivity symptoms. These results may be because parents assume that their children with NF1 and pNFs are more affected by having a chronic illness, which similarly affects both males and females. Future research may examine factors that explain why parent-child discrepancies in rating depression, but not other BASC domains, were different for male and female children.

Our finding that child age did not impact discrepancies in ratings in any domain did not support our second hypothesis. While some studies have shown an effect of age on parent-child rating discrepancies (De Los Reyes & Kazdin, 2006; De Los Reyes & Ohannessian, 2016), other research studies have mixed results (Affrunti & Woodruff-Borden, 2015). These inconsistencies across studies may be due to the assessment of different domains (e.g., depression vs. anxiety vs. overall functioning) and the use of different measures of psychosocial functioning. In the present study, age may not have significantly impacted parent versus child reporting given that we generally had a younger sample of children compared to other reports of parent-child discrepancies. Future studies could examine the longitudinal effects of child age on parent-child discrepancies to identify whether there are changes in concordance across different developmental periods.

Child ADHD diagnosis was associated with greater differences in ratings of hyperactivity whereby parents rated higher overall symptoms in comparison to child self-report. Results are consistent with the findings of Rietman et al. (2018) whereby an ADHD diagnosis was related to higher parent-reported but not child self-reports of attention problems. Similarly, greater difficulties in executive functioning in the current study were associated with more parent-child discrepancies in ratings. This finding further highlights that groups with specific characteristics may be more prone to discrepancies in certain domains than others. Our results also suggest that individuals with ADHD or greater executive functioning difficulties may have more limited awareness of their psychosocial functioning (Factor, Rosen, & Reyes, 2016) or underreport symptoms as a self-protective mechanism (Rietman et al., 2018).

In partial support of our third hypothesis regarding family factors, we found that higher parental stress significantly predicted discrepancies between child self- and parent proxy-report of atypicality. These results are consistent with prior literature, suggesting an association between parental stress and parent child-discrepancies in rating psychosocial functioning. Specifically, De Los Reyes and Kazdin (2006) found that parents who are more stressed may be more likely to report poorer child functioning compared to those who are less stressed. Furthermore, poorer family functioning significantly predicted discrepancies between child and parent reports of anxiety, possibly due to weaknesses in family communication or an overall elevated level of family conflict. Since disease severity may affect parental stress and family functioning, future studies should explore whether illness severity (e.g., disease severity, physical complications, tumor visibility, activity limitations) contributes to discrepancies in ratings (Pinquart, 2018).

Across the aforementioned variables, results suggest that psychologists or other mental health professionals should identify families with NF1 who have greater discrepancies in their ratings to improve communication and cohesion. In turn, this strategy may result in fewer discrepancies and more agreement regarding psychosocial functioning of the child, which will help identify children most in need of interventions and unite families in their pursuit of psychological support.

Finally, our exploratory aim examined how community factors predicted discrepancies in parent proxy- and child self-report. Exploring the role of SES, our results did not suggest the presence of significant associations between DCI categories and ratings of parent-child discrepancies. However, there are multiple indices of SES and community health outside of the DCI, and future studies should use these to further explore associations between SES and discrepancies in reporting.

This study contained some observable limitations. First of all, the BASC-2 only has four scales that are rated by both children and parents, thus limiting the domains that we were able to analyze for discrepancies. Considering the significant link between cognitive and social-emotional functioning in NF1 (Martin et al., 2012), future studies could incorporate objective measures of child cognitive functioning to evaluate how these measures may relate to reporting differences. Furthermore, this study contained a relatively homogenous sample, with a majority of parental reporters being mothers and being of the same race and family structure, which limits our ability to assess across group differences. While we did not see a significant difference between mothers and fathers, it would be interesting to examine how different family structures influence rates of discrepancies. Parental psychopathology also may play a role in rating discrepancies (Affrunti & Woodruff-Borden, 2015), and future investigations are encouraged to explore this line of research. We also acknowledge that having only one index of SES is a limitation, and having other indices of SES and community health, such as household income, is important for future studies. In addition, we chose to measure discrepancies through the absolute value of score differences between parent proxy- and child self-report; future research could use latent profile analysis to categorize discrepancies across multiple domains. Finally, this study contained a fairly large age range, with children ranging from middle childhood to late adolescence; considering the large differences in development during these years, future studies with larger samples could separate age cohorts to examine discrepancies across developmental periods.

In summary, this study highlights the importance of multi-informant reporting of social-emotional functioning in youth with NF1. In other populations, discrepancies between child and parent report are associated with a litany of psychosocial problems and conflicts, such as child depression and anxiety. This work adds to that literature by directly examining these ratings in the NF1 population and examining child, family, and community factors that may lead to higher discrepancies. In the research community, garnering this information from multiple informants to gather their unique perspectives will lead to a more holistic and accurate representation of a given population. In practice, knowing which groups and domains are more prone to discrepancies may help clinicians to better assess and treat the mental health concerns of children and adolescents and their parents. In research, collecting both parent and child reports of child psychosocial functioning may be helpful for evaluating outcomes in psychosocial and medical interventional trials.

Author Note

Nour Al Ghriwati and Paige Little contributed equally to this article. Some of the results in this article were presented at the Society for Pediatric Psychology Annual Conference in March 2022. The multidisciplinary research study from which these data were obtained was preregistered on https://ClinicalTrials.gov (NCT00924196). The data that support the findings of this study are available from the corresponding author upon reasonable request.

This research was supported by the Intramural Research Program of the National Institutes of Health, the National Cancer Institute, and the Pediatric Oncology Branch. This project has been funded in whole or in part with federal funds from the National Cancer Institute, National Institutes of Health, under Contract No. (75N91019D00024). The content of this publication does not necessarily reflect the views or policies of the Department of Health and Human Services nor does mention of trade names, commercial products, or organizations imply endorsement by the U.S. government.

The authors thank the children and caregivers who participated in this study. We also thank all members of the Health Psychology and Neurobehavioral Research Group and the Neurofibromatosis Team of the Pediatric Oncology Branch, Center for Cancer Research, National Cancer Institute for their assistance with data collection and the care of patients on the NF1 Natural History study (https://ClinicalTrials.gov identifier NCT00924196).

Nour Al Ghriwati played a lead role in formal analysis and an equal role in conceptualization, writing–original draft, and writing–review and editing. Paige Little played a lead role in writing–review and editing and an equal role in conceptualization and writing–original draft. Staci Martin played a supporting role in conceptualization and writing–original draft and an equal role in methodology, supervision, and writing–review and editing. Mary Anne Tamula played a supporting role in project administration, writing–original draft, and writing–review and editing. Brigitte C. Widemann played an equal role in investigation and writing–review and editing. Pamela L. Wolters played a lead role in supervision and an equal role in conceptualization, methodology, writing–original draft, and writing–review and editing.

Footnotes

The authors have no conflicts of interest to report.

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