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Patient-Proxy Agreement on Mental Health and Neuropsychological Symptoms Among Youth with Juvenile Fibromyalgia Syndrome

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Background/Purpose: Adolescents with juvenile fibromyalgia syndrome (JFMS) present with a myriad of mental health and neuropsychological symptoms, including dyscognition (“brain fog”); however, patient-proxy agreement on the severity of these co-morbidities is unclear as are their psychosocial contributors. We aimed to 1) determine patient-proxy agreement on mental health and neuropsychological co-morbidities, and 2) identify psychosocial factors associated with the presence of subjective dyscognition in JFMS.

Methods: This was a cross-sectional cohort study of children 12-17 years old diagnosed with JFMS according to the 2010 ACR criteria, seen in a specialty pediatric rheumatology pain clinic from 7/2017-9/2019, and one of their caregivers. Exclusion criteria included non-English speaking patients, use of stimulant medication, and a medical condition precluding completion of assessments. All subjects completed study questionnaires, complemented by retrospective chart review. Subjects with elevated scores (≥ 65) on The Children’s Depression Inventory (CDI-2) or endorsing item 8 (suicidal thoughts) on the CDI-2 triggered the study’s mental health (MH) safety check and were assessed for active suicidal ideation. We assessed patient-proxy agreement using intra-class correlation coefficients (ICCs) and mean differences between patient and proxy scores using Wilcoxon signed-rank test. We also performed multivariate linear regression analyses to identify factors independently associated with subjective dyscognition as measured by the Pediatric Quality of Life (PedsQL) Cognitive Functioning Scale.

Results: Thirty-one patients completed the study. The majority were female (87%), non-Hispanic (90%), and Caucasian (81%). Median age at enrollment was 15 years (IQR: 14-16). Patients had a median pain duration of 12 months (IQR: 6-36) and a median visual analog pain score (0-100) of 59 (IQR: 32-68). According to the Resilience Scale 14-item, patient resilience was low (mean=69, SD=16) and parental resilience was moderate (mean=81, SD=12). Sixteen subjects (52%) triggered a mental health safety check, 8 of whom endorsed suicidal ideation on the CDI-2. None had an active plan or intent. Patient-proxy agreement was good to excellent for all measures (Table 1) and this agreement was stronger among patients who triggered the MH safety check (Table 2). In bivariate analyses, greater anxiety (β=-0.76 [-1.30, -0.21]), depression (β=-0.87 [-1.51, -0.23]), functional disability (β=-0.87 [-1.72, 0.02]) and lower patient resilience (β=0.49 [-0.06, 1.04]) were significantly associated with dyscognition (Table 3). In adjusted analyses, depression (β=-1.88 [-3.21, -0.55]) remained independently associated with greater dyscognition (p=0.01).

Conclusion: Patient-proxy agreement on mental health and neuropsychological symptoms was good to excellent in JFMS. This agreement was strengthened among adolescents with greater depression, which was also found to be an independent predictor of dyscognition. These findings suggest that parental reports of symptoms among youth with JFMS are reliable, especially when there is a large mental health burden among affected youth.

Legend: Functional disability inventory (FDI) scores range from 0_60 with greater scores indicating more functional disability. Pediatric Quality of Life Inventory (PedsQL) Multidimensional Fatigue Scale (MFS) range from 0_100 where higher scores indicate less symptoms/problems. Behavior Rating Inventory of Executive Function_2 (BRIEF_2) is a standardized rating scale used to assess children’s executive functioning where T scores from 60 to 64 are considered mildly elevated, and T scores from 65 to 69 are considered potentially clinically elevated. The Children’s Depression Inventory, 2nd Edition (CDI_2) is an assessment of depressive symptoms, where T_scores ≥ 65 identify potentially clinically depressed individuals. The Multidimensional Anxiety Scale for Children, 2nd Edition (MASC_2) assesses anxiety symptoms in youth where T_scores ≥ 60 indicate increased likelihood of at least one anxiety disorder in the subject. Difference between patient and proxy mean scores were assessed with the Wilcoxon signed_rank test (2 tailed). Intra_class correlation coefficients (ICCs) were rated as follows: poor agreement (≤0.40), fair agreement (0.41 to 0.59), good agreement (0.60 to 0.74), and excellent agreement (≥0.75).

Legend: pᶺ = Non_significant p_value. Functional disability inventory (FDI) scores range from 0_60 with greater scores indicating more functional disability. Pediatric Quality of Life Inventory (PedsQL) Multidimensional Fatigue Scale (MFS) range from 0_100 where higher scores indicate less symptoms/problems. Behavior Rating Inventory of Executive Function_2 (BRIEF_2) is a standardized rating scale used to assess children’s executive functioning where T scores from 60 to 64 are considered mildly elevated, and T scores from 65 to 69 are considered potentially clinically elevated. The Children’s Depression Inventory, 2nd Edition (CDI_2) is an assessment of depressive symptoms, where T_scores ≥ 65 identify potentially clinically depressed individuals. The Multidimensional Anxiety Scale for Children, 2nd Edition (MASC_2) assesses anxiety symptoms in youth where T_scores ≥ 60 indicate increased likelihood of at least one anxiety disorder in the subject. Intra_class correlation coefficients (ICCs) were rated as follows: poor agreement (≤0.40), fair agreement (0.41 to 0.59), good agreement (0.60 to 0.74), and excellent agreement (≥0.75).

Legend: pƗ = Significant p_value. Anxiety was removed from the multivariate model due to co_linearity. FDI=functional disability inventory. CDI_2= The Children’s Depression Inventory, 2nd Edition. MASC_2 = The Multidimensional Anxiety Scale for Children, 2nd Edition. RS_14=Resilience Scale 14_item. The Peds Cognitive Functioning Scale ranges from 0_100 where higher scores indicate less symptoms/problems.

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