Background

We aimed to examine symptom load in a clinical adolescent population at three-year follow-up and explore associations with standard care treatment procedures and resilience factors upon first presenting at Child and Adolescent Mental Health Services.

Methods

This study is part of a prospective longitudinal cohort study: The Health Survey in Department of Children and Youth, St. Olavs hospital, Norway. A clinical population of 717 (43.5% of eligible) adolescents aged 13-18 years participated in the first study visit (T₁, 2009-2011). Of these, 447 adolescents with psychiatric disorders, with treatment history from medical records and self-reported resilience factors (Resilience Scale for Adolescents; READ) at T₁, reported symptom load (Achenbach System of Empirically Based Assessment - Youth Self Report; YSR) three years later aged 16-21 years (T₂).

Result

At T₁, 93.0% received individual treatment. The frequency of psychotherapy and medication varied by disorder group and between genders. Overall, psychotherapy was more frequent among girls, whereas medication was more common among boys. Total READ mean value (overall 3.5, SD 0.8), ranged from patients with mood disorders (3.0, SD 0.7) to patients with Attention Deficit Hyperactivity disorder (3.7, SD 0.7), and was lower for girls than boys in all diagnostic groups. At T₂, the YSR Total Problem mean T-score ranged across the diagnostic groups (48.7, SD 24.0 to 62.7, SD 30.2), with highest symptom scores for those with mood disorders at T₁, of whom 48.6% had T-scores in the borderline/clinical range (≥60) three years later. Number of psychotherapy sessions was positively associated and Total READ score was negatively associated with the YSR Total Problems T-score (regression coefficient β = 0.5, CI (0.3 to 0.7), p < 0.001 and β = - 15.7, CI (- 19.2 to - 12.1), p < 0.001, respectively). The subscale Personal Competence was associated with the lowest Total Problem score for both genders.

Conclusions

Self-reported symptom load was substantial after three years, despite comprehensive treatment procedures. Higher self-reported resilience characteristics were associated with lower symptom load after three years. These results highlight the burden of adolescent psychiatric disorders, the need for extensive interventions and the importance of resilience factors for a positive outcome.

BACKGROUNDUndifferentiated systemic autoinflammatory diseases (USAIDs) present diagnostic and therapeutic challenges. Chronic interferon (IFN) signaling and cytokine dysregulation may identify diseases with available targeted treatments.METHODSSixty-six consecutively referred USAID patients underwent underwent screening for the presence of an interferon signature using a standardized type-I IFN-response-gene score (IRG-S), cytokine profiling, and genetic evaluation by next-generation sequencing.RESULTSThirty-six USAID patients (55%) had elevated IRG-S. Neutrophilic panniculitis (40% vs. 0%), basal ganglia calcifications (46% vs. 0%), interstitial lung disease (47% vs. 5%), and myositis (60% vs. 10%) were more prevalent in patients with elevated IRG-S. Moderate IRG-S elevation and highly elevated serum IL-18 distinguished 8 patients with pulmonary alveolar proteinosis (PAP) and recurrent macrophage activation syndrome (MAS). Among patients with panniculitis and progressive cytopenias, 2 patients were compound heterozygous for potentially novel LRBA mutations, 4 patients harbored potentially novel splice variants in IKBKG (which encodes NF-κB essential modulator [NEMO]), and 6 patients had de novo frameshift mutations in SAMD9L. Of additional 12 patients with elevated IRG-S and CANDLE-, SAVI- or Aicardi-Goutières syndrome-like (AGS-like) phenotypes, 5 patients carried mutations in either SAMHD1, TREX1, PSMB8, or PSMG2. Two patients had anti-MDA5 autoantibody-positive juvenile dermatomyositis, and 7 could not be classified. Patients with LRBA, IKBKG, and SAMD9L mutations showed a pattern of IRG elevation that suggests prominent NF-κB activation different from the canonical interferonopathies CANDLE, SAVI, and AGS.CONCLUSIONSIn patients with elevated IRG-S, we identified characteristic clinical features and 3 additional autoinflammatory diseases: IL-18-mediated PAP and recurrent MAS (IL-18PAP-MAS), NEMO deleted exon 5-autoinflammatory syndrome (NEMO-NDAS), and SAMD9L-associated autoinflammatory disease (SAMD9L-SAAD). The IRG-S expands the diagnostic armamentarium in evaluating USAIDs and points to different pathways regulating IRG expression.TRIAL REGISTRATIONClinicalTrials.gov NCT02974595.FUNDINGThe Intramural Research Program of the NIH, NIAID, NIAMS, and the Clinical Center.