Body-Focused Repetitive Behavior Disorders in Children and Adolescents: Clinical Characteristics and Treatment Outcomes in a Naturalistic Setting.

Body-focused repetitive behavior disorders, including trichotillomania (hair-pulling disorder) and excoriation (skin picking) disorder, typically emerge in early adolescence, but little is known about the clinical characteristics and treatment outcomes of these disorders in young people, particularly in real-world clinical settings. Participants were 63 children and adolescents (51 girls; age range 9-17) with a diagnosis of trichotillomania (n = 33) and/or skin-picking disorder (n = 33) attending a specialist outpatient clinic in Stockholm, Sweden. Demographic and clinical characteristics were gathered at the initial assessment. Of the 63 assessed youths, 56 received manual-based behavior therapy mainly focusing on habit reversal training, which was combined with medication when deemed appropriate. The mean clinician-reported trichotillomania and skin-picking disorder symptom severity at baseline (n = 63) was in the moderate range. We observed high rates of psychiatric comorbidity (63.5%) and use of psychiatric medication (54.8%). For the 56 individuals undertaking treatment at the clinic, mixed-effects regression models showed a significant decrease in symptom severity from baseline to posttreatment, with gains maintained up to the 12-month follow-up. Substantial and durable improvements were also seen on self-reported symptoms, self-reported depression, and global functioning. Specialist care should be made more widely available to improve the prognosis and quality of life of young people with trichotillomania and skin-picking disorder.

Effectiveness of Multimodal Treatment for Young People With Body Dysmorphic Disorder in Two Specialist Clinics.

Body dysmorphic disorder (BDD) typically originates in adolescence and is associated with considerable adversity. Evidence-based treatments exist but research on clinical outcomes in naturalistic settings is extremely scarce. We evaluated the short- and long-term outcomes of a large cohort of adolescents with BDD receiving specialist multimodal treatment and examined predictors of symptom improvement. We followed 140 young people (age range 10-18) with a diagnosis of BDD treated at two national and specialist outpatient clinics in Stockholm, Sweden (n = 96) and London, England (n = 44), between January 2015 and April 2021. Participants received multimodal treatment consisting of cognitive behavior therapy and, in 72% of cases, medication (primarily selective serotonin reuptake inhibitors). Data were collected at baseline, posttreatment, and 3, 6, and 12 months after treatment.The primary outcome measure was the clinician-rated Yale-Brown Obsessive-Compulsive Scale Modified for BDD, Adolescent version (BDD-YBOCS-A). Secondary outcomes included self-reported measures of BDD symptoms, depressive symptoms, and global functioning. Mixed-effects regression models showed that BDD-YBOCS-A scores decreased significantly from baseline to posttreatment (coefficient [95% confidence interval] = -16.33 [-17.90 to -14.76], p<0.001; within-group effect size (Cohen's d) = 2.08 (95% confidence interval, 1.81 to 2.35). At the end of the treatment, 79% of the participants were classified as responders and 59% as full or partial remitters. BDD symptoms continued to improve throughout the follow-up. Improvement was also seen on all secondary outcome measures. Linear regression models identified baseline BDD symptom severity as a predictor of treatment outcome at posttreatment, but no consistent predictors were found at the 12-month follow-up. To conclude, multimodal treatment for adolescent BDD is effective in both the short- and long-term when provided flexibly within a specialist setting. Considering the high personal and societal costs of BDD, specialist care should be made more widely available.

Feasibility of internet-delivered cognitive-behavior therapy for obsessive-compulsive disorder in youth with autism spectrum disorder: A clinical benchmark study.

Obsessive-compulsive disorder (OCD) is a treatable condition that often requires specialist care, particularly when comorbid with autism spectrum disorder (ASD). However, specialist clinics are few and typically located in large medical centers. To increase availability of evidence-based treatment for OCD in individuals with ASD, we adapted an internet-delivered cognitive behavior therapy (ICBT) protocol to suit the needs of these individuals and conducted a feasibility study (N = 22). The primary outcome was the clinician-rated Children's Yale-Brown Obsessive-Compulsive Scale (CY-BOCS), administered at pre- and post-treatment as well as 3 months after treatment. ICBT was deemed acceptable and was associated with clinically significant improvements in CY-BOCS scores, corresponding to a large within-group effect size (Cohen's d = 1.33). Similarly, significant improvements were observed in most of the secondary parent- and self-rated measures. Approximately 60% of the participants were classed as treatment responders and 50% were in remission from their OCD at the 3-month follow-up. To provide a meaningful benchmark, we also analyzed data from a specialist clinic that regularly treats individuals with comorbid OCD and ASD (N = 52). These analyses indicated that specialized in-person CBT produced significantly larger effects (d = 2.69) while being markedly more resource demanding, compared to ICBT. To conclude, ICBT can be successfully adapted to treat OCD in youth with ASD and may be a viable alternative for those who do not have direct access to highly specialized treatment. Further improvements of the treatment protocol based on participant and therapist feedback are warranted, as is a formal test of its efficacy and cost-effectiveness in a randomized controlled trial.

Insomnia in Tourette Syndrome and Chronic Tic Disorder.

BACKGROUND: Insomnia is common in Tourette syndrome (TS) and chronic tic disorder (CTD), but precise prevalence estimates are lacking. OBJECTIVE: In this Swedish register-based cohort study, we estimated the prevalence of insomnia in TS/CTD and quantified the magnitude of this association, accounting for familial confounders and relevant somatic and psychiatric comorbidities. METHODS: Of 10,444,702 individuals living in Sweden during the period from 1997 to 2013, 5877 had a diagnosis of TS/CTD and were compared to unexposed individuals from the general population on the presence of insomnia using logistic regression models. RESULTS: Individuals with TS/CTD had a period prevalence of insomnia of 32.16%, compared to 13.70% of the unexposed population. This translated into a 6.7-fold increased likelihood of insomnia in TS/CTD (odds ratio adjusted [aOR] for sex, birth year, birth country, and somatic disorders = 6.74; 95% confidence interval [CI], 6.37-7.15). A full sibling comparison, designed to adjust for shared familial factors, attenuated the estimates (aOR = 5.41; 95% CI, 4.65-6.30). When individuals with attention-deficit/hyperactivity disorder (ADHD) and pervasive developmental disorders were excluded, the association was also attenuated, whereas exclusion of other psychiatric comorbidities had minimal impact. Having persistent TS/CTD, comorbid ADHD, and taking ADHD medication greatly increased the likelihood of insomnia. CONCLUSIONS: Insomnia is significantly associated with TS/CTD, independently from somatic disorders, familial factors or psychiatric comorbidities, although familial factors, neurodevelopmental comorbidities, and ADHD/ADHD medication may explain part of the association. Insomnia should be routinely assessed and managed in TS/CTD, particularly in chronic patients and in those with comorbid ADHD. Other sleep disorders require further study. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.

Clinical characteristics of 172 children and adolescents with body dysmorphic disorder.

Body dysmorphic disorder (BDD) often starts in childhood, with most cases developing symptoms before age 18. Yet, BDD research has primarily focused on adults. We report the clinical characteristics of the world's largest cohort of carefully diagnosed youths with BDD and focus on previously unexplored sex and age differences. We systematically collected clinical data from 172 young people with BDD consecutively referred to 2 specialist pediatric obsessive-compulsive and related disorders outpatient clinics in Stockholm, Sweden and in London, England. A series of clinician-, self-, and parent-reported measures were administered. The cohort consisted of 136 girls, 32 boys, and 4 transgender individuals (age range 10-19 years). The mean severity of BDD symptoms was in the moderate to severe range, with more than one third presenting with severe symptoms and more than half showing poor or absent insight/delusional beliefs. We observed high rates of current psychiatric comorbidity (71.5%), past or current self-harm (52.1%), suicide attempts (11.0%), current desire for cosmetic procedures (53.7%), and complete school dropout (32.4%). Compared to boys, girls had significantly more severe self-reported BDD symptoms, depression, suicidal thoughts, and self-harm. Compared to the younger participants (14 or younger), older participants had significantly more severe compulsions and were more likely to report a desire for conducting cosmetic procedures. Adolescent BDD can be a severe and disabling disorder associated with significant risks and substantial functional impairment. The clinical presentation of the disorder is largely similar across sexes and age groups, indicating the importance of early detection and treatment. More research is needed specifically focusing on boys and pre-pubertal individuals with BDD.

Effect of an Internet-Delivered Stepped-Care Program vs In-Person Cognitive Behavioral Therapy on Obsessive-Compulsive Disorder Symptoms in Children and Adolescents: A Randomized Clinical Trial.

Importance: In most countries, young people with obsessive-compulsive disorder have limited access to specialist cognitive behavioral therapy (CBT), a first-line treatment. Objective: To investigate whether internet-delivered CBT implemented in a stepped-care model is noninferior to in-person CBT for pediatric obsessive-compulsive disorder. Design, Setting and Participants: A randomized clinical noninferiority trial conducted at 2 specialist child and adolescent mental health clinics in Sweden. Participants included 152 individuals aged 8 to 17 years with obsessive-compulsive disorder. Enrollment began in October 2017 and ended in May 2019. Follow-up ended in April 2020. Interventions: Participants randomized to the stepped-care group (n = 74) received internet-delivered CBT for 16 weeks. Nonresponders at the 3-month follow-up were then offered a course of traditional face-to-face treatment. Participants randomized to the control group (n = 78) immediately received in-person CBT for 16 weeks. Nonresponders at the 3-month follow-up received additional face-to-face treatment. Main Outcomes and Measures: The primary outcome was the masked assessor-rated Children's Yale-Brown Obsessive-Compulsive Scale (CY-BOCS) score at the 6-month follow-up. The scale includes 10 items rated from 0 (no symptoms) to 4 (extreme symptoms), yielding a total score range of 0 to 40, with higher scores indicating greater severity. Assessors were masked to treatment allocation at pretreatment, posttreatment, 3-month follow-up, and 6-month follow-up assessments. The predefined noninferiority margin was 4 points on the CY-BOCS. Results: Among the 152 randomized participants (mean age, 13.4 years; 94 [62%] females), 151 (99%) completed the trial. At the 3-month follow-up, 34 participants (46%) in the stepped-care group and 23 (30%) in the in-person CBT group were nonresponders. At the 6-month follow-up, the CY-BOCS score was 11.57 points in the stepped-care group vs 10.57 points in the face-to-face treatment group, corresponding to an estimated mean difference of 0.91 points ([1-sided 97.5% CI, -∞ to 3.28]; P for noninferiority = .02). Increased anxiety (30%-36%) and depressive symptoms (20%-28%) were the most frequently reported adverse events in both groups. There were 2 unrelated serious adverse events (1 in each group). Conclusions and Relevance: Among children and adolescents with obsessive-compulsive disorder, treatment with an internet-delivered CBT program followed by in-person CBT if necessary compared with in-person CBT alone resulted in a noninferior difference in symptoms at the 6-month follow-up. Further research is needed to understand the durability and generalizability of these findings. Trial Registration: ClinicalTrials.gov Identifier: NCT03263546.

Effectiveness of Behaviour Therapy for Children and Adolescents with Tourette Syndrome and Chronic Tic Disorder in a Naturalistic Setting.

It is unclear if the results of randomised controlled trials (RCTs) of behaviour therapy (BT) for Tourette syndrome (TS) and chronic tic disorder (CTD) can be generalised to naturalistic clinical settings and are durable long-term. In this naturalistic study, 74 young people with TS/CTD received BT at a specialist clinic. Data were collected at baseline, post-treatment, and at 3-, 6-, and 12-month follow-ups. Measures included the Yale Global Tic Severity Scale (YGTSS) and the Clinical Global Impression-Improvement scale (CGI-I), amongst others. Tic severity and tic-related impairment improved after treatment, with large within-group effect sizes. At post-treatment, 57% of the participants were classified as treatment responders according to the CGI-I. Tic severity and tic-related impairment improved further through the follow-up, with 75% treatment responders at the 12-month follow-up. BT is an effective and durable treatment for young people with TS/CTD in a naturalistic specialist clinical setting, with comparable effects to RCTs.

Prevalence and impact of insomnia in children and adolescents with body dysmorphic disorder undergoing multimodal specialist treatment.

Pediatric body dysmorphic disorder (BDD) is challenging to treat. This study aimed to establish the prevalence of insomnia in youth with BDD and explore its impact on clinical outcomes. Sixty-six children and adolescents with BDD consecutively referred to a specialist clinic completed a range of clinical measures, including the Yale-Brown Obsessive-Compulsive Scale Modified for BDD-Adolescent Version (BDD-YBOCS-A), and the Insomnia Severity Index (ISI). Patients with clinical insomnia (ISI score ≥ 9) were compared to the rest of the sample on socio-demographic and clinical features. Fifty-six patients who received multimodal treatment were re-assessed post-treatment. A mixed-model ANOVA was performed to compare treatment outcomes between the insomnia vs. no insomnia groups, and Chi-squared tests were used to compare response and remission rates. According to the ISI, 48% of the sample qualified as having insomnia at baseline. These participants showed significantly higher self-reported BDD symptom severity, more depressive symptoms, and more functional impairment in daily activities. Patients with insomnia improved less on the BDD-YBOCS-A than those without insomnia, although the difference did not reach statistical significance. The rates of responders and remitters were lower in the insomnia group, compared to the non-insomnia group. Insomnia is prevalent in pediatric BDD, and is associated with more severe psychopathology and worse functioning in daily activities. Furthermore, youth experiencing BDD and insomnia may benefit from multimodal treatment to a lesser extent than those without insomnia. If these results are replicated in larger samples, treatment refinements for pediatric BDD could include specific modules to directly target insomnia.

Stepped Care Internet-Delivered vs Face-to-Face Cognitive-Behavior Therapy for Pediatric Obsessive-Compulsive Disorder: A Trial Protocol for a Randomized Noninferiority Trial.

Importance: Internet-delivered cognitive behavior therapy is an effective treatment for children and adolescents with obsessive-compulsive disorder and has the potential to markedly increase access to treatment for patients while being cost-effective for health care organizations. Objective: To investigate whether internet-delivered cognitive behavior therapy implemented within a stepped care model is noninferior to, and cost-effective compared with, the gold standard of face-to-face cognitive behavior therapy for pediatric obsessive-compulsive disorder. Design, Setting, and Participants: Multicenter, single-blind, randomized clinical noninferiority trial implemented at 2 specialist pediatric obsessive-compulsive disorder clinics in Stockholm and Gothenburg, Sweden. Participants are 152 children and adolescents aged 7 to 17 years with obsessive compulsive disorder, recruited through the 2 clinics and online self-referral. Patients will be randomized 1:1 to the stepped care intervention or face-to-face therapy. Blind evaluations will be conducted after treatment and at 3-month and 6-month follow-ups. At the 6-month follow-up (primary end point), noninferiority will be tested and resource use will be compared between the 2 treatment groups. Data will be analyzed according to intention-to-treat principles. Intervention: Patients randomized to stepped care will first receive internet-delivered cognitive behavior therapy for 16 weeks; patients who are classified as nonresponders 3 months after treatment completion will receive additional face-to-face therapy. The control group will receive 16 weeks of face-to-face cognitive behavior therapy immediately following randomization and nonresponders at the 3-month follow-up will, as in the stepped care group, receive additional face-to-face therapy. Main Outcomes and Measures: Noninferiority is defined as a 4-point difference on the primary outcome measure (Children's Yale-Brown Obsessive Compulsive Scale). Discussion: Recruitment started October 6, 2017, and was completed May 24, 2019. Results from the primary end point will be available by May 2020. The naturalistic follow-ups (1, 2, and 5 years after the end of treatment) will continue to 2025. There are no interim analyses planned or stopping rules for the trial. Trial Registration: ClinicalTrials.gov identifier: NCT03263546.

Establishing a Pediatric Acute-Onset Neuropsychiatric Syndrome Clinic: Baseline Clinical Features of the Pediatric Acute-Onset Neuropsychiatric Syndrome Cohort at Karolinska Institutet.

Objectives: Pediatric acute-onset neuropsychiatric syndrome (PANS) is a descriptive clinical entity defined by the abrupt onset of psychiatric and somatic symptoms leading to significant loss of function. Data on well-characterized PANS patients are limited, biomarkers have yet to be identified, and a solid evidence base to guide treatment is lacking. In this study, we present our experience of a systematic evaluation of the first 45 patients included in a Swedish cohort. Methods: During the period 2014-2018, our clinic received 100 referrals regarding suspected PANS. All patients underwent a standardized psychiatric/medical evaluation by a child/adolescent psychiatrist and a clinical psychologist or a nurse. Those with severe symptoms were also assessed by a pediatric neurologist and a pediatric rheumatologist. Laboratory tests were obtained at different time points in an attempt to capture an active disease state. Results: Of the 100 referrals, 45 met strict PANS criteria and consented to participate in a long-term follow-up study. The median age at intake was 7.2 years (range 3.0-13.1) and 56% were male. Ninety-three percent fulfilled both criteria for acute/atypical onset of PANS symptoms and having had an infection in relation to onset. Sixteen percent had an onset of an autoimmune or inflammatory disorder in temporal relation to the onset of PANS-related symptoms. The most common onset symptoms were obsessive-compulsive disorder (89%), anxiety (78%), and emotional lability (71%). Twenty-four percent had a preexisting autoimmune disease (AD) and 18% a preexisting psychiatric/neuropsychiatric diagnosis. Sixty-four percent of biological relatives had at least one psychiatric disorder and 76% at least one AD or inflammatory disorder. Complement activation (37%), leukopenia (20%), positive antinuclear antibodies (17%), and elevated thyroid antibodies (11%) were the most common laboratory findings. Conclusions: In our PANS cohort, there was a strong indication of an association with AD. Further work is needed to establish whether any of the potential biomarkers identified will be clinically useful. Long-term follow-up of these patients using the Swedish national registers will enable a deeper understanding of the course of this patient group.

Insomnia in pediatric obsessive-compulsive disorder: prevalence and association with multimodal treatment outcomes in a naturalistic clinical setting.

OBJECTIVE: Little is known about the prevalence and impact of insomnia on clinical outcomes in youth with obsessive-compulsive disorder (OCD). This study aimed to investigate this subject. PATIENTS/METHODS: A total of 193 patients from a specialist pediatric OCD clinic completed a range of diagnostic and clinical measures, including the Insomnia Severity Index (ISI). Patients scoring above a previously validated cut-off on the ISI (score ≥9) were compared to the rest of the sample on sociodemographic and clinical characteristics. In a subsample of 143 (from the initial 193) patients who were treated at the clinic, a mixed-model analysis of variance (ANOVA) was used to compare the outcomes of multimodal OCD treatment in the insomnia (N = 60) vs no insomnia (N = 83) groups. The primary outcome measure was the clinician-administered Children's Yale-Brown Obsessive-Compulsive Scale (CY-BOCS) at post-treatment and at three-month follow-up. RESULTS: The psychometric properties of the ISI in our sample were excellent. At baseline, 42% (81/193) of the sample scored above the ISI cut-off for clinical insomnia. These participants had significantly higher OCD severity, higher rates of psychiatric comorbidities, more severe depressive symptoms, poorer general functioning, and were more likely to take sleep medications, compared to those who scored below the ISI cut-off. In the treated subsample, while the insomnia group remained more severely affected through the three time-points, both groups improved similarly on the CY-BOCS at post-treatment and at three-month follow-up. CONCLUSION: Insomnia is relatively common in pediatric OCD and is associated with more severe psychopathology. However, with adequate multimodal, evidence-based treatment, these patients can improve as much as those without insomnia.