A systematic review of the psychometric properties of tools for measuring depression in youths with intellectual disability.

While youths with intellectual disability (ID) have increased vulnerability for depressive disorders, cognitive problems and combined functional barriers make them less prone to receive adequate treatments. A systematic review of the literature was conducted (PROSPERO Registration number: CRD42022347703) based on several databases from 1980 to 2022 to examine the quality of tools for measuring depression in children and adolescents with ID. The COSMIN (COnsensus-based Standards for the selection of health status Measurement Instruments) checklist was used to assess several psychometric domains. Twelve studies evaluated the properties of six tools for measuring depression in youths with ID. The Center for Epidemiologic Studies Depression Scale-Intellectual Disability (CESD-ID) was the only scale with at least five domains of psychometric properties assessed to have strong or moderate evidence. Based on the reviewed findings, tools specifically developed for populations with developmental disabilities should be considered first in order to screen depression in youths with ID. Much work is required to confirm their validity in clinical samples with patients with a complex form of developmental disabilities. As a complement to self- and caregivers-report questionnaires, clinician rating scales were considered useful to catch the full picture of depression in youths with ID, in particular associated behavioral expressions. Their validity received little scrutiny and certainly deserve more attention to improve care practice of youths with ID.

Emotions and behaviours of child and adolescent psychiatric patients during the COVID-19 pandemic.

BACKGROUND: Previous pandemics have had negative effects on mental health, but there are few data on children and adolescents who were receiving ongoing psychiatric treatment. AIMS: To study changes in emotions and clinical state, and their predictors, during the COVID-19 pandemic in France. METHOD: We administered (by interview) the baseline Youth Self-Report version of the CoRonavIruS Health Impact Survey v0.3 (CRISIS, French translation) to 123 adolescent patients and the Parent/Caregiver version to evaluate 99 child patients before and during the first 'lockdown'. For 139 of these patients who received ongoing treatment in our centre, treating physicians retrospectively completed longitudinal global ratings for five time periods, masked to CRISIS ratings. RESULTS: The main outcome measure was the sum of eight mood state items, which formed a single factor in each age group. Overall, this score improved for each age group during the first lockdown. Clinician ratings modestly supported this result in patients without intellectual disability or autism spectrum disorder. Improvement of mood states was significantly associated with perceived improvement in family relationships in both age groups. CONCLUSIONS: Consistent with previous studies of clinical cohorts, our patients had diverse responses during the pandemic. Several factors may have contributed to the finding of improvement in some individuals during the first lockdown, including the degree of family support or conflict, stress reduction owing to isolation, limitations of the outcome measures and/or possible selection bias. Ongoing treatment may have had a protective effect. Clinically, during crises additional support may be needed by families who experience increased conflict or who care for children with intellectual disability.

Hard-to-treat or hard-to-catch? Clinical features and therapeutic outcomes of help-seeking foster care youths with mood disorders.

Introduction: The high level of emotional problems in youths placed in foster care contrasts with the limited use of evidence-based treatments. This study aims to better characterize the clinical features and therapeutic outcomes of foster care youths with mood disorders. Methods: A secondary analysis of data collected in the context of a French-Canadian clinical research network on pediatric mood disorders in four sites was conducted to compare three groups of patients with depressive or bipolar disorder: those without exposure to child welfare intervention (WCWI, n = 181), those who received non-placement psychosocial intervention (NPI, n = 62), and those in placement interventions (PI, n = 41). Results: We observed a very high rate of academic problems in patients in the groups NPI/PI compared to those in the WCWI group. Patients in the PI group had more disruptive behavioral disorders (OR = 6.87, 95% CI [3.25-14.52]), trauma-related disorders (OR = 3.78, 95% CI [1.6-8.94]), and any neurodevelopmental disorders (OR = 2.73, 95% CI [1.36-5.49]) compared to the other groups (NPI/WCWI). Among inpatients, the Clinical Global Impression-Improvement scale and the change in the Children Global Assessment Scale during the hospital stay did not differ across the three groups. We observed a higher prescription rate of antipsychotics in the PI group compared to the NPI/WCWI groups, but no significant difference for antidepressants and mood stabilizers. Discussion: These findings support the view that, when provided with dedicated support, fostered inpatient youths can improve in a range comparable to other inpatients. Undetected neurodevelopmental disorders and academic problems are likely important contributors of the burden of mood disorders in these youths.

Corrigendum: Prader-Willi syndrome: Symptoms and topiramate response in light of genetics.

[This corrects the article DOI: 10.3389/fnins.2023.1126970.].

Down syndrome regression disorder, a case series: Clinical characterization and therapeutic approaches.

Down syndrome (DS) is one of the most frequent genetic disorders and represents the first cause of intellectual disability of genetic origin. While the majority of patients with DS follow a harmonious evolution, an unusual neurodevelopmental regression may occur, distinct from that described in the context of autism spectrum disorders, called down syndrome regression disorder (DSRD). Based on four patients, two males and two females, with age range between 20 and 24, treated at the Reference Center for Rare Psychiatric Disorders of the GHU Paris Psychiatry and Neurosciences [Pôle hospitalo-universitaire d'Évaluation Prévention et Innovation Thérapeutique (PEPIT)], we describe this syndrome, discuss its etiologies and propose therapeutic strategies. DSRD often occurs in late adolescence. There is a sudden onset of language disorders, loss of autonomy and daily living skills, as well as behavioral symptoms such as depression, psychosis, or catatonia. These symptoms are non-specific and lead to an overlap with other diagnostic categories, thus complicating diagnosis. The etiologies of the syndrome are not clearly identified but certain predispositions of patients with trisomy 21 have suggested an underlying immune-mediated mechanism. Symptomatic therapeutic approaches (serotonergic antidepressants, atypical antipsychotics, benzodiazepines) were not effective, and generally associated with poor tolerance. Etiological treatments, including anti-inflammatory drugs and corticosteroids, led to partial or good recovery in the four cases. Early recognition of regressive symptoms and rapid implementation of adapted treatments are required to improve the quality of life of patients and their families.

Prader-Willi syndrome: Symptoms and topiramate response in light of genetics.

Introduction: Prader-Willi Syndrome (PWS) is a rare genetic condition, which affects one in 25,000 births and results in various phenotypes. It leads to a wide range of metabolic and endocrine disorders including growth delay, hypogonadism, narcolepsy, lack of satiety and compulsive eating, associated with mild to moderate cognitive impairment. Prognosis is especially determined by the complications of obesity (diabetes, cardiorespiratory diseases) and by severe behavioral disorders marked by impulsivity and compulsion. This heterogeneous clinical picture may lead to mis- or delayed diagnosis of comorbidities. Moreover, when diagnosis is made, treatment remains limited, with high interindividual differences in drug response. This may be due to the underlying genetic variability of the syndrome, which can involve several different genetic mutations, notably deletion or uniparental disomy (UPD) in a region of chromosome 15. Here, we propose to determine whether subjects with PWS differ for clinical phenotype and treatment response depending on the underlying genetic anomaly. Methods: We retrospectively included all 24 PWS patients who were referred to the Reference Center for Rare Psychiatric Disorders (GHU Paris Psychiatrie and Neurosciences) between November 2018 and July 2022, with either deletion (N = 8) or disomy (N = 16). The following socio-demographic and clinical characteristics were recorded: age, sex, psychiatric and non-psychiatric symptoms, the type of genetic defect, medication and treatment response to topiramate, which was evaluated in terms of eating compulsions and impulsive behaviors. We compared topiramate treatment doses and responses between PWS with deletion and those with disomy. Non-parametric tests were used with random permutations for p-value and bootstrap 95% confidence interval computations. Results: First, we found that disomy was associated with a more severe clinical phenotype than deletion. Second, we observed that topiramate was less effective and less tolerated in disomy, compared to deletion. Discussion: These results suggest that a pharmacogenomic-based approach may be relevant for the treatment of compulsions in PWS, thus highlighting the importance of personalized medicine for such complex heterogeneous disorders.

Relevance of Brain 18F-FDG PET Imaging in Probable Seronegative Encephalitis With Catatonia: A Case Report.

Autoimmune encephalitis (AIE) is a rare, severe, and rapidly progressive encephalopathy, and its diagnosis is challenging, especially in adolescent populations when the presentation is mainly psychiatric. Currently, cerebral 18-fluorodeoxyglucose positron emission tomography (18F-FDG-PET) imaging is not included in the diagnosis algorithm. We describe a 16-year-old patient with probable seronegative encephalitis with catatonia for which several cerebral PET scans were relevant and helpful for diagnosis, treatment decision making, and follow-up monitoring. The patient recovered after 2 years of treatment with etiologic treatment of AIE and treatment of catatonia. This case suggests a more systematic assessment of the clinical relevance of 18F-FDG-PET imaging in probable seronegative AIE.

Missing the forest for the trees? A high rate of motor and language impairments in Disruptive Mood Dysregulation Disorder in a chart review of inpatient adolescents.

Youths with severe and persistent irritability have a particularly high rate of school failures and learning difficulties. The aim of this study was to determine whether inpatient adolescents with Disruptive Mood Dysregulation Disorder (DMDD) have more motor and/or language impairments compared to patients with other psychiatric disorders. A retrospective chart review of all consecutive cases admitted in two adolescent inpatient units between January 2017 and December 2018 was conducted (N = 191). All patients received multi-disciplinary clinical and developmental assessments. For a subtest of subjects, additional standardized tests were used to document motor and language impairments. In this clinical chart 53 adolescents with a DMDD (mean age 13.6 ± 1.5, min 12, max 16, 70% males) were compared to patients with a major depressive disorder (MDD, n = 64, mean age 15.3 ± 1.6, 52% males) and patients with a non-mood disorder (NMD, n = 61, mean age 14.4 ± 1.55, 59% males). Among inpatients with DMDD, 71% had an associated motor and/or language disorder, with combined forms in around two-thirds of cases. Compared to youths with MDD, participants with DMDD were more likely to have an associated developmental coordination disorder (67% vs. 22%, OR = 4.7) and a written language disorder (35% vs. 10%, OR = 4.6). While 31% of inpatients with DMDD had an associated communication/oral language disorder, this rate was not statistically different from those observed in the MDD group (11%, OR = 3.2). The frequencies of motor and language impairments were not statistically different between participants in the DMDD group and in the NMD group. The high rate of motor and written language disorders found in DMDD patients may partly account for their academic difficulties. Such finding, if confirmed, supports systematic screening of motor and written language impairments in youths with chronic irritability and suggests remediation potential.

Psychiatric autoimmune conditions in children and adolescents: Is catatonia a severity marker?

OBJECTIVES: Patients with autoimmune encephalitis (AE) are likely to exhibit an acute onset of severe psychiatric features, including psychosis and/or catatonia. Based on the high prevalence of catatonia in AE and our clinical experience, we hypothesized that catatonia might be a marker of severity requiring more aggressive treatment approaches. METHODS: To reach a sufficient number of cases with brain-autoimmune conditions, we pooled two samples (N = 58): the first from the French National Network of Rare Psychiatric diseases and the second from the largest Italian neuro-pediatrics center for encephalopathies. Autoimmune conditions were diagnosed using a multidisciplinary approach and numerous paraclinical investigations. We retrospectively compared patients with and without catatonia for psychiatric and non-psychiatric clinical features, biological and imaging assessments, type of immunotherapy used and outcomes. RESULTS: The sample included 25 patients (43%) with catatonia and 33 (57%) without catatonia. Forty-two patients (72.4%) had a definite AE (including 27 anti-NMDA receptor encephalitis) and 16 (27.6%) suspected autoimmune encephalitis. Patients with catatonia showed significantly more psychotic features [18 (72%) vs 9 (27.3%), p < 0.001)] and more movement disorders [25 (100%) vs 20 (60.6%), p < 0.001] than patients without catatonia. First line (corticoids, immunoglobulin and plasma exchanges) and second line (e.g., rituximab) therapies were more effective in patients with catatonia, with 24 (96%) vs 22 (66.7%) (p = 0.006) and 17 (68%) vs 9 (27.3%) (p = 0.002), respectively. However, those with catatonia received more combinations of first and second line treatments and had more relapses during outcomes. CONCLUSION: Despite its exploratory design, the study supports the idea that autoimmune catatonia may be a marker of severity and morbidity in terms of initial presentation and relapses, requiring the need for early and aggressive treatment.

Borderline personality disorder and prior suicide attempts define a severity gradient among hospitalized adolescent suicide attempters.

BACKGROUND: Borderline personality disorder (BPD) and history of prior suicide attempt (SA) have been shown to be high predictors for subsequent suicide. However, no previous study has examined how both factors interact to modify clinical and suicide severity among adolescents. METHODS: This study presents a comprehensive assessment of 302 adolescents (265 girls, mean age = 14.7 years) hospitalized after a SA. To test clinical interactions between BPD and history of prior SA, the sample was divided into single attempters without BPD (non-BPD-SA, N = 80), single attempters with BPD (BPD-SA, N = 127) and multiple attempters with BPD (BPD-MA, N = 95). RESULTS: Univariate analyses revealed a severity gradient among the 3 groups with an additive effect of BPD on the clinical and suicide severity already conferred by a history of SA. This gradient encompassed categorical (anxiety and conduct disorders and non-suicidal-self-injury [NSSI]) and dimensional comorbidities (substance use and depression severity) and suicide characteristics (age at first SA). According to regression analyses, the BPD-MA group that was associated with the most severe clinical presentation also showed specific features: the first SA at a younger age and a higher prevalence of non-suicidal self-injury (NSSI) and anxiety disorders. The BPD-MA group was not associated with higher impulsivity or frequency of negative life events. CONCLUSIONS: Based on these findings and to improve youth suicide prevention, future studies should systematically consider BPD and the efficacy of reinforcing early interventions for anxiety disorders and NSSI.

[Antidepressant medication in children]. / Quelle prescription d'antidépresseurs chez l'enfant ?

When should we use antidepressant medications in children? Antidepressant medication may not be considered as a first-line treatment in children; psychotherapeutic treatments should always be preferentially used. At this age, the efficacy of SSRI is regarded as low to moderate for depression, but moderate to high for Obsessive Compulsive Disorder (OCD) and anxiety disorders. When an antidepressant medication is prescribed, a SSRI should always be used first. In particular, fluoxetine is the most studied SSRI and the only medication who received approval by the French regulatory authority. Sertraline and fluvoxamine which have been approved for OCD should preferentially be used for that purpose. During the first 4 weeks, clinicians should actively monitor the onset of side effects, especially mood swings and suicidal behavior. The onset or increase of suicidal thoughts during SSRI treatment would concern about 1 out of 100 young patients treated. This risk is maximal during the first four weeks following the introduction of the SSRI and should progressively decrease after one month. When used in children, antidepressant medication can only be used in association with psychotherapeutic treatments and psychosocial interventions targeting the maintaining factors perpetuating the cycle of affective symptoms.

Quelle prescription d'antidépresseurs chez l'enfant ? Les antidépresseurs ne sont jamais un traitement de première intention chez l'enfant et l'adolescent. La balance bénéfice-risque varie selon les indications : faible à modérée pour la dépression, mais modérée à élevée pour les troubles obsessionnels compulsifs (TOC) et les troubles anxieux. Quand un traitement est prescrit, les inhibiteurs sélectifs de la recapture de la sérotonine (ISRS) sont toujours à privilégier. En particulier la fluoxétine est le traitement dont le rapport bénéfice-risque est le plus favorable dans la dépression, et c'est la seule molécule disposant d'une autorisation de mise sur le marché (AMM). La sertraline et la fluvoxamine sont utilisées préférentiellement pour les TOC, pour lesquels elles ont une AMM. Les modalités de prescription des ISRS chez l'enfant sont globalement similaires à l'adulte. Deux effets indésirables nécessitent une surveillance hebdomadaire les 4 premières semaines : le risque de virage de l'humeur et de conduites suicidaires. L'apparition ou l'augmentation d'idées suicidaires en début de traitement concerne environ 1 jeune traité sur 100. Ce risque est surtout élevé dans les 4 semaines qui suivent l'introduction, alors que la réduction du risque suicidaire sous ISRS apparaît généralement après un mois de traitement. D'une façon générale, un traitement antidépresseur ne peut s'envisager que dans le cadre d'une prise en charge associant des propositions psychothérapeutiques adaptées au niveau de développement et des interventions familiales et psychosociales ciblant les facteurs de pérennisation des symptômes anxieux et/ou dépressifs.

Suicide Attempts Among French and Brazilian Adolescents Admitted to an Emergency Room. A Comparative Study of Risk and Protective Factors.

BACKGROUND: Suicide is the second most common cause of preventable mortality among Brazilian and French adolescents. The aim of the current study was to compare the main risk and protective factors associated with a suicide attempt (SA) and to highlight differences based on geographical characteristics. METHOD: We compared a Brazilian sample (N = 45) of adolescents admitted to the emergency room of a public hospital in São Paulo for SA to a French sample (N = 320) of adolescents hospitalized for SA across 5 paediatric departments. Then, we ran several multivariate models to examine how each selected variable was related to geographic origin and to the other selected variables linked to geographic origin. RESULTS: The two samples presented no significant differences regarding gender, age or schooling. Both samples had high rates of depressive disorders, anxiety disorders, substance use, disruptive disorders, borderline psychopathology, and lifetime SAs. However, the Brazilian sample presented significantly higher levels of psychopathology and had more insecure attachment relationships (fearful and detached), whereas the French sample had a more secure attachment style. Brazilian adolescents had more recourse to spiritual beliefs and spiritual support, whereas the French adolescents had higher scores on the Reasons for Living Inventory and used more help-seeking strategies from their social network, mainly close friends. Multivariate models showed that two productive coping strategies (seeking spiritual support and social action) and the dependence score were significantly associated with membership in the Brazilian cohort, whereas a secure attachment style and depression severity (evaluated by the Beck Depression Inventory) were significantly associated with membership in the French cohort. CONCLUSION: Despite presenting similar psychopathologies, Brazilian adolescents presented a more insecure attachment style and used the religious kind of coping more commonly than their French counterparts. We hypothesize that religion may compensate for the social vulnerabilities present in a middle-income country such as Brazil. More transcultural studies may help to elucidate this phenomenon.

High diagnostic value of plasma Niemann-Pick type C biomarkers in adults with selected neurological and/or psychiatric disorders.

Late-onset Niemann-Pick type C (NP-C) is a rare, underdiagnosed lysosomal disease with neurological manifestations. A specific treatment, miglustat, can stabilize the disease if given early. Recently, three plasma screening biomarkers (PSBs) were developed [cholestane3ß,5α,6ßtriol (C-triol), 7-ketocholesterol (7-KC), and lysosphingomyelin-509 (LSM-509)], allowing a simpler and quite robust screening of patients suitable for genetic testing. The objective of our study was to evaluate practical utility and feasibility of large-scale PSB screening for NP-C in selected adult patients. Patients were prospectively enrolled if they showed, starting from 12 years of age, at least one of the three initial neuro-psychiatric manifestations described in NP-C: (1) gait disorder (cerebellar and/or dystonic); (2) cognitive decline with frontal lobe syndrome; (3) atypical psychosis. PSBs were measured in plasma of all patients and, if positive (LSM-509 and/or C-triol + 7-KC elevated), sequencing of NPC1 and NPC2 genes was performed. A total of 251 patients [136 males, 115 females; median age 42.1 (range 12.2-85.6) years] were screened. Six patients had positive PSBs. Two were confirmed to have NP-C (0.8% diagnostic yield, both with all three PSBs highly increased, especially LSM-509). False-positive rate was 1.2%, which was identical if only considering LSM-509. By contrast, false-positive rates were 8.1% and 5.7% for 7-KC and C-triol, respectively. We showed that selecting patients with neurologic and/or psychiatric symptoms consistent with NP-C for large-scale PSB screening is a simple and valid strategy to identify new adult NP-C patients, and would probably lead to earlier diagnosis and treatment administration if widely applied.

Repeating a suicide attempt during adolescence: risk and protective factors 12 months after hospitalization.

Suicide attempts (SAs) are a public health concern in adolescence. A brief hospitalization is recommended, but access to inpatient wards is often not available. In addition, numerous risk factors for SA recurrence have been identified, but few studies have explored protective factors. Here, we aimed to assess the role of both risk and protective factors on SA relapse in a context of free access to inpatient services. We performed a prospective follow-up study of 320 adolescents who were hospitalized for an SA between January 2011 and December 2014 in France. Assessments at baseline included socio-demographics, clinical characteristics, temperament, reasons for living, spirituality, and coping. Patients were re-evaluated at 6 months and 12 months for depression severity and SA relapse. A total of 135 and 91 patients (78 girls, 12 boys, aged 13-17) were followed up at 6 and 12 months, respectively. At the 12-month follow-up, 28 (30%) subjects had repeated an SA. Adolescents who either had a history of SA or were receiving psychotropic treatment at baseline were at higher risk of recurrence. Several variables had a protective effect: (1) productive coping skills, namely, working hard and achieving, physical recreation, and seeking relaxing diversions; (2) a particular temperament trait, namely, cooperativeness; and (3) having experienced more life events. We also found a significant interaction: the higher the depression score during follow-up, the lower the protective effect of productive coping. Our findings confirm that a history of SA and seeking psychiatric care with medication are risk factors for SA relapse. However, productive coping strategies and cooperativeness are protective factors, and the improvement of such strategies as well as treatment of persisting depression should be a goal of psychotherapy treatment offered to suicidal adolescents.