Adaptive, behavioral, and emotional outcomes following postoperative pediatric cerebellar mutism syndrome in survivors treated for medulloblastoma.

OBJECTIVE: Patients who experience postoperative pediatric cerebellar mutism syndrome (CMS) during treatment for medulloblastoma have long-term deficits in neurocognitive functioning; however, the consequences on functional or adaptive outcomes are unknown. The purpose of the present study was to compare adaptive, behavioral, and emotional functioning between survivors with and those without a history of CMS. METHODS: The authors examined outcomes in 45 survivors (15 with CMS and 30 without CMS). Comprehensive neuropsychological evaluations, which included parent-report measures of adaptive, behavioral, and emotional functioning, were completed at a median of 2.90 years following craniospinal irradiation. RESULTS: Adaptive functioning was significantly worse in the CMS group for practical and general adaptive skills compared with the group without CMS. Rates of impairment in practical, conceptual, and general adaptive skills in the CMS group exceeded expected rates in the general population. Despite having lower overall intellectual functioning, working memory, and processing speed, IQ and related cognitive processes were uncorrelated with adaptive outcomes in the CMS group. No significant group differences or increased rates of impairment were observed for behavioral and emotional outcomes. CONCLUSIONS: Survivors with CMS, compared with those without CMS, are rated as having significant deficits in overall or general adaptive functioning, with specific weakness in practical skills several years posttreatment. Findings from this study demonstrate the high risk for ongoing functional deficits despite acute recovery from symptoms of CMS, highlighting the need for intervention to mitigate such risk.

Phasic Versus Tonic Irritability and Associations with Family Accommodation Among Youth with Selective Mutism: A Latent Profile Analysis.

Clinical presentations of selective mutism (SM) vary widely across affected youth. Although studies have explored general externalizing problems in youth with SM, research has not specifically examined patterns of irritability. Relatedly, research has not considered how affected families differentially accommodate the anxiety of youth with SM as a function of the child's temper outbursts (i.e., phasic irritability) and general angry mood (i.e., tonic irritability). Data were drawn from a sample of treatment-seeking children and adolescents with a primary diagnosis of selective mutism (N = 152; Mean age = 6.12 years; 67.11% female), and their caregivers. Latent profile analysis (LPA) was used to identify distinct profiles in SM youth that were characterized by varying levels of phasic and/or tonic irritability. Analyses further examined whether these different profiles were associated with different levels of family accommodation and global impairment. LPA identified 5 profiles: SM with No irritability, SM with Low Phasic Irritability, SM with High Phasic Irritability, SM with High Phasic and Moderate Tonic Irritability, and SM with High Phasic and High Tonic Irritability. Patterns of family accommodation and global impairment were highest among youth belonging to profiles characterized by high phasic irritability. Findings highlight separable patterns of irritability across youth with SM, with phasic irritability (i.e., temper outbursts) appearing particularly linked with increased family accommodation and overall global impairment. Assessing phasic irritability is critical for optimizing treatment in youth with SM and can be useful for flagging possible patterns of family accommodation contributing to overall impairment.

Long-term outcomes of selective mutism: a systematic literature review.

BACKGROUND: Selective mutism (SM) is a childhood onset anxiety disorder, and the main symptom is not speaking in certain social situations. Knowledge about the duration and long-term outcomes of SM have been lacking and the aim of this systematic literature review was to address this gap in the literature. We investigated how long SM symptoms persisted as well as other psychiatric outcomes associated with SM in later life. METHODS: The PubMed, PsycInfo, Web of Science, Cochrane Library and Embase databases were initially searched from inception to 11 September 2023. Studies were included if they were published in English and had followed up subjects with clinically diagnosed SM for at least two years. The review followed the Preferred Reporting Items of Systematic Reviews and Meta-analyses guidelines and the protocol was registered with the Open Science Framework. The papers were assessed using the Quality Assessment with Diverse Studies tool. RESULTS: This review screened 2,432 papers and assessed 18 studies. Seven case series studies were excluded from discussion because of the low number of subjects and the fact that their findings could not be generalized to wider populations. In the end, nine clinical cohorts and two case control studies were reviewed. These provided a total of 292 subjects and the sample sizes ranged from 11-49. The overall quality of the studies was moderate. The review found that 190 of the 243 subjects in the studies that reported recovery rates showed moderate or total improvement from SM during follow up. Other anxiety disorders were the most common psychiatric disorders later in life, although these results should be interpreted with caution. Older age at baseline and parental psychopathology might predict greater impairment, but further studies are needed to confirm these results. CONCLUSIONS: Most subjects with SM recovered from this disorder during adolescence, but anxiety disorders were common in later life. Early detection and treatment are needed to prevent symptoms from persisting and other psychiatric disorders from developing.

Beyond the language barrier: A systematic review of selective mutism in culturally and linguistically diverse children.

Selective mutism is a childhood psychiatric disorder that has been associated with adverse psychological, social and educational outcomes. Although evidence suggests that culturally and linguistically diverse children might be overrepresented among children with selective mutism, a direct examination of how migration or minority status are associated with the development and persistence of the disorder is still scarce. Guided by eco-cultural perspectives of development, the current review aims to provide an overview of selective mutism in culturally and linguistically diverse children. A systematic literature review of selective mutism studies that included a group of culturally and linguistically diverse children yielded eight studies that met our inclusion criteria. Although these studies support the view that bilingualism and minority status might be associated with selective mutism, the role of sociocultural factors in the development and persistence of the disorder remained mostly unexamined. The review concludes with a discussion of potential directions for future research, including examination of the cultural and psychological meanings of silence and talk, socialization goals, gender inequality, and parental acculturation strategies.

Diagnosing selective mutism: a critical review of measures for clinical practice and research.

Selective mutism (SM) is an anxiety disorder (prevalence 1-2%), characterized by the consistent absence of speaking in specific situations (e.g., in school), while adequately speaking in other situations (e.g., at home). SM can have a debilitating impact on the psychosocial and academic functioning in childhood. The use of psychometrically sound and cross-culturally valid instruments is urgently needed.The aim of this paper is to identify and review the available assessment instruments for screening or diagnosing the core SM symptomatology. We conducted a systematic search in 6 databases. We identified 1469 studies from the last decade and investigated the measures having been used in a diagnostic assessment of SM. Studies were included if original data on the assessment or treatment of SM were reported. It was found that 38% of published studies on SM reporting original data did not report the use of any standardized or objective measure to investigate the core symptomatology. The results showed that many different questionnaires, interviews and observational instruments were used, many of these only once. The Selective Mutism Questionnaire (SMQ), Anxiety Disorders Interview Schedule (ADIS) and School Speech Questionnaire (SSQ) were used most often. Psychometric data on these instruments are emerging. Beyond these commonly used instruments, more recent developed instruments, such as the Frankfurt Scale of SM (FSSM) and the Teacher Telephone Interview for SM (TTI-SM), are described, as well as several interesting observational measures. The strengths and weaknesses of the instruments are discussed and recommendations are made for their use in clinical practice and research.

Emotion regulation in selective mutism: A comparison group study in children and adolescents with selective mutism.

A deficit in emotion regulation skills could be an important factor influencing the vulnerability and maintenance of symptoms in selective mutism (SM). Unfortunately, to date only a few studies have examined emotion regulation in SM. Therefore, the present study investigated whether SM is associated with dysfunctional emotion regulation strategies. We compared a sample of 28 children and adolescents with SM (M = 12.66 years, SD = 3.98; 18 females) to 33 controls without SM (M = 12.45 years, SD = 3.18; 21 females). Both groups were investigated for the assessment of SM, social anxiety and emotion regulation using self and parent report questionnaires. We assumed that the disorder is associated with less adaptive and more maladaptive strategies, especially maladaptive cognitive strategies. Instead of significant differences in these overall values, only significant differences in individual emotion regulation strategies were found. In terms of adaptive strategies, children and adolescents with SM reported less problem-oriented behaviour and less cognitive problem-solving. Instead, they reported the maladaptive strategy of abandonment more often than the control group. In contrast to other anxiety disorders, children and adolescents with SM did neither report maladaptive cognitive strategies nor seek support more frequently than the control group. Their emotion regulation strategies are qualitatively closely related to the symptoms of SM, which makes it difficult to determine their independent significance. Trial registration: This study is registered with the ClinicalTrials.gov number NCT04233905.

Neuropsychological Profile in Children with Posterior Fossa Tumors with or Without Postoperative Cerebellar Mutism Syndrome (CMS).

Cerebellar mutism syndrome (CMS) is a common surgical sequela in children following posterior fossa tumor (PFT) resection. Here, we analyze the neuropsychological features associated with PFT in children, focusing particularly on the differential profiles associated with the presence or absence of CMS after surgery. We further examine the effect of post-resection treatments, tumor type, and presence/absence of hydrocephalus on surgical outcome. Thirty-six patients diagnosed with PFT (19 with and 17 without CMS) and 34 age- and gender-matched healthy controls (HCs) were recruited. A comprehensive neuropsychological evaluation was conducted in all patients postoperatively and in HCs, including an assessment of general cognitive ability, motor skills, perception, language, memory, attention, executive functions, and academic competence. CMS was found to be a clinical marker of lower neuropsychological profile scores across all cognitive domains except auditory-verbal processing and visual memory tasks. PFT patients not presenting CMS exhibited milder impairment in intellectual functioning, motor tasks, reasoning, language, verbal learning and recall, attention, cognitive executive functions, and academic competence. High-grade tumors were associated with slower processing speed and verbal delayed recall as well as alterations in selective and sustained attention. Hydrocephalus was detrimental to motor functioning and nonverbal reasoning. Patients who had undergone surgery, chemotherapy, and radiotherapy presented impaired processing speed, verbal learning, and reading. In addition to the deleterious effects of PFT, post-resection PFT treatments have a negative cognitive impact. These undesired consequences and the associated tumor-related damage can be assessed using standardized, long-term neuropsychological evaluation when planning rehabilitation.

Variations in Parent and Teacher Ratings of Internalizing, Externalizing, Adaptive Skills, and Behavioral Symptoms in Children with Selective Mutism.

Selective mutism (SM) is an anxiety disorder that impacts communication. Children with SM present concerns to parents and teachers as they consistently do not speak in situations where there is an expectation to speak, such as at school, but speak in other settings where they feel more comfortable, such as at home. The purpose of this study was to investigate the relationship between parents' and teachers' perceptions of children with SM on behavioral rating scales and language measures. Forty-two children (22 boys and 20 girls, ranging from 2.4 to 13.8 years, with a mean age of 7.1 years) took part in this study. Parents and teachers completed the Behavior Assessment System for Children (BASC-3) measuring internalizing behaviors, externalizing behaviors, adaptive skills, and behavioral symptoms. Frequency of speaking and language abilities were also measured. Parents and teachers both identified withdrawal as the most prominent feature of SM but parents saw children as significantly more withdrawn than did their teachers. Both rated children similarly at-risk on scales of functional communication and social skills. Higher adaptive skills (including functional communication and social skills) were positively correlated with vocabulary, narrative language, and auditory serial memory according to teachers. Parent and teacher rating scales provide valuable information for diagnosis and progress monitoring. Children with SM can benefit from mental health practitioners who can identify and enhance their emotional well-being.

Examining a novel, parent child interaction therapy-informed, behavioral treatment of selective mutism.

BACKGROUND: The purpose of this study was to evaluate a new therapy for children with selective mutism (SM) that combines Parent-Child Interaction Therapy principles and behavioral techniques. METHOD: Children aged 4-10 with a primary diagnosis of SM were eligible to participate. Comorbidity was allowed with the exception of autism spectrum disorder, intellectual disability, mania or psychosis. Of 54 potentially eligible participants, 33 met inclusion/exclusion criteria of which 31 families consented (94%). Following assessment, children were waitlisted for an average of 4 months before receiving 16 sessions of weekly therapy at an outpatient psychiatry clinic of a children's hospital in Vancouver, Canada; all children completed treatment. Assessments were conducted at time of referral (baseline), pre-treatment, post-treatment, 3 month follow up, and 1 year follow up. Two did not complete follow up assessments (93% retention). RESULTS: Results showed significant and large (Cohen's d = 1.80) gains in speaking behaviors across contexts from pre- to post-treatment. Gains were maintained at 3-months and 1-year post-treatment. Statistically significant and large improvements were also found in post-treatment teacher and parent reports of child anxiety as well as a behavioral measure of the child speaking to an unknown adult. Parents reported high satisfaction with treatment. None of the potential predictors of treatment response examined were found to be significant. CONCLUSIONS: PCIT-SM appears to be an effective treatment for children aged 4-10 with SM.

Intensive group behavioral treatment (IGBT) for children with selective mutism: A preliminary randomized clinical trial.

OBJECTIVE: Very few controlled trials have evaluated targeted treatment methods for childhood selective mutism (SM); the availability of evidence-based services remains limited. This study is the first controlled trial to evaluate an intensive group behavioral treatment (IGBT) for children with SM. METHOD: Twenty-nine children with SM (5-9 years; 76% female; 35% ethnic minority) were randomized to immediate SM 5-day IGBT or to a 4-week waitlist with psychoeducational resources (WLP), and were assessed at Week 4 and again 8 weeks into the following school year. RESULTS: IGBT was associated with high satisfaction and low perceived barriers to treatment participation. At Week 4, 50% of the immediate IGBT condition and 0% of the WLP condition were classified as "clinical responders." Further, Time × Condition interactions were significant for social anxiety severity, verbal behavior in social situations, and global functioning (but not for SM severity, verbal behavior in home settings, or overall anxiety). School-year follow-up assessments revealed significant improvements across all outcomes. Eight weeks into the following school year, 46% of IGBT-treated children were free of an SM diagnosis. In addition, teachers in the post-IGBT school year rated less school impairment and more classroom verbal behavior relative to teachers in the pre-IGBT school year. CONCLUSIONS: Findings provide the first empirical support for the efficacy and acceptability of IGBT for SM. Further study is needed to examine mechanisms of IGBT response, and other effective SM treatment methods, in order to clarify which treatment formats work best for which affected children. (PsycINFO Database Record (c) 2019 APA, all rights reserved).

The multiple faces of catatonia in autism spectrum disorders: descriptive clinical experience of 22 patients over 12 years.

A retrospective review was conducted from the inpatient and outpatient records of twenty-two autistic youth presenting to a neurobehavioral service over a twelve-year period for combined psychiatric and behavioral pathology who also met DSM5 criteria for catatonia. Six autistic girls and 16 autistic boys ranging from ages eight to 26 years old were identified, and their variegated symptoms evaluated. Stereotypy, posturing, negativism, mutism and stupor were the most common catatonic symptoms, each present in more than half of the study patients. One patient had abnormal vital signs indicative of malignant catatonia. Twenty patients had concomitant repetitive self-injurious behaviors that had led to significant tissue injury and were refractory to psychotropic and behavioral interventions. The sample was weighted towards patients with severe self-injurious behavior, which often was the reason for admission. The many "faces" of catatonia in autism spectrum disorders are seen in this sample, and the novel recognition of repetitive self-injury as an under-recognized motor symptom of catatonia is highlighted. The preliminary findings in this study open many important future vistas for ongoing research regarding catatonia in ASDs.

A Two-Session Hierarchy for Shaping Successive Approximations of Speech in Selective Mutism: Pilot Study of Mobile Apps and Mechanisms of Behavior Change.

Selective mutism (SM) is an anxiety disorder marked by withdrawal of speech in particular social situations. Treatment is often difficult, requiring attention to several characteristics particular to the disorder. Therapeutic tools and activities such as games and mobile applications (apps) may be particularly advantageous to behavioral therapy for SM. A 2-session hierarchy for shaping successive approximations of speech in SM was piloted with 15 children, 5 to 17 years old, who were randomly assigned to shaping while using mobile apps, other therapeutic tools/activities, and reinforcement alone. Very strong treatment gains were observed: 13 of 15 (88.7%) children completed the hierarchy during the first session and 14 (93.3%) did so during the second session, with the final child completing all but the final step (i.e., to ask and respond to at least 5 open-ended questions). Moreover, all 15 children spoke to the clinician within 59 minutes of treatment (M = 17 minutes), and 14 (93.3%) children held five, 5-minute conversations with additional unknown adults during the second session. This occurred regardless of the inclusion of therapeutic tools/activities, although preliminary patterns of responding were observed such that children shaped while using mobile apps tended to show less self-reported and physiologically measured anxious distress. The utility of therapeutic activities and mobile apps when treating SM is discussed as well as areas for future research.

Effects of anxiety, language skills, and cultural adaptation on the development of selective mutism.

Although bilingual children are thought to be at higher risk for selective mutism (SM), little is known about the development of SM in this population. This study investigates the effects of children's anxiety and language skills and parents' cultural adaptation on the development of SM. 15 bilingual (11 mute, 4 speaking at the beginning of the study) and 15 monolingual children (7 mute, 8 speaking at the beginning of the study) between the ages of 3 years and 5 years 8 months were assessed longitudinally over a 9-month period. Children's anxiety and parents' cultural adaptation were examined via parent questionnaires. Receptive language skills were assessed with a standardized test. Every 3 months, parents and preschool teachers reported on the children's speaking behavior via questionnaires. Anxiety best predicted the development of mute behavior. There was no effect of bilingual status on its own. The effect of language skills did not reach significance but was considerably higher in preschool settings in comparison with family and public situations. Results also indicated an association between parents' orientation to the mainstream culture and children's speaking behavior in preschool. Level of anxiety might function as an early indicator of SM, especially in bilingual children, when information on language proficiency is scarce. There is still a need for intensive research in order to further the understanding of the development of SM in bilingual children.

[Selective mutism]. / Selektiver Mutismus.

Selective mutism was first described in the medical literature 140 years ago. The diagnosis came into the focus of adult psychiatry with the appearance of DSM-5. Henceforth, selective mutism during infancy, adolescence and also adulthood is specified as an independent anxiety disorder. It often begins in early childhood with a kind of speechlessness in certain situations. A diagnostic clarification often only takes place after school enrolment. Very often comorbid anxiety disorders, especially social phobia and depression also occur. The course is very variable and with some affected persons regression of the pathology occurs suddenly and completely and with others there is a slow regression of the symptoms. Equally the disorder can persist until adulthood. Whilst formerly a traumatic genesis was assumed, a multifactorial etiology with genetic, psychological and language-associated effects is nowadays presumed. The therapy is supported through psychotherapy, speech therapy and psychopharmacology.

Treatment of selective mutism: a 5-year follow-up study.

Selective mutism (SM) has been defined as an anxiety disorder in the diagnostic and statistical manual of mental disorders (DSM-5). Cognitive behavioral therapy (CBT) is the recommended approach for SM, but prospective long-term outcome studies are lacking. Reports from the children themselves, and the use of more global quality of life measures, are also missing in the literature. We have developed a school-based CBT intervention previously found to increase speech in a pilot efficacy study and a randomized controlled treatment study. Continued progress was found in our 1-year follow-up studies, where older age and more severe SM had a significant negative effect upon outcome. In the present study, we provide 5-year outcome data for 30 of these 32 children with SM who completed the same CBT for mean 21 weeks (sd 5, range 8-24) at mean age 6 years (10 boys). Mean age at the 5-year follow-up was 11 years (range 8-14). Outcome measures were diagnostic status, the teacher- and parent-rated selective mutism questionnaires, and child rated quality of life and speaking behavior. At the 5-year follow-up, 21 children were in full remission, five were in partial remission and four fulfilled diagnostic criteria for SM. Seven children (23%) fulfilled criteria for social phobia, and separation anxiety disorder, specific phobia and/or enuresis nocturna were found in a total of five children (17%). Older age and severity at baseline and familial SM were significant negative predictors of outcome. Treatment gains were maintained on the teacher- and parent questionnaires. The children rated their overall quality of life as good. Although most of them talked outside of home, 50% still experienced it as somewhat challenging. These results point to the long-term effectiveness of CBT for SM, but also highlight the need to develop more effective interventions for the subset of children with persistent symptoms.Clinical trials registration NCT01002196.

Latent Class Symptom Profiles of Selective Mutism: Identification and Linkage to Temperamental and Social Constructs.

Selective mutism (SM) is a stable, debilitating psychiatric disorder in which a child fails to speak in most public situations. Considerable debate exists as to the typology of this population, with empirically-based studies pointing to possible dimensions of anxiety, oppositionality, and communication problems, among other aspects. Little work has juxtaposed identified symptom profiles with key temperamental and social constructs often implicated in SM. The present study examined a large, diverse, non-clinical, international sample of children aged 6-10 years with SM to empirically identify symptom profiles and to link these profiles to key aspects of temperament (i.e., emotionality, shyness, sociability, activity) and social functioning (i.e., social problems, social competence). Exploratory and confirmatory factor analysis revealed anxiety/distress, oppositionality, and inattention domains. In addition, latent class analysis revealed nuanced profiles labeled as (1) moderately anxious, oppositional, and inattentive, (2) highly anxious, and moderately oppositional and inattentive, and (3) mildly to moderately anxious, and mildly oppositional and inattentive. Class 2 was the most impaired group and was associated with greater emotionality, shyness, and social problems. Class 3 was the least impaired group and was associated with better sociability and social competence and activity. Class 1 was largely between the other classes, demonstrating less shyness and social problems than Class 2. The results help confirm previous findings of anxiety and oppositional profiles among children with SM but that nuanced classes may indicate subtle variations in impairment. The results have implications not only for subtyping this population but also for refining assessment and case conceptualization strategies and pursuing personalized and perhaps less lengthy treatment.

Children's and parent's psychological profiles in selective mutism and generalized anxiety disorder: a clinical study.

Selective mutism (SM) is classified in DSM-5 as an anxiety disorder. The aim of the study was to investigate the psychological features of children with SM and their parental psychological profiles, compared to generalized anxiety disorder (GAD) children and their parents. The parents of 26 preschool children with SM and 32 with children with GAD filled out the child behavior check list for 1½-5 years (CBCL1½-5) and the symptom checklist-90-revised (SCL-90-R). Information about the children and their parents' histories was collected through clinical interviews. Children with SM scored higher than children with GAD on the CBCL1½-5 withdrawn scale and lower on the attention problems, aggressive behavior, and externalizing problems scales. Mothers of children with SM scored higher on the SCL-90-R obsessive-compulsive subscale and Global Severity Index than mothers of children with GAD, while fathers of children with SM scored higher on the SCL-90-R Phobic Anxiety subscale and on the Global Severity Index than fathers of children with GAD. Parents of children with SM displayed a greater presence of stressful life events than parents of children with GAD. Data appeared to confirm that SM and GAD share a common anxious core, though some differences in the children's psychological profiles and the parents' history and personality emerged. Future research should focus on the role of external factors, such as parent-child relationship, in the development of SM.

A voz no autismo: uma análise baseada em autobiografias / Voice in autism: an analysis based on autobiographies / La voz en el autismo: un análisis basado en autobiografías

Neste artigo, abordamos a voz no autismo a partir da análise de autobiografias de autistas e de seus pais. Para esta finalidade, enfocamos manifestações como o mutismo, o grito gutural, o monólogo, a expressão irruptiva de palavras esporádicas, a ecolalia, uma língua privada não compartilhada, o recurso a intermediários como o gravador, a mimetização de voz de animais, o recurso a scripts de personagens Disney ou livros. Após descrevermos e sinalizarmos alguns aspectos importantes evidenciados nessa maneira singular de se relacionar com a voz, apontaremos algumas nuanças do funcionamento psíquico do autista.

In this paper, we analyse voice in autism based on the analysis of autobiographies written by autistics and their parents. For this purpose, we focus on manifestations such as mutism, guttural scream, monologue, the irruptive expression of sporadic words, echolalia, a private non-shared language, the use of intermediaries such as tape recorders, animal voice mimicry, Disney characters or books. After describing and signaling some importante aspects evidencied in this unique way of relating to the voice, we will point out some nuances of the psychic functioning of the autistic.

En este artículo trataremos de la voz en el autismo a partir del análisis de autobiografías de autistas y de sus padres. Para tal finalidad, abordamos manifestaciones como el mutismo, el grito gutural, el monólogo, la irrupción de palabras esporádicas, la ecolalia, una lengua privada no compartida, el recurso a intermediarios como el grabador, la mimetización de la voz de animales, el recurso a guiones de personajes de Disney o a libros. Tras describir e indicar algunos aspectos importantes puestos de manifiesto en esta manera singular de relacionarse con la voz, señalaremos algunos matices del funcionamiento psíquico del autista.