Selective Mutism a Review and Integration of the Last 15 Years

Introduction

Selective mutism (SM) is a psychological condition usually occurring during childhood that is characterized by a total absenteeism of voice communication in specific social situations while speech product appears to exist normal in other situations. For instance, children with SM may non respond to a question posed by the teacher in class and/or do not speak to peers at school, merely do verbally communicate with parents, siblings, or other familiar people encountered in the domicile environment. To formally establish the diagnosis, current classification systems assume that the selective non-speaking behavior is required to be nowadays for at least 1 month, should not be attributable to a lack of knowledge of, or discomfort with, the spoken linguistic communication required in the social situation, and has to interfere significantly with daily functioning in school, work, or social life. Furthermore, the disturbance is not better explained past a advice disorder (eg, childhood-onset fluency disorder) and does not occur exclusively during the course of autism spectrum disorder, schizophrenia, or another psychotic disorder.^1,ii

SM is a relatively rare disorder. Estimates on its indicate prevalence take been obtained in clinic or school samples in various countries and typically range between 0.03% and 1.9% depending on the setting (eg, dispensary vs school/general population) and ages of the children in the sample.ⁱⁱⁱ SM is typically an early on-onset status, starting usually before the age of 5 years and often becoming a focus of clinical attention when children enter schoolhouse.¹ The course of SM is variable: some children continue to demonstrate the prototypical muteness associated with the disorder, but in many young people the selective non-speaking behavior gradually diminishes while symptoms of social reticence and social anxiety often remain.^4,5

The dramatic symptomatology of SM has puzzled researchers and clinicians in both psychology and psychiatry for virtually i and a one-half centuries. Initially, SM was conceptualized as an oppositional behavior problem – as evidenced by previously employed labels such equally "voluntary aphasia"⁶ and "elective mutism"^vii suggesting that these children intentionally choose to remain mute in certain situations or with certain people. The current view is more neutral regarding children's motives, with the term "selective" referring to the fact that children's lack of speech just occurs in item contexts or settings.^1,8 Moreover, the mostly accepted idea is that the prototypical non-speaking behavior of children with this disorder is fueled by fear and apprehension that predominantly occurs in certain social situations, which is why SM is now regarded every bit an anxiety pathology.

In this article, we volition get-go summarize research evidence supporting that in that location indeed is an intimate link between SM and (social) feet that justifies its recent re-classification equally an anxiety disorder. We will and then talk over the implications of viewing SM as an anxiety disorder for the classification, cess, and treatment of this condition. Next, we will point out that – although we agree that anxiety is clearly implicated, there are also indications that SM is a heterogeneous disorder in which a number of other psychopathological and developmental phenomena are possibly involved. We contend that this related phenomenology should not be neglected in the assessment and clinical management of children with this debilitating disorder.

Empirical Evidence on the Link Between SM and (Social) Anxiety

Evidence for the relation between SM and (social) anxiety essentially comes from three lines of inquiry. The first research line is concerned with the investigation of the comorbidity rates betwixt SM and anxiety pathology. A recently published meta-analysis nicely summarizes the results of 22 studies in which clinical interviews were conducted in children with SM to establish the co-occurrence of other anxiety disorders.⁹ The results showed that 80% of the children with SM as well fulfilled the diagnostic criteria for at least one other anxiety disorder, and in the vast majority of cases (69%) this involved social anxiety disorder (SAD). A second line of enquiry has focused on the content of the fears and fear-related cognitions of children with SM. For example, Vogel et al interviewed children with SM near their fears and worries in speech-related situations and assessed their levels of social fear-related cognitions in comparison to typically developing children and children with SAD.^x The qualitative interviews revealed that the content of the fears reported by children with SM were predominantly focused on typical social feet themes (ie, fear of being scrutinized and being critically or negatively evaluated by other people). The quantitative comparisons revealed that children with SM reported equally high levels of negative fright-related cognitions as children with Lamentable, with both clinical groups displaying college scores than typically developing children. A third and last line of inquiry has to exercise with the temperament characteristics of children with SM. There is increasing show that children with SM display high levels of shyness, get easily distressed when facing novelty, and have a tendency to withdraw from unfamiliar situations.^11–13 These are all defining features of the temperament typology of "behavioral inhibition to the unfamiliar"¹⁴ which has been demonstrated as an important vulnerability cistron of anxiety pathology, and SAD in particular.^15,16

Taken together, it can exist concluded that there is a articulate link between SM and fear and anxiety, which justifies the decision of electric current classification systems to categorize SM every bit an anxiety disorder.^1,2,17 Farther, SM appears to accept a special relation with social anxiety, which has prompted some scholars to argue that SM should be regarded every bit a variant of, and more than in specific a developmental precursor of Sorry,^eighteen which seems to be supported by its early historic period-of-onset in combination with the ascertainment that full muteness usually disappears when children become older.^4,v This link has been confirmed past empirical studies which have noted that there are clear similarities between children with SM and children with Sad in terms of fearfulness content and levels of fear and anxiety.^x,19–22 The master implication of this observation for clinicians is that when they encounter a child with SM they should at least initially manage the case as any other anxiety disorder.^iii,17 Of class, if co-occurring disorders besides are present they too volition need to get part of the clinical management picture. What that implies volition be the topic of our next section.

Clinical Management of SM as an Anxiety Disorder

Classification and Assessment

Clinicians can use the SM module of the Anxiety Disorders Interview Schedule for Children and Parents (ADIS-C/P)²³ or the Schedule for Melancholia Disorders and Schizophrenia for Children (Kiddie- or K-SADS)²⁴ to formally establish the diagnosis of the disorder. Other modules of these instruments tin also exist used to check frequent comorbid conditions, notably SAD and other anxiety issues. Usually, these semi-structured clinical interviews are administered to both the child and the parent. However, given the key symptom of the status (ie, the non-speaking behavior), it is likely that one volition demand to rely but on the parent equally the principal informant.

To get an impression of the severity of the trouble and to eventually monitor treatment progress, the Selective Mutism Questionnaire (SMQ)²⁵ can exist administered. This parent-based scale consists of 17 items measuring the frequency of children'due south not-speaking behavior across various settings, ie, at home, in school, and in other public/social situations. The SMQ is a reliable calibration and there is also testify for its validity.^25–27 A recently adult alternative instrument is the Frankfurt Scale of Selective Mutism (FSSM).²⁸ The FSSM is a parent-report measure that not only yields a severity index of SM symptoms but also includes a diagnostic scale that can be used to evaluate the presence of the core characteristics of the disorder and hence serves to support the establishment of the diagnosis.

Oftentimes, the lack of speech in school is the most important reason for parents to seek help for their kid, and so an cess of SM symptoms in the school situation is particularly relevant. For this purpose, an observation is indicated to actually witness the muteness in class and during other school activities. When this is non feasible, the School Speech Questionnaire (SSQ)²⁹ can be a useful tool to measure the teacher'southward perception of the frequency of children's non-speaking behavior.

In spite of the fact that SM at present belongs to the category of anxiety disorders, it is remarkable that its defining criteria practice non explicitly refer to fear and feet. This means that clinicians need to use other assessment instruments to obtain information on this important aspect of the problem. Given the young age of many children with SM, parent-written report questionnaires are the main source of information,³⁰ just as children become older it is as well possible to employ self-report scales to assess the intensity and frequency of fear and feet symptoms, in particular in the social domain. The Social Feet Scale for Children³¹ and the Social Phobia and Anxiety Inventory³² are excellent choices for this purpose.

Treatment

With the acknowledgement that SM primarily is a fearfulness- and anxiety-driven trouble, it is skilful to see that – at least in the scientific literature – cerebral-behavioral therapy (CBT) is more often than not recognized every bit the most feasible intervention for children with this disorder. Briefly, CBT for SM consists of the aforementioned components that also constitute CBT for other anxiety disorders,³³ namely (1) psycho-education – defining SM as an expression of feet and specifically of social anxiety; (two) physiological training – animate and muscle relaxation; (3) behavioral training – contingency direction, hierarchical exposure, modeling, shaping, and gradual desensitization; (4) cerebral training – positive self-talk and cognitive restructuring; and (5) parent grooming – enhancing parents' skills in assisting their kid and gradually discontinuing the mutism behaviors.³⁴

Figure 1 provides an overview of the psychosocial approaches that were investigated as treatments for children with SM in studies conducted in the fourth dimension periods 1980–1996, 1995–2005, and 2005–2015.^35–37 Information technology can be seen that ever since 1980, CBT interventions for SM have always been the almost pop in research settings; moreover, during the final decade well-nigh no intervention written report can be found that did not include strategies such every bit reinforcement, exposure, and cognitive restructuring. Information technology is also clear that researchers have focused less on the psychodynamic treatment of SM but have gained increasing interest in the systems approach (see Figure 1).

Figure 1 Overview of research on psychosocial interventions for children with SM. For each period, the percentages of studies incorporating various handling modalities are shown. Notation: Data from these studies.35–37

The research on the outcome of interventions for children with SM is even so somewhat inconclusive. This is because many studies have just described the effects of treatment in a single case or but a few children with SM, which of course hinders the generalization to other youngsters suffering from the condition. Meanwhile, 3 controlled trials accept been published and all have evaluated the efficacy of CBT on the non-speaking beliefs and associated feet symptomatology of children with SM. In the first investigation, Bergman et al evaluated a twenty-session CBT-based program that mainly consisted of behavioral techniques.³⁸ 20-one children with SM were randomly assigned to CBT or a waitlist control status. Information technology was found that 67% of the children who had received CBT no longer fulfilled the diagnostic criteria of SM, whereas 0% of the children in the waitlist condition attained such a diagnosis-free status. Moreover, on standardized measures completed past parents and teachers, the children treated with CBT were reported to display increased functional speaking and decreased levels of social anxiety, which were positive developments that did not occur in children in the waitlist status. Similar findings were obtained in 2 other controlled investigations past Cornacchio et al³⁹ and Oerbeck et al^twoscore that as well compared the effects of a CBT for children with SM versus a waitlist control condition. Children of the latter study were re-assessed 5 years subsequently termination of treatment,⁴¹ and the results revealed that 70% were no longer diagnosed with SM at this long-term follow-up, whereas a further 17% of the children were in partial remission. Meanwhile, 23% of children met the criteria for SAD, and 50% found it challenging to talk outside of home. This indicates that although the positive effects of the intervention were largely maintained, a substantial number of the children showed persistent bug, which of class highlights the demand for developing even more than effective treatments.

For other childhood anxiety disorders, pharmacotherapy, and in particular handling with selective serotonin reuptake inhibitors (SSRIs), is considered a viable intervention option.⁴² The general clinical guideline is that SSRIs are indicated when CBT yields an insufficient treatment response,⁴³ but in that location are also indications that a combination of CBT and pharmacotherapy may be even more effective in treating anxiety disorders in children and adolescents than each of both monotherapies on their own.⁴⁴ Enquiry on the efficacy of SSRIs in children with SM is thin, which is not that surprising given the possible side effects of this type of medication⁴² and the fairly young age of most children with this condition.^three The few studies that take been conducted are limited by small numbers, heterogeneous designs, the absenteeism of a comparison grouping, and lack of consistent result measures. Nonetheless, the results advise that SSRIs yield symptomatic comeback, although information technology remains largely unclear how many children achieve full remission.⁴⁵ So far, support for a combination treatment of CBT and SSRI in the treatment of SM is missing.⁴⁶

There are indications that outcome is better when children with SM are treated at a younger age.⁴¹ This as well raises the question of whether it is possible to implement early interventions thereby averting the development of the persistent pattern of not-speaking beliefs associated with the disorder. Given the strong relationship betwixt SM and behavioral inhibition, this temperament trait might be an important target for such a preventive approach. The trait is easily detectable and can be finer addressed past ways of a brief parent-based intervention program that aims to promote exposure, adjust negative thinking, and enhance social skills.⁴⁷ In a serial of studies, it has been demonstrated that this intervention delivered to inhibited preschool children can reduce the development of babyhood anxiety disorders both on the short- and the long-term,^48–52 and it would be interesting to study whether this approach is also useful of altering the trajectory of SM.

Taken together, feet is a prominent symptom of children with SM and as such the most logical approach is to care for the condition not unlike that of whatsoever other anxiety disorder. CBT-based interventions are certainly the number one option for treating SM, and it is good to see that this notion has increasingly percolated into ongoing research.³⁷ This does non automatically imply that this is also truthful for clinical practise. Kazdin⁵³ rightly noted that there still exists a huge gap between assessments and treatments that accept been demonstrated every bit "bear witness-based" and what is really used past clinicians in their daily practice. For case, many clinicians still rely on psychodynamic-based interventions such as play therapy when dealing with children with SM. It is not our intention to downplay the importance of play therapy – especially when working with young children this is certainly a valuable method, but given the extant empirical evidence, it is strongly recommended to comprise CBT elements within such an intervention.⁵⁴

Another case is concerned with unremarkably encountered advice on how therapists should approach a child with SM. For instance, on the basis of their clinical expertise, Oerbeck et al⁴⁰ take avant-garde the principle of "defocused communication", which requires the therapist

to sit abreast rather than reverse the child, to conduct a joint activeness ..., to think aloud rather than asking the child straight questions, to give the child enough time to respond ..., and try to receive a verbal respond in a neutral way. (p. 195)

Unfortunately, in that location is no empirical testify for the unique upshot of defocused communication in the treatment of children with SM. Further, although it seems a good strategy to "break the water ice" during the initial stage of therapy, the technique seems less appropriate when treatment progresses to a point that exposure exercises take to be conducted during which the child is required to speak and to respond to questions in real-life situations.

SM: More than Than an Anxiety Disorder?

Although the part of fear and anxiety is prominent in SM, it is skilful to exist aware that at that place is considerable evidence indicating that other psychopathologies or difficulties may fuel children's apprehension for speaking in social situations. In this section, nosotros will talk over a number of such problems and also address implications for the clinical direction of children with SM.

Speech and Language Bug and Developmental Filibuster

One case in point are speech and language problems, which have been shown to be nowadays in a considerable proportion of children with SM. For case, Steinhausen and Juzi⁵⁵ examined the clinical characteristics of 100 children with SM. It was found that 38% of the children with SM also displayed early speech and language disorders. In item, articulation disorder (20%) and expressive language disorder (28%) were highly prevalent. In some other study,⁵⁶ 54 children with SM and 108 control children were subjected to a comprehensive assessment while their parents were interviewed by means of a diagnostic interview. The results showed that children with SM were more than ofttimes characterized by a developmental delay – and this was often concerned with language issues (51.9%), as compared to children in the control grouping (xi.1%). In terms of comorbid diagnoses, the DSM-Iv classifications of phonological disorder (42.6%), mixed receptive-expressive language disorder (17.3%), and expressive language disorder (eleven.5%) were clearly more than prevalent among the children with SM (percentages in the control group beingness 10.ii%, 1.0%, and one.0%, respectively). In further research, Manassis and colleagues⁵⁷ employed standardized language tests to explore whether children with SM differed from (socially) anxious and normal command children with regard to their language skills and abilities. It was establish that children with SM relative to children in the other groups consistently displayed a poorer performance on linguistic tasks, although it was also noted that this not always unsaid that they all suffered from speech and language disorders. A terminal investigation past Cohan et al⁵⁸ adopted a quantitative arroyo in which parent-based measures were used to appraise social feet, behavioral problems, and linguistic communication delays in 130 children with SM. Subtypes of SM were identified past means of a cluster analysis. The results revealed that besides subtypes of exclusively anxious children and anxious-mildly oppositional children, bear witness was institute for a tertiary subgroup of children with SM showing a mix of anxiety and (sub)clinical levels oral communication and syntax problems.

Collectively these findings indicate that there is a subset of children with SM who display delayed and impaired oral communication and language skills. It is easy to see how these difficulties prompt children to become apprehensive of school and other social situations in which they are required to speak, and this may be peculiarly true for those who too have a (genetic/temperamental) susceptibility for anxiety. Two obvious clinical implications follow from the observation that voice communication and language problems are present in some children with SM. The first 1 is that it would be adept practice to conduct a proper clinical assessment in order to get a picture of a child's full general developmental level, which as well includes a careful evaluation of oral communication, receptive language, expressive language, and phonology.⁵⁹ Receptive linguistic communication tests such as the Peabody Picture Vocabulary Test⁶⁰ tin be administered in a non-verbal way, but the evaluation of other aspects of language and spoken communication require the child to speak. To deal with this problem, the clinician could ask parents to make an audiotape recording of the child speaking in the dwelling house situation, or train parents to administer the standardized test materials, a method that has been proven to yield reliable and valid data about children's level of voice communication and linguistic communication.⁶¹ The second implication only applies if the assessment indeed reveals the presence of voice communication and language problems in a child with SM. In that case, a spoken language therapist should exist involved to remediate the speech and language difficulties and eternalize the confidence of the kid in its voice communication and linguistic abilities.⁵⁹

Autism Spectrum Problems

Although the presence of autism spectrum disorder (ASD) is considered as an exclusion benchmark of SM, the more dimensional approach of psychopathology taken by the current DSM-5¹ makes it difficult for clinicians to establish a clear boundary between these two conditions. Meanwhile, there is increasing research evidence showing that there is a clear relation between SM and ASD. For example, Steffenburg et al⁶² conducted a detailed analysis of the medical records of 97 children who had been referred to the dispensary with SM as the primary diagnosis. The post hoc analysis revealed that 62% of the children with SM could too be diagnosed with ASD: in DSM-IV terms: 29% had autistic disorder, 4% had Asperger's syndrome, and 29% had pervasive developmental disorder-not otherwise specified. Further, an additional 20% of children clearly showed autistic features but did not receive a formal ASD diagnosis, which means that in this sample only eighteen% showed no overt signs of ASD. In another study by Klein et al⁶³ that included 42 clinically referred children with SM, parents and teachers completed the Behavior Assessment System for Children (BASC),⁶⁴ a standardized scale for measuring internalizing symptoms, externalizing problems, and adaptive skills. Interestingly, the BASC likewise contains a clinical index that consists of specific items relating to developmental social problems, atypicality, and withdrawal that would exist indicative of the presence of ASD. Findings indicated that 80% of the children with SM scored above the cut-off of this autism probability alphabetize, with many of the children showing signs of social and communication problems and stereotyped interests and behaviors. A farther investigation by Nowakowski et al⁶⁵ had the purpose to analyze the interactions of children with SM and their parents by focusing on joint attention abilities. Joint attention is defined as the shared focus of two individuals on an object or event that is achieved when one individual alerts the other person by means eye-gazing, pointing, and exact directions. Importantly, this ability has been shown to be impaired in children with ASD. In the Nowakowski et al report, joint attention abilities of children with SM, children with other feet problems, and control children were assessed under ii conditions: during unstructured free play and during a number of structured tasks (eg, the parent had to set the child for a speech in front of the camera). No differences were found betwixt the three groups with regard to the level of joint attention behaviors during the unstructured gratuitous play condition. Yet, under more structured conditions, children with SM appeared to constitute significantly fewer joint attending episodes post-obit parental initiation as compared to children and parents in the other two groups, which tentatively indicates that children with SM (to some extent) display a cognitive deficit that has also been observed in children with ASD. A final study by Stein et al⁶⁶ adopted a pathophysiological approach to written report the link between SM and ASD. In 99 families that included 106 children with SM, a number of unmarried nucleotide polymorphisms (SNPs) in the contactin-associated protein-like ii (CNTNAP2) were genotyped. The results showed that the SNP rs2710102 was significantly associated with the presence of SM. Because the CNTNAP2 is known as a susceptibility gene for ASD, Stein et al concluded that some forms of SM share a similar genetic liability with disorders in the autism spectrum.

Although studies on the link betwixt SM and autism spectrum bug are subject to various methodological shortcomings,⁶⁷ findings bespeak that some children with SM do testify clear signs or even see total criteria of this neurodevelopmental disorder. In a recent review,⁶⁷ we have advocated to revise the current classification criteria of SM and to allow SM and ASD to be comorbid atmospheric condition, simply as is currently done with SAD and ASD. The ascertainment that ASD is present in some children with SM has a number of clinical implications. To begin with, it is critical to assess symptoms of ASD during the diagnostic evaluation of children who do not speak in specific social situations. To conduct such assessment in an constructive and economic mode, a two-pace procedure equally described by Volkmar et al⁶⁸ could be adopted. This procedure consists of a first screening of ASD symptoms past means of a scale that has been specifically developed for this purpose (eg, the Social Advice Questionnaire),⁶⁹ followed by a more extensive diagnostic evaluation by means of the Autism Diagnostic Observation Calibration (ADOS)⁷⁰ and the Autism Diagnostic Interview (ADI),⁷¹ which are currently considered the gilded standard cess instruments for establishing ASD. The employ of more objective measures such as the ADOS and ADI is not only helpful to plant the presence of autistic features in children with SM only could also identify children who only take SM and who are currently – by some clinicians – erroneously labelled as cases of ASD.

The presence of ASD volition besides have repercussions for the treatment of SM. The delivery of the regular CBT intervention needs to be optimized by increasing the use of visual aids, providing more than structure, incorporating actress sessions, and adding more than relaxation exercises.⁷² Furthermore, it may exist necessary to add special treatment components that target specific social problems that occur in children with this neurodevelopmental disorder. For example, Pallathra et al⁷³ noted that individuals with ASD show specific impairments in social cognition, social skills, and social motivation, all of which require attention during handling. Moreover, the often inflexible and rigid behavior of children with ASD may also require clinical attention, either by specific behavioral interventions or by prescribing antipsychotic medication.

Conclusion

SM is a rare but debilitating disorder that has puzzled researchers and clinicians for a long time. Empirical insights indicate that SM is mainly fear- and anxiety-driven and as such clinicians need to approach the status equally an anxiety disorder.^3,17 In a nutshell this implies that the assessment procedure besides an index of speaking frequency should incorporate (social) fright and feet scales, while treatment has to be CBT-based as this currently is the best available empirically-supported intervention for childhood anxiety disorders. Meanwhile, the cautionary note has to be made that this may not exist the full story. As pointed out by various authors,^74,75 SM is likely to exist a heterogeneous disorder. This means that while in some children fright and anxiety may be the sole basis of not-speaking behavior in specific social situations, there are other children for whom other psychopathologies and difficulties contribute to the etiology and expression of SM. In this cursory article, nosotros have discussed the examples of speech and language issues, developmental filibuster, and autism spectrum disorder in the hope that clinicians will address these factors in their assessment, and ultimately deploy the nearly optimal treatments in children with SM.

Disclosure

The authors report no conflicts of interest in this work.

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