International Journal of Pediatric Otorhinolaryngology 78 (2014) 334–338

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Language regression in children with Autism Spectrum Disorders Suman Kumar *, Probir Karmakar, Akhil Mohanan Ali Yavar Jung National Institute for the Hearing Handicapped, Eastern Regional Centre, Kolkata, India

A R T I C L E I N F O

A B S T R A C T

Article history: Received 19 July 2013 Received in revised form 30 November 2013 Accepted 3 December 2013 Available online 12 December 2013

Objectives: Regression in autism applies to the phenomenon of apparently normal early development followed by the loss of previously acquired skills and manifestation of symptoms of autism. Estimates of the frequency of regression in autism range from 10% to 50%. Although there are tools available to evaluate and diagnose Autism Spectrum Disorders, however, there is no published tool available in Indian context to identify the children with ASD at an early age. The study was aimed to describe the differences in language regression between children with ASD and typically developing children and also to determine the age of regression. Methods: Regression screening tool, a questionnaire was developed based on Regression Supplement Form (Goldberg et al., 2003). The skills were validated by five Clinical Psychologists. It comprised of 16 skills which included domains like, ‘spoken language and non verbal communication’, ‘social interest and responsiveness’ and ‘play and imagination’. This retrospective study was conducted on a single group. The participants consisted of parents of 30 children with ASD (22 males and 8 females). Results: The findings revealed a significant regression in children with ASD. The mean regression age is 20.19 months (SD-5.2). The regression profile of the children with ASD revealed regression of language skills occurred at 19.16 months followed by non language skills at 20.5 months. Conclusions: Based on the findings it can be stated that inclusion of regression screening tool will offer clinicians a convenient tool to examine the phenomena of regression in children with ASD and identify them as early as 21 months of age for early intervention. ß 2013 Elsevier Ireland Ltd. All rights reserved.

Keywords: Autism Spectrum Disorder Linguistic and non linguistic regression Regression screening tool

1. Introduction Autism Spectrum Disorder (ASD) is a behaviourally defined syndrome characterized by qualitative impairments of social interaction and communication accompanied by restricted, repetitive and/or stereotyped patterns of behaviour and interests [1,2]. The slow, limited or otherwise faulty psychological development in these key behavioural domains is thought to result from subtle anomalies in brain development [3–5] that hamper the normal elaboration of neurofunctional networks mediating social cognition, verbal and gestural communication, and self regulation/awareness [6–9]. Based on methodological differences in sampling and/or geographical variability in identification and diagnosis prevalence estimates for ASD vary from study to study. Current prevalence estimates for ASD in the United States are 34 per 10,000 or 0.34% based on a recently reported study by the Centre for Disease Control and Prevention [10]. Large epidemiologic studies have

* Corresponding author at: AYJNIHH, ERC, B.T. Road, Bon Hooghly, Kolkata 700090, India. Tel.: +91 08981812836. E-mail address: [email protected] (S. Kumar). 0165-5876/$ – see front matter ß 2013 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijporl.2013.12.004

reported increases from estimates of 2–5 per 10,000 for autistic disorder in the 1970s to 6–22 per 10,000 by the turn of the century [11,12]. Regression occurs in approximately 15–40% of children with autism [13]. While it is common for parents to report dramatic changes in their child’s behaviour and skill level, some report a more gradual regression. Functioning is often perceived as normal prior to regression; however, retrospective methods suggest subtle preexisting abnormalities [13]. Regression is typically succeeded by a regaining of skills to some extent; however, researchers have reported various outcomes. For instance, Kobayashi and Murata [14] examined the long-term prognosis of children with regressive autism and found the majority to have higher rates of epilepsy and a lower language achievement. However, Lord et al. [15] found that the time between word loss and the reemergence of words was only 4–5 months in children who would go on to speak in phrases at 5 years of age. Regression is also displayed in children with CDD who experience several years of seemingly normal development, followed by a broad deterioration that occurs between 3 and 5 years of age [16]. The mean age range for autistic regression falls between 18 and 24 months of age [17]. It is usually diagnosed around 21 months of age, but has been detected as early as 15 months of age [18]. The

S. Kumar et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 334–338

loss of language is most easily noticed by the parents; however, it rarely occurs in isolation [15,19,20]. In fact, 76–93% of children who lost language skills had an associated loss of another nonlanguage skill [19,21] such as direct gaze, orientation to name, spontaneous imitation or response to social overture. The general approach of retrospective studies that have used interviews or questionnaires has been to ask about the child’s behaviour in the first 2 or 3 years of life. Few studies have focused exclusively on younger ages. An exception is the study of Vostanis et al. [22], in which the parents of referred children with ASD completed a questionnaire on concerns regarding their child’s development between 12 and 18 months. Play behaviour items and lack of referential gestures were found to best predict the diagnosis of autism. Gillberg [23] studied symptoms reported by the parents of children with autism younger than 2–3 years. The item ‘‘Does not play like other children’’ was among the three items with the strongest discriminatory power. Early detection of autism is important because early behavioural intervention can have a significant impact on the long-term prognosis of many children with autism. Unfortunately, autism is not typically diagnosed in children until around 3–4 years of age [24]. Therefore many children do not receive intervention as early as would be optimal. Although there are tools available to evaluate and diagnose Autism Spectrum Disorders, there are very limited tools available to explore parent reported regression in depth. Regression was earlier reported only in Rett’s syndrome and childhood disintegrative disorder. But of late, regression was reported across spectrum of autism disorders. Characteristics of different sub types of ASD have been listed in Table 1. However, there is no published tool available in Indian context to identify the children with ASD at early age. So there is a need to develop regression screening tool which may identify children with ASD as early as possible. The primary aim of the study is to describe language regression in children who are later diagnosed with ASD and whether this language regression can be an early indicator of Autism Spectrum Disorders. The main objectives are (1) To study the language regression in children with Autism Spectrum Disorders and (2) To determine the age of regression of language skills (Fig. 1). 2. Methodology 2.1. Participants Participants comprised of parents of 30 children diagnosed with ASD. The parents of only those children were included as a part of the study who met the following criteria: 1. The diagnosis of ASD has been further confirmed by the researchers with an experienced Clinical Psychologist in the field of autism. 2. All the children had a history of regression.

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Fig. 1. Age of regression reported in different studies.

3. Age range of the children was between 3 and 8 years. 4. No associated problem. 5. Mothers of the children were homemakers. The study was accomplished in two phases. 2.2. Phase-I: development of the test material  A questionnaire, consisting of language and non language skills was developed based on the Regression Supplement Form (RSF) given by Goldberg et al. [19] (Appendix-II).  The questionnaire consisted of nineteen skills in the following domains like: spoken language (at least five single words, 2–3 word phrases) and nonverbal communication (nonverbal communicative gesture), social interest and responsiveness (interest in children, offers to share, shares enjoyment with others), and play and imagination (displays imaginative or pretend play).  The questionnaire was given to five Clinical Psychologists to validate the skills used. They were asked to rate the skill on 3 point rating scale, 1-most relevant, 2-relevant and 3-not relevant. Skills, rated as most relevant by at least 4 judges were included in the questionnaire. By this process 16 skills were selected from the entire domain. These 16 skills are mentioned in Table 1.  The questionnaire was then translated in Bangla by 5 Speech Language Pathologists and retranslated in English by another 5 Speech Language Pathologists to ensure the content.

Table 1 Characteristics of different subtypes of ASD. Landau–Kleffner syndrome

Childhood disintegrative disorder

Rett’s syndrome

Autistic disorder

Normally established skills gradually or suddenly develop a progressive aphasia, most commonly between 3 and 7 years of age. Initially, loss of verbal understanding, followed by deterioration in expressive communication ability and affected children may become completely mute and fail to respond even to nonverbal sounds.

Previously completely normal children who undergo massive regression between 2 and 10 years of age and mental retardation.

Characterized by normal early growth and development followed by a slowing of development, loss of purposeful use of the hands, distinctive hand movements, slowed brain and head growth, problems with walking, seizures, and intellectual disability.

Severe qualitative deficits in social interaction and communication skills, restricted and stereotyped patterns of behaviour, interests, and activities and Onset before the age of 3 years.

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Table 2 Frequencies of commonly reported losses. Skills

Frequency (n)

Age of loss Mean (M)

Standard deviation (SD)

2–3 words phrases Respond to own name At least 5 single words including mama, dada Direct gaze in social communication Social games/play Appropriate response to social overtures from adults Child was just as active as other children about surroundings Spontaneous imitation of non-taught action Social smiling Non verbal communicative gesture Interest in/watches children Share enjoyment with others Offer to share Imaginative/pretend play Show and direct attention to object when offered Asks for help by socially acceptable means

28 27 27 26 26 22 18 13 13 11 8 8 6 5 5 4

19.16 21 19.16 20.06 20.15 20.5 20.40 19.56 20.06 20.07 20.68 20 19.82 19.42 20.78 22.2

4.79 4.9 7.23 5.2 3.66 3.8 5.2 4.8 3.2 7.3 6.6 4.45 2.8 4.2 6.55 8.57

2.3. Phase-II: data collection Children with ASD who were attending special schools or centre like Autism Society of India (Kolkata), Child Development Centre (CDC), and Pradip School for Autism (Kolkata) were taken up for the present study. The parents of the children with ASD were contacted through schools. Written consent was obtained from each parent prior to the interview. Structured interviews were conducted by the examiner and purpose of the study was explained to the parents. Demographic data like date of birth of the child, educational qualification of parents, socioeconomic status were recorded. During interview, the parents were explained about the content of the questionnaire and asked to report whether the mentioned skills were lost or not and if yes, at what age. It took about 20–25 min to conduct each interview. The questionnaires were provided to the participants to report about the skills. The participants were given seven days to recall and report about each skill retrospectively. 2.4. Statistical analysis Standard statistical analysis was carried out to obtain the mean regression age and standard deviation (SD) of the collected data using SPSS version11.

3. Results The mean age of the children with ASD was 64.6 months (5 years 4 months). Out of 30 children with ASD 22 were male and 8 were female. Out of the 30 children only one child was diagnosed as Asperger’s syndrome. The rest 29 children were diagnosed as autism by a different Clinical Psychologist. Twenty-six (26) parents were graduates (86.67%). The socioeconomic status of all families was middle class. Monthly income of the parents was considered to obtain the socio economic status. The collected data was subjected to statistical analysis. It is inferred from the analyzed data that specific age related regression have been found in all 30 children with ASD. Similar finding has been reported by Osterling and Dawson [25] who reported that differences between normally developing children and children with autism can be identified by 1 year of age. Differences were found between the groups in three general categories of behaviours: social, joint attention, and certain autistic behaviours. In the present study the mean age of regression in children with ASD in all skills was 20.19 months (SD = 5.2). The mean age of only

language regression was 19.16 months whereas the mean age of only other skill regression was 20.5 months. The finding of the present study is in consonance with guidelines [26] and similar to the mean age reported by parents in the Cooper et al. [27] and Goldberg et al. [19] studies of language and non language skills. Hoshino et al. [28] stated that regression usually occurs in an age range from 15 months to 30 months old, while in other studies most cases regressing in a period between 18 and 24 months Davidovitch et al. [41,17,20,29]. Wilson et al. [21] reported mean age of onset of language regression in a mixed group of Regressive ASD and non-ASD children was 21.2 months with SD = 10.5 while studies of regressive ASD have reported a mean age of onset ranging from 18 months [30] to 25 months [13]. Responses to regression screening tool revealed several types of regression. The regression in children with ASD was described in terms of type of loss and age of loss to know types and age of regression. The regression was subdivided into three categories, i.e. regression in all skills, only language regression and only other non language skill regression. The finding of present study in terms of loss in all three categories is comparable with previous studies [15,19,21,31,32]. Regression in all skills was reported in 28 children (93.3%) however there was no regression found in only language skill. Other skills regression was reported in 2 children (6.7%). The result was consistent with findings of Goldberg et al. [19] where only language regression was found in 4.8% and full regression of language and non language skills was found in 35.7%. Amongst all the skills, 2–3 word phrase loss were most frequently reported by the parents (28 of 30) followed by respond to own name and at least 5 single word including mama, dada (27 of 30). Loss in skills in item’s like ‘‘child was just as active as other children about surroundings’’ (18 out of 30), ‘‘spontaneous imitation of non-taught action’’ (13/30) and ‘‘social smile’’ (13/ 30) were reported by almost half of the participants. Few skills like: ‘‘asks for help by socially acceptable means’’, ‘‘Show and direct attention to object when offered’’ and ‘‘Imaginative/pretend play skills’’ were least commonly reported by the participants (4, 5 and 5, respectively). To get a closer look at the types of losses which are most common, a rank order of the reported frequencies for each of the 16 skills is tabulated in Table 2. 4. Discussion Though several studies have been done on regression of children with ASD however, relatively few studies detailed the nature of this loss. When the age of regression was reported most of the children have progressed beyond the stage of complex

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babbling and proto-word production, and are often functioning at the single word level, with emerging or expanding expressive vocabularies of words that they use spontaneously and meaningfully. During the course of the regression, children cease to produce words that previously seemed a permanent part of their vocabulary while others showed marked reductions in their verbalizations, often to the point where they discontinue speaking altogether. Kurita [30] and Rutter [33] stated that around 20–40% of children, who had regression, lost all expressive language. Although regression age of language domain was 19.16 months in present study however large scale study done by Kurita [30] found 37.2% of 261 children lost speech preceding to 30 months of age. Wilson et al. [21] stated that the parents of children with autism reported language regression between 18 and 24 months of age, and 95% before age 3 years which was also similar to current findings. In another study only 13% of children with Landau– Kleffner syndrome regress before the age of 24 months [17,34–37]. In Landau–Kleffner syndrome behavioural problems develop as a reaction to the child’s emotional frustration at being unable to communicate adequately, and to the experience of social rejection, isolation, and low self esteem. However, in autism, behavioural regression and language regression occur simultaneously [30,37,38]. The finding of present study revealed that non verbal communicative gesture and asking for help were regressed at 20.07 months and 22.2 months of age, respectively. While Swinkel et al. [39] reported regression of protodeclarative pointing in more than 90% of the children after 13 months and instrumental pointing at about 14–15 months along with 25% of the parents apparently did not observe pretend play by age 18 months. Several reports have suggested that most children who regress in language demonstrated at least mild to moderate regression in social skills, evident in poor eye gaze, loss of social smiling, decreased spontaneous imitation of actions, loss of interest in others, withdrawn or aloof behaviour, and problems involving play skills [19,21]. 5. Conclusion At any age incidence of language regression should be a cause for timely concern. Regression is not a characteristic of simple developmental delay and is only rarely a characteristic of developmental language disorders [38,40]. Language regression usually occurs in the context of a more global autistic regression in younger children [21]. The finding of present study revealed that the mean regression age was 20.19 months with SD 5.2. It also indicated that the regression of language skills (19.16 months) was earlier than non language skills (20.5 months) though many studies reported simultaneous regression in both domains. It can be inferred from the study that language regression characteristic is the most important feature by which the children with ASD can be identified as early as by 21 months in the developing period from typically developing children. There is great need of further study on regression characteristics of children with ASD as the information about regression is commonly obtained retrospectively many months to years’ postonset. Due to the imprecise onset and prolonged time course that characterizes the regression in many cases. Parents may remember only that they slowly came to the realization that their child was no longer using words previously elicited frequently and meaningfully at an earlier stage of development. Variability in parent’s recognition and accuracy in reporting symptoms may make it difficult to separate age of onset from age of recognition. Research on children with regression is needed to be undertaken to differentiate language regression from other types of regression.

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Language regression in children with Autism Spectrum Disorders.

Regression in autism applies to the phenomenon of apparently normal early development followed by the loss of previously acquired skills and manifesta...
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