556782

research-article2014
AUT0010.1177/1362361314556782AutismNorrelgen et al.

Original Article
Autism

Children with autism spectrum disorders 2015, Vol. 19(8) 934­–943

© The Author(s) 2014
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DOI: 10.1177/1362361314556782

preschool years aut.sagepub.com

Fritjof Norrelgen1,2, Elisabeth Fernell2,3, Mats Eriksson1,2,4, Åsa

Hedvall1,2, Clara Persson4, Maria Sjölin4, Christopher Gillberg2
and Liselotte Kjellmer1,2,4

Abstract
There is uncertainty about the proportion of children with autism spectrum disorders who do not develop phrase
speech during the preschool years. The main purpose of this study was to examine this ratio in a population-based
community sample of children. The cohort consisted of 165 children (141 boys, 24 girls) with autism spectrum disorders
aged 4–6 years followed longitudinally over 2 years during which time they had received intervention at a specialized
autism center. In this study, data collected at the 2-year follow-up were used. Three categories of expressive language
were defined: nonverbal, minimally verbal, and phrase speech. Data from the Vineland Adaptive Behavior Scales-II were
used to classify expressive language. A secondary objective of the study was to analyze factors that might be linked to
verbal ability, namely, child age, cognitive level, autism subtype and severity of core autism symptoms, developmental
regression, epilepsy or other medical conditions, and intensity of intervention. The proportion of children who met
the criteria for nonverbal, minimally verbal, and phrase speech were 15%, 10%, and 75%, respectively. The single most
important factor linked to expressive language was the child’s cognitive level, and all children classified as being nonverbal
or minimally verbal had intellectual disability.

Keywords
autism, intellectual disability, minimally verbal, nonverbal, preschool, Vineland

Introduction
In recent years, the research community has highlighted
the lack of knowledge about children with autism
spectrum disorders (ASD) who do not develop spoken lan-
guage, or only minimally so, during the preschool years
(Kasari et al., 2013; Tager-Flusberg and Kasari, 2013).
Estimates of the proportion of children with ASD who do
not acquire communicative spoken language before the
age of 5 years vary considerably. A recurring estimation in
the slightly older literature states that approximately 50%
of children with autism never develop functional speech
(Prizant, 1996; Rutter, 1978; Sigman and McGovern,
2005; Volkmar et al., 1994). Authors of more current arti-
cles, however, claim that this figure likely has decreased
14%–20% of the studied children were still nonverbal at
the age of 9 years. Additionally, 11%–14% of the children
only had minimal speech using words but not three-word
phrases. The authors who reported these data themselves
argue that the results are not necessarily representative of
the entire ASD population since their two-cohort sample
also included a group that did not meet diagnostic criteria
for ASD. To the best of our knowledge, only one additional
study published after Lord et al. (2004) has reported
1Karolinska University Hospital, Sweden
2Universityof Gothenburg, Sweden
3Skaraborg Hospital, Sweden

considerably in recent years (Eigsti et al., 2011; Landa, 4Karolinska Institutet, Sweden

2007; Luyster et al., 2008; Tager-Flusberg et al., 2005;

Corresponding author:
Tager-Flusberg and Kasari, 2013). All the mentioned arti- Fritjof Norrelgen, Department of Speech and Language Pathology,
cles refer back to (directly or indirectly) one seminal study Karolinska University Hospital, 17176 Stockholm, Sweden.
by Lord et al. (2004), which found that approximately Email: fritjof.norrelgen@karolinska.se

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 Norrelgen et al.
proportions of nonverbal or minimally verbal children
with ASD (Anderson et al., 2007). When scrutinized, the
data reported by Anderson and colleagues seem to come
from the exact same two research cohorts as in Lord et al.
(2004), although presented in a slightly different way:
expressive language level at the age of 9 years was pre-
sented separately for children with autism, pervasive
developmental disorder—not otherwise specified (PDD-
NOS), and nonspectrum developmental disabilities.
Collapsing data for children with autism and PDD-NOS,
20% were found to be nonverbal (using no or few consist-
ent words) and an additional 19% only used minimal
amounts of speech (using words but not sentences).
Several researchers have proposed possible causes for
the suggested decrease in number of children with ASD
who do not acquire spoken language: (1) Children with
ASD are diagnosed earlier today and thus gain access to
earlier intervention, which in turn may result in more chil-
dren developing phrase speech (Goldstein, 2002); (2)
intervention may be more efficient due to improved treat-
ment strategies, which may make a big difference for
many children who might otherwise remain nonverbal/
minimally verbal (Koegel and Koegel, 1995; Rogers and
Vismara, 2008; Smith and Eikeseth, 2011); (3) the updated
diagnostic criteria and broader definitions for ASD in the
revised Diagnostic and Statistical Manual of Mental
Disorders (3rd ed.; DSM-III-R; American Psychiatric
Association (APA), 1987) and then in the Diagnostic and
Statistical Manual of Mental Disorders (4th ed.; DSM-IV;
APA, 1994) have led to an increased prevalence of ASD,
including more verbal individuals (CDC, 2012). With the
total ASD population thus becoming larger, the propor-
tions of nonverbal children as well as children with intel-
lectual disability are assumed to decrease in the total
group (Bryson et al., 1988; Kasari et al., 2013; Lord et al.,
2004; Tager-Flusberg et al., 2005). With these three expla-
nations in mind, it does not seem unreasonable that the
proportion of nonverbal children in the total spectrum of
ASD would have declined in recent years. In addition,
although developing speech after the preschool years is
considered rare for children with ASD, Pickett et al.
(2009) in their substantial review showed that it is not
impossible. In the 64 published materials reviewed, they
found a total of 167 individuals with ASD who developed
speech after the age of 5 years.
One crucial point when researching children with ASD
who fail to develop spoken language is how the borders
between verbal and nonverbal children are defined.
Noteworthy, studies have used different terms for, seem-
ingly, the same phenomenon. Terms such as nonverbal,
non-functional speech, little or no functional speech, and
lack of expressive language have been used (Lord et al.,
2004; Sigman and McGovern, 2005; Thurm et al., 2007;
Venter et al., 1992). Moreover, the criteria and borders
vary between studies, and nonverbal can mean anything
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935
from few or no words to include any child who does not
have enough language to be understood in communication
with people who do not know the child well. More recently,
the term “minimally verbal” has been used to describe
these children (Kasari et al., 2013; NIH-Workshop, 2013;
Tager-Flusberg and Kasari, 2013). Kasari et al. (2013)
define the minimally verbal child with ASD as having “a
very small repertoire of spoken words or fixed phrases that
are used communicatively” (p. 480); however, the exact
number of words and/or fixed phrases may vary. In addi-
tion, echolalic or stereotyped language may also be pre-
sent. Yet, the terms “nonverbal” and “minimally verbal”
do seem to suggest two different states or degrees of
impaired speech, such that nonverbal individuals do not
use any words at all, while “minimally verbal” individuals
have a certain, although very small, amount of speech
(Tager-Flusberg and Kasari, 2013).
Another way to classify the verbal ability of children
with ASD was proposed by Tager-Flusberg et al. (2009)
who suggested the use of benchmark criteria to categorize
the child’s expressive language into different develop-
mental levels using recommended assessment methods.
For example, a child must have reached an expressive
age equivalent of >15 months within the domains of
vocabulary and pragmatics before one can assume that it
has left the first phase, “preverbal communication,” and
moved into the second phase, “the first words,” and have
reached an expressive age equivalent of >24 months
within the domains of vocabulary, grammar, and prag-
matics before one can assume that the child has moved
into the third phase of language development, that is,
“combination of words.”
Currently, it is not entirely clear why many children
with ASD remain nonverbal or minimally verbal even
after having received intervention (Kasari et al., 2013;
Tager-Flusberg and Kasari, 2013), but it has been sug-
gested that several factors probably interact and that these
factors may have different importance for different indi-
viduals (Tager-Flusberg et al., 2005). Numerous studies
have shown a positive relationship between general cogni-
tive ability and language development. Children in the
ASD population who have higher IQs generally exhibit
better language skills (Charman et al., 2003; Kjelgaard and
Tager-Flusberg, 2001; Kjellmer et al., 2012a; Luyster
et al., 2008, 2007), but the linguistic heterogeneity within
the ASD population may not be entirely explained by dif-
ferences in IQ (Chan et al., 2005; Kjelgaard and Tager-
Flusberg, 2001; Thurm et al., 2007). Indeed, the majority
of children with ASD who remain nonverbal at later pre-
school age may show low cognitive ability, but some may
have worse language ability than would be expected from
their nonverbal mental age (Lord et al., 2004). Children
with ASD and a medical/genetic condition, including epi-
lepsy, as well as children with ASD and a history of devel-
opmental regression tend to have poorer cognitive and
 936
Table 1. Number of children and mean age divided by gender
and age group.
Gender Age Total
4 years 5 years 6 years
Girls 24 (4:7) 0 0 24 (4:7)
Boys 18 (4:8) 74 (5:6) 49 (6:4) 141 (5:8)
Total 42 (4:7) 74 (5:6) 49 (6:4) 165 (5:6)
adaptive outcomes (Bernabei et al., 2007; Eriksson et al.,
2013), which likely includes poorer language outcomes.
With regard to the relationship between language and type
of ASD, children with a diagnosis of PDD-NOS generally
show better language ability than those with autistic disor-
der (e.g. Anderson et al., 2007; Thurm et al., 2007). This
may hold true also when controlling for nonverbal mental
age between these two diagnostic groups (e.g. Luyster
et al., 2007) as well as when examining children with ASD
without intellectual disability, controlling for general cog-
nitive level (Kjellmer et al., 2012b).
The main objective of this study was to analyze the pro-
portion of nonverbal or minimally verbal children in a
population-based community sample of preschoolers with
ASD, who had received early intervention and were
assessed at a 2-year follow-up. In addition, we wanted to
investigate the relation between verbal ability and cogni-
tive level, type of ASD, severity of core autism symptoms,
developmental regression, associated medical disorders,
and type of intervention given.
Subjects and methods
Subjects
Participants in this study were 165 children with ASD who
had been recruited from a population-based community
sample. At the time of the study, they were 4 to 6.5 years
old. The original community sample consisted of all 313
children, born between 2002 and 2006, who had been diag-
nosed with ASD before the age of 4.5 years in the Stockholm
County (28,000 births per year). Of these 313 children, 288
had been referred to the Autism Center for Young Children
(ACYC) to receive interventions. The remaining 25 had
been referred to their local habilitation center, mainly due
to more complex medical needs. Of the 288 children, 24
had been referred to the center prior to study start and could
not be included. Of the remaining 264 children, 37 parents
declined participation, 15 could not communicate in
Swedish or English, 2 families moved abroad, and another
2 children had been referred back to their local habilitation
center due to their complex medical needs. The remaining
208 children were assessed and followed over a 2-year
period by a research team at the ACYC. The cohort is
described in detail in Fernell et al. (2010, 2011). At the
ACYC, the children received intervention based on Applied
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Autism 19(8)
Table 2. Distribution and proportions of cohort by cognitive/
developmental quotient (IQ/DQ).
IQ/DQ classification N
⩾70 81 (49.1%)
50–69 27 (16.4%)
<50 52 (31.5%)
<70a 5 (3.0%)
Total 165 (100%)
aThe assessment had been done by external psychologist and detailed
data regarding the cognitive level were not available.
Behavior Analysis (ABA) at different levels of intensity
(Fernell et al., 2011). Research data were collected at
ACYC intake (Time 1) and at follow-up 2 years later (Time
2). At Time 2, all families were invited for a comprehensive
follow-up assessment by the clinical research team consist-
ing of physicians (n = 4), psychologists (n = 2), and speech-
language pathologists (n = 2), who were blind to the type
and extent of intervention the child had received. Of the
208 children, 198 participated in the follow-up assessment
(Fernell et al., 2011). In all, 33 children were excluded: 21
were found not to meet full criteria for ASD, 1 family
declined reassessment of their child’s ASD, 8 were younger
than 4 years of age at follow-up (only children at or above
the age of 4 years were included), and for 3 children data
were incomplete. The remaining 165 children, thus drawn
from a population-based community sample of children
diagnosed with ASD, were included in the study. The distri-
bution of ASD diagnoses in the study cohort was as fol-
lows: 97 children with autistic syndrome (58.8%), 56
children with PDD-NOS (33.9%), and 12 children with
Asperger’s syndrome (7.3%). In Tables 1 and 2, the distri-
butions of age, gender, and developmental status are shown.
Methods
Classification of speech. At follow-up, all children were
assessed by the parent interview Vineland Adaptive
Behavior Scales-II (VABS-II) administered by a medical
doctor, a psychologist, or a speech-language pathologist
(Sparrow et al., 2005). Data from the VABS expressive
sub-domain were used for classification of speech (based
on individual items within as well as the age equivalent of
this sub-domain). Three subgroups were defined: (1) non-
verbal: using fewer than three words and an expressive
age equivalent corresponding to below 15 months; (2)
minimally verbal: using at least three words but never or
only sometimes/partially two-word phrases and an expres-
sive age equivalent corresponding to below 24 months;
and (3) phrase speech: using two-word phrases and an
expressive age equivalent at or above 24 months. In some
of the analyses, the nonverbal and the minimally verbal
groups were collapsed into one and contrasted with the
phrase speech subgroup.
 Norrelgen et al.
Assessment of cognitive level, type of ASD, and severity of core
autism symptoms. To determine the child’s intellectual/
developmental quotient (IQ/DQ) at follow-up, all children
in the study group had been tested by a psychologist using
the Wechsler Preschool and Primary Scale of Intelligence
(WPPSI-III; Wechsler, 2005) or the Griffiths’ develop-
mental scales (Alin-Åkerman and Nordberg, 1991) in
those who could not take the WPPSI-III. The children’s
IQ/DQ status was classified into three categories: ⩾70,
50–69, and <50. In addition, performance IQ data from
WPPSI-III or from the Griffiths’ developmental scales
were used. Performance IQ data from the Griffiths’ were
extracted and converted from Scale D (Eye and Hand
Coordination) and Scale E (Performance; Engman et al.,
2008). The children’s performance IQ status was classified
into three categories: ⩾70, 50–69, and <50. In the statisti-
cal analyses that involved IQ/DQ and performance IQ, the
continuous scores were used.
Assessment of the child’s type of ASD at follow-up was
based on parent interview using the Diagnostic Interview
of Social and Communication Disorders (DISCO-10;
Nygren et al., 2009; Wing et al., 2002), administrated by
medical doctor, psychologist, or speech-language patholo-
gist, in combination with other compiled clinical data. A
classification of ASD type was then performed according
to DSM-IV (APA, 1994) by a medical doctor, psycholo-
gist, and speech-language pathologist. Additional informa-
tion about the severity of core autism symptoms was
obtained by interviewing the parents according to the
Autistic Behavior Checklist (ABC; Krug et al., 1980).
Developmental regression. Information regarding develop-
mental regression was obtained from the parent interview
at the ACYC and records from Child Health Centers
(CHC; Eriksson et al., 2013). Parents were interviewed by
a medical doctor regarding a history of regression in the
child and this information was compared to available data
from CHC and medical records. Consistency was required
between parental information and the notes in the records
from CHC. Regression was defined as loss of more than
five spoken words used communicatively in children more
than 15 months of age. In children younger than 15 months,
regression was determined when there was a clear indica-
tion of loss of social interest and contact.
Medical examination, including assessment of epilepsy and of
cerebral palsy. Each child had a medical/neurological
examination by a medical doctor with regard to medical
conditions, including epilepsy. Medical and genetic condi-
tions were defined as (1) significant intrauterine harmful
exposure, (2) severe prematurity (gestational age less than
29 weeks), (3) identified genetic disorder including signifi-
cant genomic imbalances identified with array compara-
tive genomic hybridization (CGH), (4) abnormal brain
magnetic resonance imaging (MRI) findings, and/or (5)
clinical diagnosis of epilepsy.
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937
Behavioral intervention. Type of intervention that the child
had received over the 2-year observation period was cate-
gorized into one of two groups: (1) Intensive intervention
based on ABA given at the preschool and by the parents at
home with assistance from the ACYC, for at least 15 h and
up to 40 h per week, or (2) non-intensive targeted interven-
tion based on ABA, consisting of different targeted types of
training, for example, toilet training, speech and language
training, training of compliance, or other specific training
that the child was deemed to need (see Fernell et al., 2011).
Statistical analyses. To analyze the differences of overall IQ
scores, performance IQ scores, age, and ABC scores in
relation to speech classification, ANOVAs were per-
formed. For post hoc analyses, Fisher’s least significant
difference (LSD) was used with a significance level of
p < 0.05. Age effects in those analyses were controlled for
by separate analyses of covariance (ANCOVAs) with age
as covariate. To assess the influence of the dichotomous
variables, developmental regression and medical condition
odds ratios and their 95% confidence intervals (CIs) were
calculated by logistic regression analyses.
Ethics. The study was approved by the ethics committee at
Karolinska University Hospital, Stockholm.
Results
Overall distribution by speech classification and
by general cognitive level
In all, 25 children (5 girls and 20 boys; i.e. 15% of the 165
children) were classified as nonverbal. In addition, 17
children (1 girl and 16 boys; i.e. 10%) were classified as
being minimally verbal. The remaining 123 children (18
girls and 105 boys; i.e. 75%) had phrase speech (see Table
3). Of the 165 children, 84 (51%) had IQ/DQ <70, 46
(28%) between 70 and 84, and 35 (21%) had IQ/DQ ⩾85.
All children classified as being nonverbal or minimally
verbal had IQ/DQ below 70. Between-group analysis
of variance (ANOVA) based on IQ/DQ continuous
scores and speech classification was significant
(F(2, 155) = 92.25, p < 0.001), and a post hoc analysis
showed that the differences in IQ/DQ scores were signifi-
cant between all of the three speech groups (p < 0.05). The
distribution of IQ/DQ in relation to verbal classification is
shown in Table 3.
Distribution by speech classification and
performance IQ level
Performance IQ data were available for 40 of the 42 chil-
dren who were classified as nonverbal or minimally ver-
bal. Of these 40 children, 2 (5%) had a performance
IQ >70 and the remaining 38 children had a perfor
mance IQ <70. A between-group ANOVA based on
 938 Autism 19(8)

Table 3. Distribution and proportions of the cohort by cognitive/developmental level (IQ/DQ) and by performance IQ level
divided by speech classification.

Speech classification Total

Nonverbal Minimally verbal Phrase speech

IQ/DQ classification
⩾70 0 (0.0%) 0 (0.0%) 81 (49.1%) 81 (49.1%)
50–69 0 (0.0%) 2 (1.2%) 25 (15.2%) 27 (16.4%)
<50 24 (14.5%) 15 (9.1%) 13 (7.9%) 52 (31.5%)
<70a 1 (0.6%) 0 (0.0%) 4 (2.4%) 5 (3.0%)
Performance IQ classification
⩾70 0 (0.0%) 2 (1.2%) 92 (55.8%) 94 (57.0%)
50–69 4 (2.4%) 3 (1.8%) 16 (9.7%) 23 (13.9%)
<50 19 (11.5%) 12 (7.3%) 10 (6.1%) 41 (24.9%)
Missing data 2 (1.2%) 0 (0.0%) 5 (3.0%) 7 (4.2%)
Total 25 (15.2%) 17 (10.3%) 123 (74.5%) 165 (100%)
aThe assessment had been done by external psychologist and detailed data regarding the cognitive level were not available.

Table 4. Distribution and proportions of the cohort by speech classification and ASD diagnosis.

ASD diagnosis Speech classification Total

Nonverbal Minimally verbal Phrase speech

Autistic syndrome 24 (24.7%) 16 (16.5%) 57 (58.8%) 97 (100%)
PDD-NOS 1 (1.8%) 1 (1.8%) 54 (96.4%) 56 (100%)
Asperger’s syndrome 0 (0.0%) 0 (0.0%) 12 (100.0%) 12 (100%)
Total 25 (15.2%) 17 (10.3%) 123 (74.5%) 165 (100%)

ASD: autism spectrum disorders; PDD-NOS: pervasive developmental disorder—not otherwise specified.

performance IQ continuous scores and speech classifica-
tion was significant (F(2, 155) = 70.4, p < 0.001). A post
hoc analysis revealed that the performance IQ scores
were significantly different between all three speech
groups (p < 0.05). The distribution of performance IQ in
relation to verbal classification is displayed in Table 3.
Age distribution in relation to speech
classification
Age distribution differed between the verbal (phrase
speech), nonverbal, and minimally verbal groups (F(2,
162) = 5.16, p < 0.01). Pairwise comparisons (Fisher’s
LSD) showed that the mean age was higher in the group
with phrase speech compared with both the group of non-
verbal and the group of minimally verbal children
(p < 0.05).
Since there were age differences between the speech
classification groups, we repeated the two between-group
ANOVAs of speech classification in relation to IQ/DQ
scores and performance IQ scores presented above with
age as a covariate to analyze for potential interaction
effects. Neither of these ANCOVAs were significant,
p = 0.86 and p = 0.75, respectively.
Distribution by ASD diagnosis and speech
classification
Of the total group of 165 children, 97 had autistic disor-
der, 56 had PDD-NOS, and 12 had Asperger’s syndrome.
Of the 42 children, 40 (95%) who were nonverbal or
minimally verbal had autistic disorder and 2 had PDD-
NOS. Of the 123 children with phrase speech, 57 had
autistic disorder (46%), 54 had PDD-NOS (44%), and 12
had Asperger’s syndrome (10%). These results are shown
in Table 4.
ABC data in relation to speech classification
Children with phrase speech had fewer core autism symp-
toms (ABC) than those who were nonverbal or minimally
verbal (F(2, 157) = 12.12, p < 0.001). Pairwise compari-
sons (Fisher’s LSD) showed that there was no significant
difference between the ABC scores of the nonverbal and
the minimally verbal groups of children but between both
these groups and the group with phrase speech (p < 0.05).
To control for potential interaction effects of age in these
analyses, an ANCOVA with age as a covariate was per-
formed (p = 0.39).

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 Norrelgen et al.
Developmental regression
Of the 165 children, 39 had developmental regression
(24%). Among the 42 children who were nonverbal or
minimally verbal, 19 had had a reported regression (45%).
The corresponding rate among the 123 with phrase speech
was 18% (20/123); odds ratio = 4.25, 95% CI = 1.96–9.22,
p < 0.001). Conversely, 49% (19/39) of children with and
18% (23/126) of those without developmental regression
were minimally verbal or nonverbal (p < 0.001).
Medical conditions including epilepsy
A known medical condition including epilepsy was identi-
fied in 24% of children who were either nonverbal or mini-
mally verbal and in 17% of children with phrase speech
(n.s.). Given that all nonverbal and minimally verbal chil-
dren had an IQ/DQ below 70, we were also interested in
comparing these proportions in the group with IQ/DQ <70
and with phrase speech. This analysis revealed that of the
44 children with IQ/DQ below 70 but with phrase speech, 8
(19%) had a known medical condition (n.s.). No child had
cerebral palsy or other causes of oral motor difficulties.
Type of intervention and expressive speech ability
Of the 42 nonverbal or minimally verbal children, 22
(52%) had received intensive intervention and 20 (48%)
targeted intervention (Fernell et al., 2011). The corre-
sponding rates among those with phrase speech were 58
(47%) and 65 (53%), respectively.
Discussion
This study is the first in recent years to use a population-
based community sample in order to estimate the propor-
tion of children with ASD who at late preschool age have
not developed phrase speech. Our data indicated that about
one in four of the children were nonverbal or only mini-
mally verbal. In agreement with Lord et al. (2004) and
Anderson et al. (2007), we therefore conclude that nowa-
days considerably fewer than 50% of children with ASD—
defined according to DSM-IV—remain nonverbal or
minimally verbal at the age of starting school, regardless
of whether a stricter or a slightly broader definition of
“nonverbal” (i.e. including children that we here have
defined as being minimally verbal) is used.
In closer comparison, 14%–20% of the children in the
Lord et al. (2004) study and 20% in the Anderson et al.
(2007) study were classified as nonverbal based on stricter
criteria of using no or very few consistent words. An addi-
tional 11%–14% (Lord et al., 2004) and 19% (Anderson
et al., 2007) of the children in respective study only exhib-
ited minimal amounts of speech using words but not
sentences. In our study, 15% were judged to be nonverbal
and another 10% as being minimally verbal. When
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939
comparing the results of the three studies, one has to bear
in mind the considerable differences between them. First,
the definitions of nonverbal/minimally verbal were not
identical across studies. In fact, they may not even have
been exactly identical between the Lord et al. (2004) and
Anderson et al. (2007) studies, even though they report
data from the same two cohorts. This reflects the lack of
agreed terms and definitions for nonverbal/minimally ver-
bal in the earlier ASD literature. Second, categorization
was based on results from different assessment instru-
ments. The two previous studies used a combination of
a standardized parent interview (Autism Diagnostic
Interview–Revised (ADI-R)) and direct observation
(Autism Diagnostic Observation Schedule (ADOS)),
whereas this study only used a standardized parent inter-
view instrument (VABS). This difference could in part
contribute to the lower rate of nonverbal/minimally verbal
children identified in this study.
Another important distinction between the studies was
differences in age groups assessed: 4- to 6-year-olds in this
study and 9-year-olds in the Lord and Anderson studies.
Our data showed an age effect, such that the children in
the phrase speech group had a higher mean age than both
the nonverbal and minimally verbal speech groups. The
observed age differences could indicate that some children
with ASD will acquire phrase speech during the late pre-
school years before starting school. Indeed, some children
in our sample are likely preverbal (i.e. they will go on to
develop speech) rather than truly nonverbal (i.e. they will
not develop speech), an important but difficult to define
distinction (Tager-Flusberg and Kasari, 2013). Moreover,
the children in our study will probably receive continued
communication-enhancing interventions of some kind
and, with or without such treatment, a few more of them
will possibly develop phrase speech during the school
years. Such a positive language development after pre-
school age for some children with ASD is in line with the
findings of Pickett et al. (2009). However, since our sam-
ple was drawn from the community population of all chil-
dren aged 22–54 months diagnosed with ASD in Stockholm
County during a certain time period, the age differences
found could also, hypothetically, be a reflection of the pos-
sibility that children who have been diagnosed with ASD
very early in life have more severe problems than those
who are detected later. A previous study of the cohort
(from which the sample of this study was drawn) found
that treatment outcome was significantly poorer for chil-
dren who had been referred at an earlier age compared to
those who had been referred at a later age (Eriksson et al.,
2013). In addition, there were no interaction effects of age
in the between-group analyses of IQ/DQ and performance
IQ, respectively, in this study.
Furthermore, the sample in the study by Lord et al.
(2004) was not representative of the ASD population since
it also included a group of children who did not meet
 940
diagnostic criteria for ASD although all children had been
referred for possible autism at the age of 2 years. In the
study by Anderson et al. (2007), data for the ASD groups
were presented separately; yet, the sample was not com-
pletely community based. In this study, participants were
children aged 4–6.5 years sampled from a complete com-
munity based population who had been diagnosed with
ASD before or at the age of 4.5 years (the representative-
ness of our sample will be discussed in further detail
below). In addition, the two cohorts in the Lord and
Anderson studies did not include any children with a diag-
nosis of Asperger’s syndrome and our sample included
only a few. Both children with Asperger’s syndrome who
are commonly diagnosed at school age (Coo et al., 2012) as
well as other children diagnosed with ASD at a later age are
likely to be verbal. Thus, the total proportion of nonverbal/
minimally verbal children with ASD is, in all probability,
even smaller than found both in the current and in the Lord
and Anderson studies if one were to assess a group repre-
sentative of older school-age children with ASD.
Previous research has clearly shown that early expres-
sive language ability is strongly linked to later social and
adaptive skills, academic success, and independence in
adulthood (Gillberg, 1991; Lord et al., 2004; Venter et al.,
1992). In general, individuals within the autism spectrum
with an IQ above 50 at initial diagnosis, who have devel-
oped communicative speech before the age of 6 years,
have better prospects of achieving social and academic
success (Gillberg and Steffenburg, 1987). In our study, all
children classified as being nonverbal or minimally verbal
had intellectual disability, the vast majority of whom are in
the severe range. A similar pattern was seen with regard to
performance IQ, such that only 2 of 40 children (5%) clas-
sified as nonverbal or minimally verbal had a performance
IQ score >70. Negative prognostic factors have been
linked to low general cognitive function combined with
low level of communicative phrase speech at the age of
6 years (Billstedt et al., 2005). The findings highlight the
importance of assessing expressive language skills in addi-
tion to IQ/DQ in preschool children with ASD.
Children with a history of regression significantly
more often were nonverbal or minimally verbal compared
to children without regression. In fact, about half the
group with regression had no phrase speech, whereas this
applied in fewer than one in five of those without regres-
sion. The subgroup with regression in the sample is cur-
rently the subject of a detailed analysis of comprehensive
clinical and etiological background. It is already clear that
this group with ASD, regression, and lack of phrase
speech is among the most severely disabled of all within
the autism spectrum.
With regard to type of ASD, almost all of the 42 children
in our study classified as being nonverbal or minimally ver-
bal had autistic disorder, 2 had PDD-NOS, and none had a
diagnosis of Asperger’s syndrome. Our findings are in
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Autism 19(8)
keeping with, for example, Thurm et al. (2007) and
Anderson et al. (2007). In addition, the severity of core
autism symptoms was also found to be higher in the non-
verbal/minimally verbal speech group of this study.
We did not find any correlation between identified
medical disorders and a child being nonverbal or mini-
mally verbal. The rate of identified genetic and other medi-
cal disorders was about 20% in the total group, and in
many children, despite severe autism combined with ID,
no etiology had been revealed at the time of this study.
Reviews of studies of early intensive behavioral inter-
vention (EIBI) have shown considerable variability in out-
come and that the child’s IQ is an important factor for
prognosis (Howlin et al., 2009). Importantly, our study
was not a randomized controlled trial designed to assess
the effects of EIBI, but our results, nevertheless, are inter-
esting and we think worth mentioning. Our analyses
revealed that in the group of children who were nonverbal
or minimally verbal as well as in the group with phrase
speech, the distribution was almost even between those
who had received EIBI for 15–40 h/week and those who
had received non-intensive, problem-targeted intervention
during the 2 years at the habilitation center (for more
details about treatment protocols, see Fernell et al. (2011)).
The focus of EIBI mainly involves training of language,
communication, play, and social skills. Therefore, an
expected result would have been that the proportion of
children classified as nonverbal or minimally verbal would
be smaller in the EIBI group than in the non-intensive
group after 2 years of intervention (which was not the
case). However, in a previous study of treatment effects in
the whole cohort (Fernell et al., 2011), no significant dif-
ference between the intensive and non-intensive groups
was found with regard to adaptive outcome.
A limitation of this study is that language assessment
was based on parent report rather than on formal testing or
a combination of both. Assessing the language of mini-
mally verbal children with ASD brings about challenges,
which has been discussed by several researchers (e.g.
Kasari et al., 2013; Tager-Flusberg et al., 2009). The num-
ber of words the child uses on a daily basis (according to
responses from semi-structured parent interviews) and lin-
guistic age equivalents (measured with formal, standard-
ized language test) appear to be the most common ways to
measure the expressive ability in order to distinguish ver-
bal from more or less nonverbal children with ASD and
there are documented pros and cons of both. When the
child is at an early linguistic stage, it is sometimes difficult
to make use of formal testing, and therefore, parent reports
are often used as a supplement or an alternative (Charman
et al., 2003). The formal test situation is generally new and
unnatural to the child, risking a non-representative perfor-
mance. A disadvantage of parent reports is a risk of parents
overestimating their child’s abilities; however, it has
been found that primarily receptive abilities tend to be
 Norrelgen et al.
overestimated since understanding of contextual cues
(such as gestures and everyday routines) often can be mis-
interpreted as language comprehension (Tomasello and
Mervis, 1994). Parent assessments of expressive language,
on the other hand, have been found to correspond well
with the results of standardized tests (Charman et al., 2003;
Luyster et al., 2008). Thus, although it is not entirely pos-
sible to conclude whether formal language testing of chil-
dren at an early linguistic stage provides more accurate
results than parent report, these findings suggest that
responses from parental interviews are a valid and reliable
measure when the objective is to identify severe limita-
tions in expressive speech and language. Importantly,
researchers today recommend a much more comprehen-
sive assessment of language than simply administrating
the VABS, or any other parent report measure, when inves-
tigating nonverbal/minimally verbal children with ASD
(e.g. Kasari et al., 2013).
The conclusions that can be drawn from this study with
regard to the representativeness of the data need to be con-
sidered. We obtained data of 165 children out of the origi-
nal cohort of 313 children born between 2002 and 2006,
who had been diagnosed with ASD before the age of
4.5 years in the Stockholm County (Fernell et al., 2010).
Thus, the number of children who did not take part in the
study was 148. There is reason to assume that the 25 chil-
dren who had been referred to other habilitation centers,
mainly because of complex medical needs, represent dif-
ferent characteristics than the remainder of the cohort.
However, there are no indications that the characteristics
of the other children who did not participate in the study
would be substantially different than those who did partici-
pate. The causes for not participating at the study start or at
the follow-up are detailed in the “Methods” section. The
cohorts in the studies by Lord et al. (2004) and Anderson
et al. (2007) were not completely population based, con-
sisted of children diagnosed before the age of 2 years, and
included children who did not meet diagnostic criteria for
ASD (although data were presented separately for the dif-
ferent groups in the Anderson study). The cohort in this
study was based on a community sample of all children in
the Stockholm County who had been diagnosed with ASD
(based on DSM-IV) before or at the age of 4.5 years and
included children across the IQ range, across ASD diag-
nostic classifications as well as children with different
medical conditions. Taken together, these aspects imply
that the cohort in this study may have a higher representa-
tive value than earlier studies. Nevertheless, it is important
to keep the mentioned limitations in mind when consider-
ing the generalizability of the results in this study.
In conclusion, this study showed that the proportion of
children with ASD diagnosed at an early age who had not
developed phrase speech during the preschool years was
about 25% and clearly most important factor linked to ver-
bal ability was the child’s general cognitive level such that
all children classified as nonverbal or minimally verbal
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Rafaela Rossini Rosa - rafarossini@hotmail.com - IP: 177.22.171.178
941
also had an intellectual disability. Nonverbal IQ also had a
strong impact since 95% of the children who had not
developed phrase speech scored ⩽70. “Regressive autism”
constituted a large proportion of all those classified as non-
verbal or minimally verbal. Future studies with representa-
tive samples and preferably longitudinal designs are
needed to further investigate how many children with ASD
remain without phrase speech at school age. Perhaps the
most important focus in such studies would be early iden-
tification of which children are at risk of not developing
phrase speech and to determine what type of intervention
would best benefit those children.
Funding
This research received no specific grant from any funding agency
in the public, commercial, or not-for-profit sectors.
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