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Attention in selective mutism—An exploratory case-control study
Beate Oerbeck *, Hanne Kristensen
Centre for Child and Adolescent Mental Health, Eastern and Southern Norway, Norway
Received 11 December 2006; received in revised form 13 April 2007; accepted 20 April 2007
Abstract
The aim of the study was to explore the association between selective mutism (SM) and attention. In SM social anxiety seems
central but language impairment and motor problems are also reported. Attention problems have been described in parental
behavioral ratings, while neuropsychological studies are lacking. A neuropsychological test (the Trail Making Test) and parental
ratings of attention- and anxiety problems were administered to a clinical sample of 23 children with SM (aged 7–16 years, 12 boys
and 11 girls) and 46 non-referred matched controls.
The SM group differed from controls on the Trail Making Test, but the group difference disappeared, when controlling for motor
function and IQ. Parental ratings of attention problems were not significantly associated with the neuropsychological attention
measure. Neuropsychological studies of attention controlled for IQ and motor function are needed as well as tests that measure
different aspects of attention.
# 2007 Elsevier Ltd. All rights reserved.
Keywords: Neuropsychology; Attention; Anxiety; Child; Selective mutism
Selective mutism (SM) is a childhood condition
characterized by a persistent lack of speech in certain
situations despite the ability to comprehend and use
language. Children with SM usually speak to family
members at home, but fail to speak in kindergarten or at
school (American Psychiatric Association, 1994). The
etiology of SM is still unclear but most probably SM
represents a heterogeneous condition. SM is frequently
associated with social anxiety both in children (Black &
Uhde, 1995) and in adults (Steinhausen, Wachter,
Laimbock, & Metzke, 2006) and it has been argued that
SM represents a form of social anxiety disorder (SAD)
(Anstendig, 1999; Sharp, Sherman, & Gross, 2007).
However, the relationship between SM and SAD is still
under debate (Yeganeh, Beidel, Turner, Pina, &
Silverman, 2003).
SM is also associated with neurodevelopmental
disorder/delay (Kristensen, 2000) in both language and
motor function. Whether children with SM may suffer
from subtle attention deficits is less studied. To explore
the possibility of various neuropsychological impair-
ments in SM may throw light on the etiology of the
disorder and consequently have implications for
treatment. Naturally, due to the mutism, language
function has been most frequently studied in children
with SM, and language impairment has been consis-
tently reported in clinical studies (McInnes, Fung,
Manassis, Fiksenbaum, & Tannock, 2004; Steinhausen
& Juzi, 1996). However, language measures applied in
SM studies may have required visuo-motor abilities as
well (Manassis et al., 2003). Thus, in a former study we
Journal of Anxiety Disorders 22 (2008) 548–554
* Corresponding author at: Centre for Child and Adolescent Mental
Health, Eastern and Southern Norway, P.O. Box 4623, Nydalen, N-
0405 Oslo, Norway. Tel.: +47 22586000; fax: +47 22586001.
E-mail address: beate.orbeck@r-bup.no (B. Oerbeck).
0887-6185/$ – see front matter # 2007 Elsevier Ltd. All rights reserved.
doi:10.1016/j.janxdis.2007.04.008
explored aspects of nonverbal cognition and found that
children with SM did not differ from controls on tests of
visual memory span and visual memory, while verbal
memory span was reduced (Kristensen & Oerbeck,
2006).
Motor deviance/delay is reported in studies including
larger samples of children with SM (Kolvin & Fundudis,
1981; Steinhausen & Juzi, 1996) both with and without
comorbid language disorder (Kristensen, 2002).
Impaired motor function represents an important co-
morbidity in language disorders without SM (Webster,
Majnemer, Platt, & Shevell, 2005). In addition to motor
impairment, language disorders are also associated with
attention deficits and social difficulties (Snowling,
Bishop, Stothard, Chipchase, & Kaplan, 2006). Whether
children with SM may have subtle attention deficits is
thus animportantquestiontoexplore. Shyness,whichisa
hallmark of SM, has also been reported to co-vary with
attention problems in normal girls (Caspi & Silva, 1995).
Likewise, attention problems in early childhood years
significantly predicted symptoms of anxiety and depres-
sion in a large cohort study of preadolescent boys and
girls (Leech, Larkby, Day, & Day, 2006).
With regard to attention problems in SM, parental
behavioral ratings suggesting attention deficits have
been reported (Kristensen, 2001; Steinhausen & Juzi,
1996). To our knowledge, neuropsychological test
results from case-control studies have not been
published, and it is not known whether behavioral
ratings of attention deficits and anxiety co-vary in this
patient group. However, a study including two twin
pairs with SM revealed an attentional problem in three
of the four twins on a continuous performance test
(Gray, Jordan, Ziegler, & Livingston, 2002).
In this article, we wanted to study the neuropsycho-
logical aspect of attention in a group of SM and matched
controls and to examine whether motor function,
performance IQ, and a diagnosis of language disorder
and/or anxiety disorder were important predictors of
performance on the attention test. We also wanted to
explore whether the parental behavioral ratings co-
varied with neuropsychological measures.
1. Methods
1.1. Subjects
The index group consisted of 23 children with SM, 12
boys and 11 girls, mean age11.6 years (S.D. 2.7, range 7–
16 years) while the control group comprised 24 boys and
22 girls, mean age 11.8 years (S.D. 2.6, range 7–16
years). The present study sample consists of children
from age 7 years who were able to complete The Trail
Making Test and had CBCL parental behavioral ratings
(SM group: n = 23; control group: n = 46) from a larger
sample of referred children with SM (age range 4–16
years, N = 54 and 108 matched controls). The children
with SM were recruited nation-wide from out-patient
clinics and school psychology services. The control
groupconsisted of children whowere asked to participate
by the teachers of the children with SM. For each child
with SM, two non-referred children (without SM)
matched for gender, age, geographical site and SES
were recruited. SES was assessed by the occupations of
the parents and rated in accordance with the guidelines of
the governmental statistics Norway and collapsed into
four classes ranging from: 1 = upper status unto
4 = lower status, with no significant group differences
[x2
(3,69) = 2.1, ns]. Details on the recruitment and SES
procedures are described in a previous paper (Kristensen,
2000). As one of the matched controls failed to complete
the most complex part of the Trail Making Test (part B),
the number of controls was reduced from 46 to 45 on this
subtest. The parents of one matched control did not fill
outthe Child Behavior Checklist,hence N was reduced to
45 on this measure.
1.2. Background assessment and present measures
1.2.1. Diagnostic procedure
A DSM-IV diagnosis of SM (APA, 1994) was
assigned using the following procedure:
1. Discussion of the SM symptoms with the referring
therapists. 2. A structured interview with parents and
teachers on SM symptoms (Cline & Baldwin, 1994). 3.
Direct observation.
1.2.2. Assessment of intelligence, motor function
and behavior problems, and whether a diagnosis of
a language disorder and/or an anxiety disorder was
present
In addition to the neuropsychological test presented
in this paper (the Trail Making Test), the examination
included assessment of nonverbal IQ, as measured by
performance IQ (PIQ) and motor function. PIQ was
assessed by the Wechsler Scale of Intelligence-Revised
(WISC-R; Wechsler, 1992). For further details con-
cerning the WISC-R, see Kristensen (2000).
Fine and gross motor function was evaluated by sets
of age-dependent motor items designed for the study
(6–7 years: 8 items; 8 years: 12 items; 9–16 years: 13
items). The sets of items were chosen from three
different motor assessment batteries (Gillberg, 1995;
Oseretsky, 1936; Touwen, 1979). Each item was scored
B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554 549
on a scale from 1 to 4. (1: failing the test; 2: poor
performance or not at expected age level; 3: medium
performance at expected age level; 4: good performance
at expected age level). Each child was given a total
motor score (sum score  10 divided by number of
items) with a maximum score of 40. To give an
example: For age 6 through 7 years these items were
included: Pencil grip, cutting a paper circle, diadocho-
kinesis, sequential opposition of thumb to same-hand
fingers, standing on one leg, jumping on one foot,
walking on lateral aspects of feet, and balancing along a
line. For more information about the motor tests
applied, see Kristensen (2002). The mothers filled out
the Achenbach Child Behavior Check List; CBCL
(Achenbach, 1991). We present data from two CBCL
syndrome scale scores related to anxiety (anxious/
depressed and withdrawn) and the attention syndrome
scale. Parents rate their child for how true each item is
now or within the past 6 months using the following
scale: 0 = not true (as far as you know); 1 = somewhat
or sometimes true; 2 = very true or often true. Items are
summed up to form the syndrome scales, and these raw
scores are reported. For further details concerning the
CBCL, see Kristensen (2001).
Table 1 present the performance IQ, the motor score,
and the CBCL scores of the two groups.
The children were diagnosed as to whether or not
they had a language disorder using different tests, due to
a large age span of the total sample and varying
response modes in the SM group. Tests used for the
language assessment included the verbal IQ of the
WISC-R (Wechsler, 1992), the Boston Naming Test
(Kaplan, Goodglass, & Weintraub, 1983), the Reynell
Scales (Reynell, 1977) the Peabody Picture Vocabulary
Test-revised (Dunn & Dunn, 1981) and audiotapes of a
conversation at home. Eleven out of 23 SM patients
(48%) were diagnosed with a language disorder, in
contrast to 7 of the 46 controls (15%), [x2
(1,69) = 8.46,
p = .004]. The possible presence of an anxiety disorder
was assessed by the parent version of the Child
Assessment Schedule (CAS; Hodges, Kline, & Stern,
1982) adjusted according to DSM-IV criteria [regarding
separation anxiety, social phobia, specific phobia,
overanxious disorder (generalized disorder in DSM-
IV), and obsessive-compulsive disorder]. Nineteen out
of 23 SM patients (83%) fulfilled the criteria of an
anxiety disorder, in contrast to four of the 46 controls
(7%) [x2
(1,69) = 37.67, p < .001].
1.2.3. Neuropsychological measure of attention
Attention was measured using the child version of
the Trail Making Test, with number of seconds to
complete the test as our test variable (Reitan, 1971). The
Trail Making Test require in the first and simpler part A
that a line is drawn from digit 1 through 15 on a piece of
paper where the numbers are scattered within circles. In
the more complex part B, the subject is instructed to
draw the lines in sequential shifts between digits and
letters (; 1 – A, A – 2, 2 – B and so forth up to 8 – H). The
Trail Making Test provides information regarding
attention and visual scanning along with speed of both
eye–hand coordination and information processing, and
it is sensitive to cerebral dysfunction (Reitan & Wolfson,
1985). Studies have reported associations with motor
function and IQ scores, and part B can also be seen as a
measure of executive functioning as it requires alternate
shifts between sets of stimuli; (Lezak, Howieson, &
Loring, 2004; Mitrushina, Boone, & Delia, 1999; Spreen
& Strauss, 1998; Warner et al., 2006).
1.3. Procedure
One examiner (the second author) assessed all the
children. The participating families were asked to
choose the location where they thought the child would
feel most comfortable. The children were left to choose
whether or not one of the parents should be present
during the assessment. In the SM group, one child was
assessed at home, nine at school and 13 at their out-
patient clinic, while the control children were mainly
seen at school. All participants received the different
tests in the same order.
B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554550
Table 1
Performance IQ, motor scores and CBCL subscale raw scores in children with SM and controls
SM (n = 23) mean (S.D.) Controls (n = 46) mean (S.D.) t-test
Wechsler performance IQ 96.39 (18.34) 108.35 (14.74) t = À2.93, p = .01
Motor function score 31.43 (4.68) 37.15 (3.18) t = À5.28, p < .001
CBCL withdrawna
6.83 (3.68) 1.0 (1.38) t = 7.34, p < .001
CBCL anxious-depresseda
7.13 (5.93) 1.60 (2.65) t = 4.26, p = .01
CBCL attentiona
3.78 (4.09) 1.20 (1.31) t = 2.95, p = .008
a
Higher score indicates more problems.
1.4. Statistics
The data were analyzed using the Statistical Package
for the Social Sciences; SPSS version 11 (SPSS, 2002).
Means and standard deviations are reported and the
significance level was set to 0.05. Due to skewness, the
Trail Making Test data were log transformed. Inde-
pendent t-tests were used to assess possible group
differences. Linear regression analysis was employed to
study the impact of SM on attention controlled for
motor function, performance IQ, and the presence of
language- or anxiety disorders. We used bivariate
correlation analysis (Pearson) to study whether the
neuropsychological test data and the behavioral rating
data co-varied. The x2
test was used to assess whether
the groups differed significantly in SES and the
presence of deviant scores on the individual CBCL
attention subscale items.
2. Results
2.1. Performance on the Trail Making Test
The SM group differed significantly from the control
group on the Trail Making Test. Mean results (raw
scores reported as seconds to complete the test) and
analysis of group differences with independent t-tests
(using log transformed TMT data) are presented in
Table 2.
However, the group difference disappeared in
multiple regression analysis including IQ and motor
function. Motor function and IQ were both significant
predictors of the TMT A performance, while IQ
significantly predicted the TMT B performance. The
results from the regression analysis are presented in
Table 3.
One could question the use of performance IQ as a
control for IQ in our study, since this score to some
extent is time dependent (better scores with quick
performance), as is the Trail Making Test itself. We
therefore also performed the regression analyses with
another measure related to IQ (the Peabody Picture
Vocabulary Test-revised), that is time independent,
rendering similar results (results not shown).
Analysis of whether or not the presence of a
language disorder or an anxiety disorder influenced the
attention test results by including them in the regression
analysis (using the enter method) proved that none were
significant predictors (all standardized betas <.23, ns).
Analysis of whether or not the presence of the
parental CBCL ratings influenced the attention test
results by including them in the regression analysis
(using the enter method) proved that none were
significant predictors (all standardized betas <.30, ns).
2.2. The relation between neuropsychological
measures and questionnaire data
The neuropsychological measure of attention pro-
blems (the Trail Making Test) did not co-vary with the
parental ratings of problems with attention or anxiety as
measured by the CBCL syndrome scales. However, the
B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554 551
Table 2
Mean results (time to finish in seconds; raw scores) on the Trail Making Test (part A and part B) in children with SM (n = 23) and controls (n = 46)
and results from group comparison using independent t-tests on logtransformed data
SM (n = 23)
mean (S.D.)
Controls (n = 46)
mean (S.D.)
Mean difference (95% CI)
on logtransformed data
t (dfa
) p-value
TMT, part A 26.0 (12.5) 19.9 (7.4) 0.22 (0.002 to 0.45) 2.01 (67) .05
TMT, part B 62.4 (31.8) 43.1 (16.9) 0.31 (0.09 to 0.54) 2.48 (66) .008
a
df: degrees of freedom.
Table 3
Multiple regression analysis (enter method): effects of patient status and performance IQ and motor score on log transformed Trail Making Test (part
A and part B) data
Dependent variable: Trail Making Test, part A Dependent variable: Trail Making Test, part B
Standardized coefficient (b) p-value Standardized coefficient (b) p-value
Patient status .154 .226 À.114 .423
Performance IQ À.356 .002 À.268 .029
Motor score À.463 <.001 À.193 .173
R2
adjusted .320 .170
Significance of model F(3,65) 11.65, p < .001 F(3,64) 5.43, p = .002
CBCL parent reported attention problems were asso-
ciated with the two other neuropsychological measures;
motor problems and reduced nonverbal IQ (Table 4).
The parent reported syndrome scale scores on
attention and anxiety (anxiety/depression and with-
drawn) also correlated significantly (R = .67 and .58,
respectively, p < .001).
The specific items from the CBCL syndrome scale of
attention problems that best differentiate SM from
controls are presented in Table 5.
3. Discussion
3.1. Performance on the Trail Making Test
This is the first case-control study exploring a
neuropsychological measure of attention in children
with SM. Because studies on SM that include
comprehensive neuropsychological assessment with
sufficient samples sizes are lacking, our report on
specific aspects of attentional functioning in SM may be
important. This added knowledge may contribute to the
understanding of possible pathways of symptom
development and to the identification of possible
subgroups of children with SM.
The present study found an attention deficit in SM, as
measured by the Trail Making Test. However, motor
problems and reduced nonverbal IQ were central factors
in explaining this deficit, emphasizing the need to
control for these factors in future measures of attention.
Our results are in line with a study of 38 anxious/
depressed boys (age 9–11 years) who performed the
Trail Making Test part B significantly slower than the
control group (Emerson, Mollet, & Harrison, 2005). No
attempts were made to assess the impact of IQ and
motor function in this study. Hence whether anxiety
actually explained the mentioned deficit, is not known.
3.2. The relation between neuropsychological
measures and questionnaire data
The neuropsychological measure of attention did not
co-vary with the parent reported attention or anxiety
problems. This finding, along with the fact that neither a
language disorder, nor the presence of an anxiety
disorder significantly predicted the Trail Making Test
performance, could suggest that the neuropsychologi-
cally measured attention deficit represent an important
dimension in SM worth further studying. Whether
possible attention deficits will present as a separate
B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554552
Table 4
Correlations between parental ratings (three CBCL syndrome scales) and neuropsychological measures (log-transformed Trail Making Test score,
motor score and Performance IQ)
Parental ratings Neuropsychological measures
CBCL syndrome scales TMT A TMT B Motor score Performance IQ
Attention problems .168, ns .079, ns À.514**
, p < .001 À.253*
, p = .037
Anxiety/depression .054, ns .057, ns À.412**
, p < .001 À.139, ns
Withdrawn .041, ns .068, ns À.485**
, p < .001 À.232, ns
*
p < .05.
**
p < .001.
Table 5
The number and percentage of children with SM (n = 23) and controls (n = 45) with positive item scores (1 = somewhat or sometimes true, 2 = very
true) on the CBCL attention syndrome subscale items
SM (n = 23) N (%) Controls (n = 45) N (%) x2
p-value
Item 1, Acts too young for his age 10 (43) 4 (8) 11.14 .004
Item 45, Nervous 10 (43) 5 (11) 10.42 .005
Item 13, Confused, seems to be in a fog 6 (26) 1 (2) 9.49 .009
Item 62, Poorly coordinated, clumsy 4 (17) 0 8.32 .004
Item 8, Can’t concentrate 10 (43) 8 (18) 7.01 .031
Item 17, Daydreams, gets lost in his thoughts 11 (48) 10 (22) 6.91 .032
Item 80, Stares blankly 2 (9) 0 4.03 .045
Item 61, Poor schoolwork 7 (30) 5 (11) 4.76 .093
Item 46, Nervous movements or twitching 3 (13) 1 (2) 3.59 .17
Item 41, Impulsive, acts without thinking 6 (26) 12 (27) 2.11 .35
Item 10, Can’t sit still, restless, hyperactive 1 (4) 4 (9) 0.68 .71
Group differences explored with Chi-square analysis (descending x2
).
problem or embedded within motor and intellectual
impairment, as in our study, remains to be seen.
Whether acute anxiety influenced the results in our
study is not known, since we regretfully did not include
a state measure of anxiety. Interestingly, in anxious
adults presented with somewhat threatening auditory
stimuli, only state anxiety but not trait anxiety
influenced attention, as measured by event-related
potentials (Mercado, Carretie´, Tapia, & Go´mez-Jarabo,
2006). Gray et al. (2002) described an attentional test
pattern indicative of what he calls an anxious response
style in three of the four twins with SM he studied. This
response style was characterized by slow reaction
times, numerous omission errors (errors due to non
responding) along with hardly any commission errors
(false positive errors). This could suggest over-
cautiousness and anxiety linked to being afraid of
making mistakes in the test situation. However, an
anxiety state measure was not included in this study
either.
The parent reported attention problems were not
associated with the neuropsychological measure of
attention problems used in our study. However, the
parent reported attention problems was associated with
the parents’ own ratings of anxiety problems and with a
reduced nonverbal IQ and motor problems (Table 4).
That reduced IQ and motor problems were associated
with parental ratings of attention problems is not
surprising given the individual items comprising the
CBCL attention problem syndrome scale that signifi-
cantly differed between SM and controls in our study
(immaturity, clumsiness, being nervous and being
confused; Table 5). That parental CBCL rating of
anxiety and attention problems were associated with
each other, is in line with a recent study on CBCL as a
screen for psychiatric co-morbidity in pediatric patients
with ADHD (Biederman, Monuteaux, Kendrick, Klein,
& Faraone, 2005), where anxiety disorders were
predicted both by the anxious/depressed and the
attention CBCL syndrome scales.
3.3. Limitations
The present study has several limitations. It is a
clinical sample mainly recruited from out-patient
clinics and may thus represent children with increased
co-morbidity. However, the inclusion of children with
SM referred from primary care services (the school
counselling service) somewhat reduces the bias inherent
to clinical samples. Still, the results cannot be
generalised to non-referred children with SM. Interest-
ingly, an association between SM and neurodevelop-
mental disorder/delay has recently been confirmed in a
community study (Elizur & Perednik, 2003).
The mean age, 11.6 years, in the present sample may
also seem relatively high compared to other clinical
samples, but is due to the age limit set for the test
applied in the study. Our total study sample (Kristensen,
2000) corresponds fairly well with other clinic-based
samples with regard to gender ratio, age, SES, age at
symptom onset and symptom duration at assessment.
Furthermore, the assessment was conducted by a
single clinician aware of the children’s group status.
Thus, investigator bias is another possible limitation.
However, this study cannot be conducted blindly,
for obvious reasons. One could also argue that the
differences within and between groups with regard to
location of assessment may have influenced the results.
However, all the participating families chose the
location which they thought the child would feel most
comfortable with, and the children themselves chose
whether one of the parents should be present during the
assessment. It is crucial to establish rapport with
children with SM in order to be able to carry out a
comprehensive assessment. Even though this lack of
uniformity may seem non-optimal methodically, we
found the procedure essential in recruiting an adequate
sample size.
4. Conclusion
The present study explored attentional functioning in
SM, as measured by the Trail Making Test. We found
that children with SM differed from controls on the
Trail Making Test, but this deficit disappeared when
controlling for motor function and IQ in multivariate
analyses.
Parental CBCL ratings of attention problems were
associated with reduced IQ and motor problems, not
with the neuropsychological attention measure.
Further neuropsychological studies controlling for
motor function and IQ, and including measures of
different aspects of attention, are needed.
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Attention in selective mutism

  • 1. Attention in selective mutism—An exploratory case-control study Beate Oerbeck *, Hanne Kristensen Centre for Child and Adolescent Mental Health, Eastern and Southern Norway, Norway Received 11 December 2006; received in revised form 13 April 2007; accepted 20 April 2007 Abstract The aim of the study was to explore the association between selective mutism (SM) and attention. In SM social anxiety seems central but language impairment and motor problems are also reported. Attention problems have been described in parental behavioral ratings, while neuropsychological studies are lacking. A neuropsychological test (the Trail Making Test) and parental ratings of attention- and anxiety problems were administered to a clinical sample of 23 children with SM (aged 7–16 years, 12 boys and 11 girls) and 46 non-referred matched controls. The SM group differed from controls on the Trail Making Test, but the group difference disappeared, when controlling for motor function and IQ. Parental ratings of attention problems were not significantly associated with the neuropsychological attention measure. Neuropsychological studies of attention controlled for IQ and motor function are needed as well as tests that measure different aspects of attention. # 2007 Elsevier Ltd. All rights reserved. Keywords: Neuropsychology; Attention; Anxiety; Child; Selective mutism Selective mutism (SM) is a childhood condition characterized by a persistent lack of speech in certain situations despite the ability to comprehend and use language. Children with SM usually speak to family members at home, but fail to speak in kindergarten or at school (American Psychiatric Association, 1994). The etiology of SM is still unclear but most probably SM represents a heterogeneous condition. SM is frequently associated with social anxiety both in children (Black & Uhde, 1995) and in adults (Steinhausen, Wachter, Laimbock, & Metzke, 2006) and it has been argued that SM represents a form of social anxiety disorder (SAD) (Anstendig, 1999; Sharp, Sherman, & Gross, 2007). However, the relationship between SM and SAD is still under debate (Yeganeh, Beidel, Turner, Pina, & Silverman, 2003). SM is also associated with neurodevelopmental disorder/delay (Kristensen, 2000) in both language and motor function. Whether children with SM may suffer from subtle attention deficits is less studied. To explore the possibility of various neuropsychological impair- ments in SM may throw light on the etiology of the disorder and consequently have implications for treatment. Naturally, due to the mutism, language function has been most frequently studied in children with SM, and language impairment has been consis- tently reported in clinical studies (McInnes, Fung, Manassis, Fiksenbaum, & Tannock, 2004; Steinhausen & Juzi, 1996). However, language measures applied in SM studies may have required visuo-motor abilities as well (Manassis et al., 2003). Thus, in a former study we Journal of Anxiety Disorders 22 (2008) 548–554 * Corresponding author at: Centre for Child and Adolescent Mental Health, Eastern and Southern Norway, P.O. Box 4623, Nydalen, N- 0405 Oslo, Norway. Tel.: +47 22586000; fax: +47 22586001. E-mail address: beate.orbeck@r-bup.no (B. Oerbeck). 0887-6185/$ – see front matter # 2007 Elsevier Ltd. All rights reserved. doi:10.1016/j.janxdis.2007.04.008
  • 2. explored aspects of nonverbal cognition and found that children with SM did not differ from controls on tests of visual memory span and visual memory, while verbal memory span was reduced (Kristensen & Oerbeck, 2006). Motor deviance/delay is reported in studies including larger samples of children with SM (Kolvin & Fundudis, 1981; Steinhausen & Juzi, 1996) both with and without comorbid language disorder (Kristensen, 2002). Impaired motor function represents an important co- morbidity in language disorders without SM (Webster, Majnemer, Platt, & Shevell, 2005). In addition to motor impairment, language disorders are also associated with attention deficits and social difficulties (Snowling, Bishop, Stothard, Chipchase, & Kaplan, 2006). Whether children with SM may have subtle attention deficits is thus animportantquestiontoexplore. Shyness,whichisa hallmark of SM, has also been reported to co-vary with attention problems in normal girls (Caspi & Silva, 1995). Likewise, attention problems in early childhood years significantly predicted symptoms of anxiety and depres- sion in a large cohort study of preadolescent boys and girls (Leech, Larkby, Day, & Day, 2006). With regard to attention problems in SM, parental behavioral ratings suggesting attention deficits have been reported (Kristensen, 2001; Steinhausen & Juzi, 1996). To our knowledge, neuropsychological test results from case-control studies have not been published, and it is not known whether behavioral ratings of attention deficits and anxiety co-vary in this patient group. However, a study including two twin pairs with SM revealed an attentional problem in three of the four twins on a continuous performance test (Gray, Jordan, Ziegler, & Livingston, 2002). In this article, we wanted to study the neuropsycho- logical aspect of attention in a group of SM and matched controls and to examine whether motor function, performance IQ, and a diagnosis of language disorder and/or anxiety disorder were important predictors of performance on the attention test. We also wanted to explore whether the parental behavioral ratings co- varied with neuropsychological measures. 1. Methods 1.1. Subjects The index group consisted of 23 children with SM, 12 boys and 11 girls, mean age11.6 years (S.D. 2.7, range 7– 16 years) while the control group comprised 24 boys and 22 girls, mean age 11.8 years (S.D. 2.6, range 7–16 years). The present study sample consists of children from age 7 years who were able to complete The Trail Making Test and had CBCL parental behavioral ratings (SM group: n = 23; control group: n = 46) from a larger sample of referred children with SM (age range 4–16 years, N = 54 and 108 matched controls). The children with SM were recruited nation-wide from out-patient clinics and school psychology services. The control groupconsisted of children whowere asked to participate by the teachers of the children with SM. For each child with SM, two non-referred children (without SM) matched for gender, age, geographical site and SES were recruited. SES was assessed by the occupations of the parents and rated in accordance with the guidelines of the governmental statistics Norway and collapsed into four classes ranging from: 1 = upper status unto 4 = lower status, with no significant group differences [x2 (3,69) = 2.1, ns]. Details on the recruitment and SES procedures are described in a previous paper (Kristensen, 2000). As one of the matched controls failed to complete the most complex part of the Trail Making Test (part B), the number of controls was reduced from 46 to 45 on this subtest. The parents of one matched control did not fill outthe Child Behavior Checklist,hence N was reduced to 45 on this measure. 1.2. Background assessment and present measures 1.2.1. Diagnostic procedure A DSM-IV diagnosis of SM (APA, 1994) was assigned using the following procedure: 1. Discussion of the SM symptoms with the referring therapists. 2. A structured interview with parents and teachers on SM symptoms (Cline & Baldwin, 1994). 3. Direct observation. 1.2.2. Assessment of intelligence, motor function and behavior problems, and whether a diagnosis of a language disorder and/or an anxiety disorder was present In addition to the neuropsychological test presented in this paper (the Trail Making Test), the examination included assessment of nonverbal IQ, as measured by performance IQ (PIQ) and motor function. PIQ was assessed by the Wechsler Scale of Intelligence-Revised (WISC-R; Wechsler, 1992). For further details con- cerning the WISC-R, see Kristensen (2000). Fine and gross motor function was evaluated by sets of age-dependent motor items designed for the study (6–7 years: 8 items; 8 years: 12 items; 9–16 years: 13 items). The sets of items were chosen from three different motor assessment batteries (Gillberg, 1995; Oseretsky, 1936; Touwen, 1979). Each item was scored B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554 549
  • 3. on a scale from 1 to 4. (1: failing the test; 2: poor performance or not at expected age level; 3: medium performance at expected age level; 4: good performance at expected age level). Each child was given a total motor score (sum score  10 divided by number of items) with a maximum score of 40. To give an example: For age 6 through 7 years these items were included: Pencil grip, cutting a paper circle, diadocho- kinesis, sequential opposition of thumb to same-hand fingers, standing on one leg, jumping on one foot, walking on lateral aspects of feet, and balancing along a line. For more information about the motor tests applied, see Kristensen (2002). The mothers filled out the Achenbach Child Behavior Check List; CBCL (Achenbach, 1991). We present data from two CBCL syndrome scale scores related to anxiety (anxious/ depressed and withdrawn) and the attention syndrome scale. Parents rate their child for how true each item is now or within the past 6 months using the following scale: 0 = not true (as far as you know); 1 = somewhat or sometimes true; 2 = very true or often true. Items are summed up to form the syndrome scales, and these raw scores are reported. For further details concerning the CBCL, see Kristensen (2001). Table 1 present the performance IQ, the motor score, and the CBCL scores of the two groups. The children were diagnosed as to whether or not they had a language disorder using different tests, due to a large age span of the total sample and varying response modes in the SM group. Tests used for the language assessment included the verbal IQ of the WISC-R (Wechsler, 1992), the Boston Naming Test (Kaplan, Goodglass, & Weintraub, 1983), the Reynell Scales (Reynell, 1977) the Peabody Picture Vocabulary Test-revised (Dunn & Dunn, 1981) and audiotapes of a conversation at home. Eleven out of 23 SM patients (48%) were diagnosed with a language disorder, in contrast to 7 of the 46 controls (15%), [x2 (1,69) = 8.46, p = .004]. The possible presence of an anxiety disorder was assessed by the parent version of the Child Assessment Schedule (CAS; Hodges, Kline, & Stern, 1982) adjusted according to DSM-IV criteria [regarding separation anxiety, social phobia, specific phobia, overanxious disorder (generalized disorder in DSM- IV), and obsessive-compulsive disorder]. Nineteen out of 23 SM patients (83%) fulfilled the criteria of an anxiety disorder, in contrast to four of the 46 controls (7%) [x2 (1,69) = 37.67, p < .001]. 1.2.3. Neuropsychological measure of attention Attention was measured using the child version of the Trail Making Test, with number of seconds to complete the test as our test variable (Reitan, 1971). The Trail Making Test require in the first and simpler part A that a line is drawn from digit 1 through 15 on a piece of paper where the numbers are scattered within circles. In the more complex part B, the subject is instructed to draw the lines in sequential shifts between digits and letters (; 1 – A, A – 2, 2 – B and so forth up to 8 – H). The Trail Making Test provides information regarding attention and visual scanning along with speed of both eye–hand coordination and information processing, and it is sensitive to cerebral dysfunction (Reitan & Wolfson, 1985). Studies have reported associations with motor function and IQ scores, and part B can also be seen as a measure of executive functioning as it requires alternate shifts between sets of stimuli; (Lezak, Howieson, & Loring, 2004; Mitrushina, Boone, & Delia, 1999; Spreen & Strauss, 1998; Warner et al., 2006). 1.3. Procedure One examiner (the second author) assessed all the children. The participating families were asked to choose the location where they thought the child would feel most comfortable. The children were left to choose whether or not one of the parents should be present during the assessment. In the SM group, one child was assessed at home, nine at school and 13 at their out- patient clinic, while the control children were mainly seen at school. All participants received the different tests in the same order. B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554550 Table 1 Performance IQ, motor scores and CBCL subscale raw scores in children with SM and controls SM (n = 23) mean (S.D.) Controls (n = 46) mean (S.D.) t-test Wechsler performance IQ 96.39 (18.34) 108.35 (14.74) t = À2.93, p = .01 Motor function score 31.43 (4.68) 37.15 (3.18) t = À5.28, p < .001 CBCL withdrawna 6.83 (3.68) 1.0 (1.38) t = 7.34, p < .001 CBCL anxious-depresseda 7.13 (5.93) 1.60 (2.65) t = 4.26, p = .01 CBCL attentiona 3.78 (4.09) 1.20 (1.31) t = 2.95, p = .008 a Higher score indicates more problems.
  • 4. 1.4. Statistics The data were analyzed using the Statistical Package for the Social Sciences; SPSS version 11 (SPSS, 2002). Means and standard deviations are reported and the significance level was set to 0.05. Due to skewness, the Trail Making Test data were log transformed. Inde- pendent t-tests were used to assess possible group differences. Linear regression analysis was employed to study the impact of SM on attention controlled for motor function, performance IQ, and the presence of language- or anxiety disorders. We used bivariate correlation analysis (Pearson) to study whether the neuropsychological test data and the behavioral rating data co-varied. The x2 test was used to assess whether the groups differed significantly in SES and the presence of deviant scores on the individual CBCL attention subscale items. 2. Results 2.1. Performance on the Trail Making Test The SM group differed significantly from the control group on the Trail Making Test. Mean results (raw scores reported as seconds to complete the test) and analysis of group differences with independent t-tests (using log transformed TMT data) are presented in Table 2. However, the group difference disappeared in multiple regression analysis including IQ and motor function. Motor function and IQ were both significant predictors of the TMT A performance, while IQ significantly predicted the TMT B performance. The results from the regression analysis are presented in Table 3. One could question the use of performance IQ as a control for IQ in our study, since this score to some extent is time dependent (better scores with quick performance), as is the Trail Making Test itself. We therefore also performed the regression analyses with another measure related to IQ (the Peabody Picture Vocabulary Test-revised), that is time independent, rendering similar results (results not shown). Analysis of whether or not the presence of a language disorder or an anxiety disorder influenced the attention test results by including them in the regression analysis (using the enter method) proved that none were significant predictors (all standardized betas <.23, ns). Analysis of whether or not the presence of the parental CBCL ratings influenced the attention test results by including them in the regression analysis (using the enter method) proved that none were significant predictors (all standardized betas <.30, ns). 2.2. The relation between neuropsychological measures and questionnaire data The neuropsychological measure of attention pro- blems (the Trail Making Test) did not co-vary with the parental ratings of problems with attention or anxiety as measured by the CBCL syndrome scales. However, the B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554 551 Table 2 Mean results (time to finish in seconds; raw scores) on the Trail Making Test (part A and part B) in children with SM (n = 23) and controls (n = 46) and results from group comparison using independent t-tests on logtransformed data SM (n = 23) mean (S.D.) Controls (n = 46) mean (S.D.) Mean difference (95% CI) on logtransformed data t (dfa ) p-value TMT, part A 26.0 (12.5) 19.9 (7.4) 0.22 (0.002 to 0.45) 2.01 (67) .05 TMT, part B 62.4 (31.8) 43.1 (16.9) 0.31 (0.09 to 0.54) 2.48 (66) .008 a df: degrees of freedom. Table 3 Multiple regression analysis (enter method): effects of patient status and performance IQ and motor score on log transformed Trail Making Test (part A and part B) data Dependent variable: Trail Making Test, part A Dependent variable: Trail Making Test, part B Standardized coefficient (b) p-value Standardized coefficient (b) p-value Patient status .154 .226 À.114 .423 Performance IQ À.356 .002 À.268 .029 Motor score À.463 <.001 À.193 .173 R2 adjusted .320 .170 Significance of model F(3,65) 11.65, p < .001 F(3,64) 5.43, p = .002
  • 5. CBCL parent reported attention problems were asso- ciated with the two other neuropsychological measures; motor problems and reduced nonverbal IQ (Table 4). The parent reported syndrome scale scores on attention and anxiety (anxiety/depression and with- drawn) also correlated significantly (R = .67 and .58, respectively, p < .001). The specific items from the CBCL syndrome scale of attention problems that best differentiate SM from controls are presented in Table 5. 3. Discussion 3.1. Performance on the Trail Making Test This is the first case-control study exploring a neuropsychological measure of attention in children with SM. Because studies on SM that include comprehensive neuropsychological assessment with sufficient samples sizes are lacking, our report on specific aspects of attentional functioning in SM may be important. This added knowledge may contribute to the understanding of possible pathways of symptom development and to the identification of possible subgroups of children with SM. The present study found an attention deficit in SM, as measured by the Trail Making Test. However, motor problems and reduced nonverbal IQ were central factors in explaining this deficit, emphasizing the need to control for these factors in future measures of attention. Our results are in line with a study of 38 anxious/ depressed boys (age 9–11 years) who performed the Trail Making Test part B significantly slower than the control group (Emerson, Mollet, & Harrison, 2005). No attempts were made to assess the impact of IQ and motor function in this study. Hence whether anxiety actually explained the mentioned deficit, is not known. 3.2. The relation between neuropsychological measures and questionnaire data The neuropsychological measure of attention did not co-vary with the parent reported attention or anxiety problems. This finding, along with the fact that neither a language disorder, nor the presence of an anxiety disorder significantly predicted the Trail Making Test performance, could suggest that the neuropsychologi- cally measured attention deficit represent an important dimension in SM worth further studying. Whether possible attention deficits will present as a separate B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554552 Table 4 Correlations between parental ratings (three CBCL syndrome scales) and neuropsychological measures (log-transformed Trail Making Test score, motor score and Performance IQ) Parental ratings Neuropsychological measures CBCL syndrome scales TMT A TMT B Motor score Performance IQ Attention problems .168, ns .079, ns À.514** , p < .001 À.253* , p = .037 Anxiety/depression .054, ns .057, ns À.412** , p < .001 À.139, ns Withdrawn .041, ns .068, ns À.485** , p < .001 À.232, ns * p < .05. ** p < .001. Table 5 The number and percentage of children with SM (n = 23) and controls (n = 45) with positive item scores (1 = somewhat or sometimes true, 2 = very true) on the CBCL attention syndrome subscale items SM (n = 23) N (%) Controls (n = 45) N (%) x2 p-value Item 1, Acts too young for his age 10 (43) 4 (8) 11.14 .004 Item 45, Nervous 10 (43) 5 (11) 10.42 .005 Item 13, Confused, seems to be in a fog 6 (26) 1 (2) 9.49 .009 Item 62, Poorly coordinated, clumsy 4 (17) 0 8.32 .004 Item 8, Can’t concentrate 10 (43) 8 (18) 7.01 .031 Item 17, Daydreams, gets lost in his thoughts 11 (48) 10 (22) 6.91 .032 Item 80, Stares blankly 2 (9) 0 4.03 .045 Item 61, Poor schoolwork 7 (30) 5 (11) 4.76 .093 Item 46, Nervous movements or twitching 3 (13) 1 (2) 3.59 .17 Item 41, Impulsive, acts without thinking 6 (26) 12 (27) 2.11 .35 Item 10, Can’t sit still, restless, hyperactive 1 (4) 4 (9) 0.68 .71 Group differences explored with Chi-square analysis (descending x2 ).
  • 6. problem or embedded within motor and intellectual impairment, as in our study, remains to be seen. Whether acute anxiety influenced the results in our study is not known, since we regretfully did not include a state measure of anxiety. Interestingly, in anxious adults presented with somewhat threatening auditory stimuli, only state anxiety but not trait anxiety influenced attention, as measured by event-related potentials (Mercado, Carretie´, Tapia, & Go´mez-Jarabo, 2006). Gray et al. (2002) described an attentional test pattern indicative of what he calls an anxious response style in three of the four twins with SM he studied. This response style was characterized by slow reaction times, numerous omission errors (errors due to non responding) along with hardly any commission errors (false positive errors). This could suggest over- cautiousness and anxiety linked to being afraid of making mistakes in the test situation. However, an anxiety state measure was not included in this study either. The parent reported attention problems were not associated with the neuropsychological measure of attention problems used in our study. However, the parent reported attention problems was associated with the parents’ own ratings of anxiety problems and with a reduced nonverbal IQ and motor problems (Table 4). That reduced IQ and motor problems were associated with parental ratings of attention problems is not surprising given the individual items comprising the CBCL attention problem syndrome scale that signifi- cantly differed between SM and controls in our study (immaturity, clumsiness, being nervous and being confused; Table 5). That parental CBCL rating of anxiety and attention problems were associated with each other, is in line with a recent study on CBCL as a screen for psychiatric co-morbidity in pediatric patients with ADHD (Biederman, Monuteaux, Kendrick, Klein, & Faraone, 2005), where anxiety disorders were predicted both by the anxious/depressed and the attention CBCL syndrome scales. 3.3. Limitations The present study has several limitations. It is a clinical sample mainly recruited from out-patient clinics and may thus represent children with increased co-morbidity. However, the inclusion of children with SM referred from primary care services (the school counselling service) somewhat reduces the bias inherent to clinical samples. Still, the results cannot be generalised to non-referred children with SM. Interest- ingly, an association between SM and neurodevelop- mental disorder/delay has recently been confirmed in a community study (Elizur & Perednik, 2003). The mean age, 11.6 years, in the present sample may also seem relatively high compared to other clinical samples, but is due to the age limit set for the test applied in the study. Our total study sample (Kristensen, 2000) corresponds fairly well with other clinic-based samples with regard to gender ratio, age, SES, age at symptom onset and symptom duration at assessment. Furthermore, the assessment was conducted by a single clinician aware of the children’s group status. Thus, investigator bias is another possible limitation. However, this study cannot be conducted blindly, for obvious reasons. One could also argue that the differences within and between groups with regard to location of assessment may have influenced the results. However, all the participating families chose the location which they thought the child would feel most comfortable with, and the children themselves chose whether one of the parents should be present during the assessment. It is crucial to establish rapport with children with SM in order to be able to carry out a comprehensive assessment. Even though this lack of uniformity may seem non-optimal methodically, we found the procedure essential in recruiting an adequate sample size. 4. Conclusion The present study explored attentional functioning in SM, as measured by the Trail Making Test. We found that children with SM differed from controls on the Trail Making Test, but this deficit disappeared when controlling for motor function and IQ in multivariate analyses. Parental CBCL ratings of attention problems were associated with reduced IQ and motor problems, not with the neuropsychological attention measure. Further neuropsychological studies controlling for motor function and IQ, and including measures of different aspects of attention, are needed. References Achenbach, T. (1991). Integrative guide for the 1991 CBCL/4-18, YSR and TRF profiles. Burlington, VT: University of Vermont, Depart- ment of Psychiatry. American Psychiatric Association (1994). Diagnostic and statistical manual of mental disorders, 4th ed. (DSM-IV). Washington, DC: Author. Anstendig, K. D. (1999). Is selective mutism an anxiety disorder? Rethinking its DSM-IV classification. Journal of Anxiety Disor- ders, 13, 417–434. B. Oerbeck, H. Kristensen / Journal of Anxiety Disorders 22 (2008) 548–554 553
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