J Child Fam Stud (2016) 25:503–517
DOI 10.1007/s10826-015-0225-4
ORIGINAL PAPER
Effectiveness of an Individualized Case Formulation-Based CBT
for Non-responding Youths with Anxiety Disorders
Irene Lundkvist-Houndoumadi1 • Mikael Thastum1 • Esben Hougaard1
Published online: 3 June 2015
Springer Science+Business Media New York 2015
Abstract The study examined the effectiveness of an
individualized case formulation-based cognitive behavior
therapy (CBT) for youths (9–17 years) with anxiety disorders and their parents after unsuccessful treatment with a
manualized group CBT program (the Cool Kids). Out of
106 participant youths assessed at a 3-month follow-up
after manualized CBT, 24 were classified as non-responders on the Clinical Global Impression-Improvement scale
(CGI-I), and 14 of 16 non-responders with anxiety as their
primary complaint accepted an offer for additional individual family CBT. The treatment was short-term (M sessions = 11.14) and based on a revised case formulation
that was presented to and agreed upon by the families. At
post-treatment, nine youths (64.3 %) were classified as
responders on the CGI-I and six (42.9 %) were free of all
anxiety diagnoses, while at the 3-month follow-up 11
(78.6 %) had responded to treatment and nine (64.3 %) had
remitted from all anxiety diagnoses. Large effect sizes
from pre- to post-individualized treatment were found on
youths’ anxiety symptoms, self-reported (d = 1.05) as well
as mother-reported (d = .81). There was further progress at
the 3-month follow-up, while treatment gains remained
stable from post-treatment to the 1-year follow-up. Results
indicate that non-responders to manualized group CBT for
youth anxiety disorders can be helped by additional CBT
targeting each family’s specific needs.
Keywords Non-responders Anxiety disorders CBT Case formulation Individualized treatment
& Irene Lundkvist-Houndoumadi
irenelh@psy.au.dk
1
Department of Psychology and Behavioural Sciences, Aarhus
University, Bartholins Allé 9, 8000 Aarhus C, Denmark
Introduction
Anxiety disorders are amongst the most common psychiatric disorders in children and adolescents (Costello et al.
2011). They negatively influence the developmental trajectories impacting family processes, youths’ functioning
with peers, school, and recreation (Essau et al. 2000).
Furthermore, longitudinal research has found above-average levels of life interference into early adulthood (Last
et al. 1997) and anxiety disorders are considered gateway
disorders, predicting mental health problems in adulthood,
such as anxiety and depression (Kessler et al. 2009;
Merikangas et al. 2010).
During the past two decades, cognitive behavior therapy
(CBT) for anxiety disorders in children and adolescents
(from now on referred to as youths) has been evaluated in
an increasing number of randomized controlled trials
(RCTs), as evidenced by 41 studies included in a recent
Cochrane review (James et al. 2013), compared to only 18
studies included in an earlier review (James et al. 2005).
The studies in James et al. (2013) meta-analysis examined
the effectiveness of manualized CBT that varied in duration (9–20 sessions), degree of parental participation and
was either individual or group. Findings indicated that CBT
is an effective treatment for anxiety disorders in youths,
with the mean remission rate for any anxiety disorder
diagnosis being 59.4 %, while no difference was found in
the outcome of the different formats of CBT. Another
meta-analysis by Reynolds et al. (2012) reported a medium
effect size (Cohen’s d = .77) for youth-reported anxiety
symptoms after CBT. Group CBT had a medium effect
size, while individual CBT had a large effect size and
disorder-specific CBT had a larger effect size than generic
CBT. However, the only disorder that was examined both
specifically and generically was social phobia and there
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was therefore a confounding between the specificity of
treatment and the anxiety disorder treated. In contrast to
Reynolds et al. (2012), Liber et al. (2008) found no difference in the efficacy of individual CBT compared to
group CBT, while a recent effectiveness study by Wergeland et al. (2014) also did not find any significant differences between those two formats.
CBT’s effectiveness notwithstanding, about 40 % of
youths do not remit (James et al. 2013). A recent systematic literature review (Lundkvist-Houndoumadi et al. 2014)
found some evidence for pre-treatment symptomatic
severity and non-anxiety comorbidity in youths predicting
a worse end-state functioning, but not a lower degree of
improvement. Few of the included studies examined the
primary diagnosis as a predictor, but two of those that did,
found some evidence for social phobia (SoP) being associated with a worse treatment outcome (Crawley et al.
2008; Kerns et al. 2013). This finding was also supported
by two recent extensive studies (Compton et al. 2014;
Hudson et al. 2015). Lundkvist-Houndoumadi et al. (2014)
also reported finding some support that parental psychopathology, primarily anxiety and depression, was
associated with worse outcomes of CBT for youth, when
parental psychopathology was assessed through self-reports. However, two studies that assessed parental psychopathology through diagnostic interviews, reported
results that were partially in opposite direction to each
other (see Legerstee et al. 2008; Bodden et al. 2008). Thus,
no predictor of treatment outcome has been consistently
supported in the literature, a conclusion similar to the one
drawn by Taylor et al. (2012) regarding the adult literature.
They point out the need to develop strategies in order to
improve treatment outcomes if the initial treatment has not
contributed to clinical improvement, one of their suggestions being to re-evaluate the case-formulations in case of
non-response. Non-response has been defined variously
across studies, but in broad terms non-responders are participants, who might have experienced some symptom
reduction, but have not shown clinically significant
improvement, or their target symptoms are still clinically
significant after the end of treatment (Taylor et al. 2012).
Few have examined whether youths with anxiety disorders, who have not responded to a standard manualized
CBT program, can progress through additional therapy.
Perini et al. (2013) highlighted the need for developing a
stepped-care approach to treating anxious children.
Legerstee et al. (2010) offered 91 youths (aged 8–16) a
standardized stepped care CBT. As a first treatment step
youths were offered group or individual CBT following the
Friends Program (Barrett et al. 2000), consisting of ten
child sessions and four separate parent sessions. Treatment
response was defined as youths being free of all anxiety
diagnoses. Thirty-nine youths (43 %) responded to the first
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treatment step, while after the second treatment phase,
which consisted of ten standardized individual CBT sessions involving both the child and the parents, out of the 50
youths receiving the additional treatment, 37 (74 %) were
free of their anxiety disorders. Similarly, van der Leeden
et al. (2011) offered 132 youths manualized (group or
individual) CBT, which consisted of the Friends Program
(Barrett and Turner 2000). When assessments at posttreatment indicated the presence of an anxiety disorder and/
or self-reported anxiety levels above the clinical cut-off
point, a second and possibly a third treatment phase were
offered. Each of the additional treatment phases consisted
of 5 additional sessions of manualized CBT and parents
were also included in each session. They found that after
the first phase, 60 youths (45 %) had remitted from all
anxiety diagnoses, and of the 24 youths that received the
two additional phases (i.e., 10 extra sessions), 14 youths
(58 %) remitted from all diagnoses.
CBT is commonly implemented through manuals, which
have been suggested to enable evidence-based practice,
securing therapist adherence and facilitating dissemination
(Kendall et al. 2008). However, manualized treatments
have also received criticism, as they are limited in their
applicability to the broad range of problems encountered in
clinical practice (Carroll and Rounsaville 2008). As
Southam-Gerow et al. (2012) point out, the treatment
programs consider specific child disorders, without considering the multiple factors (i.e., child and family factors,
therapist factors, organization factors, and service system
factors), which all influence how potent a treatment will be.
In order to be able to individualize therapy and guide
clinicians’ decision making during treatment, Weisz et al.
(2013) proposed creating flowcharts through weekly feedback systems, based on assessments of each youth’s
treatment response. The weekly feedback system was tested together with a modular treatment protocol for anxiety,
depression, and conduct problems and showed good results
(see Weisz et al. 2012). This suggests that a more personalized approach may be powerful.
In accordance to Taylor et al.’s (2012) suggestion,
Persons (1991) advocated a case formulation model, in
order to tailor treatment to the needs of the individual
client. Treatment plans are made, by integrating in the
evidence-based treatment procedures, elements the clinician finds suitable, depending on the factors that contribute
to the maintenance or protection of the youth’s psychological problems. Arguably, this approach can easily be
implemented in clinical practice. We would suggest that
among youths who have not responded to CBT, it would
prove to be especially helpful, since it may guide clinicians
in planning the subsequent treatment step, after integrating
their knowledge on the family obtained during the first step
of treatment. Furthermore, it encourages a collaborative
J Child Fam Stud (2016) 25:503–517
stance, as the families are presented with the case formulation and invited to comment upon it, which is in accordance to participatory decision-making that according to
Chorpita and Daleiden (2014) is prioritized in the paradigm
of individualized care within service systems.
The present study can be considered to be an evaluation
of a second step of treatment, offered non-responders after
a manualized group CBT intervention, following the Cool
Kids Program (Rapee et al. 2000) that was recently evaluated in a RCT (Arendt et al. 2015). In the RCT 109 youths
(aged 7–16) with a primary anxiety disorder received
treatment and results showed that at post-treatment 27
youths (48 %) were free of all anxiety diagnoses, while at
the 3-month follow-up 55 (58 %) were. The aim of this
study was to evaluate the effectiveness of an individualized, case formulation-based CBT for youths with anxiety
disorders and their parents, after unsuccessful group treatment with manualized CBT. We hypothesized that a case
formulation-based individualized CBT would contribute to
a significant decrease in anxiety symptoms and in perceived life-interference due to the anxiety.
Method
Participants
Participant youths were recruited from the Anxiety Clinic
for Children and Adolescents at Aarhus University in
Denmark, from January 2011 to April 2012 in connection
with an RCT (Arendt et al. 2015) on a manualized CBT for
youths with anxiety disorders and their parents (the Cool
Kids Program; Rapee et al. 2000). In addition to the 109
participants from the RCT, 10 more youths treated in two
non-randomized groups were included, while 13 were
excluded, because they were lost to evaluation at the
3-month follow-up, when responder status was assessed.
Based on the Clinical Global Impressions-Improvement
scale (CGI-I; Guy 1976), 24 youths (22.6 %) were classified as non-responders. Eight youths were excluded, since
two had been admitted to treatment elsewhere and six
appeared to have had another primary difficulty than anxiety (eating disorder: 2, autism spectrum disorder: 3, severe
cognitive difficulties: 1). Of the 16 youths offered additional treatment, two declined (one family had moved to
another part of the country and the other one did not
believe further treatment was needed), resulting in our final
sample of 14 participant non-responders. The youths (eight
girls and six boys) were of Danish ethnic background and
had a mean age of 12.7 years (SD = 3.1). Almost all
youths were living with both parents (n = 12, 86 %). The
highest completed education of parents was most commonly further/higher education (mothers n = 10, 71.4 %;
505
fathers n = 8, 61.54 %), while few had a vocational education (mothers n = 2, 14.3 %; fathers n = 3, 23 %) or a
high-school equivalent (mothers n = 2, 14.3 %; fathers
n = 2, 15.4 %). Age, gender, diagnosis and CGI-scores of
participants appear in Table 1. At referral one of the youths
(ID: 8) received medication for her ADHD symptomatology and another (ID: 6) for his depressive symptoms. Three
of the non-responding youths (ID 2, 10, 12) were responders after the manualized treatment, but relapsed in the
3-month follow-up period, while three (ID 4, 7, 8) had
progressed somewhat on the CGI-I in the follow-up period,
but were still classified as non-responders.
The study was approved by the local county Ethical
Committee and by the Danish Data Protection Agency and
all parents signed an informed consent form.
Procedure
The manualized Cool Kids program (Rapee et al. 2000)
consisted of 10 weekly 2-h sessions in groups of five to
seven youths and their parents. The main treatment components were psychoeducation, cognitive restructuring and
gradual exposures.
Prior to the individualized treatment, a new case formulation and treatment plan was worked out for each
youth. Case formulations were also created prior to the
manualized CBT, but they were not systematically used in
the treatment. Most often, no major changes were made,
but additional information was added, especially as to
reasons for non-response in the manualized CBT. The
therapist from the youth’s manualized treatment provided a
draft of the new case formulation and treatment plan that
was later discussed at a clinical staff meeting. Possible
obstacles to treatment progress as well as length of treatment were also considered in that meeting. At a succeeding
meeting with the family, the therapist presented an easy to
grasp version of the proposed new case formulation and
treatment plan, which the families were encouraged to
comment on. Their suggestions, if any, were taken into
account in the final, agreed-upon formulation and treatment
plan.
The individualized treatment was based on CBT principles. For youths with a primary SoP diagnosis (ID 1–6),
disorder-specific components were added to therapy, such
as behavioral experiments with video-feedback (see Clark
2001) and social-skills training exercises. Parents of youths
with a primary separation anxiety disorder (SAD; ID 7–11)
were encouraged to use contingency management, in order
to promote youths’ independence and brave behavior (see
Silverman and Kurtines 1999). Youths with an ADHDsymptomatology (ID 2, 4, 8, 13) received extra help in
creating detailed weekly plans of the exposure exercises
the youths needed to complete. Youths with a comorbid
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Table 1 Demographic characteristics and categorical outcomes of manualized and individualized treatments
Manualized treatment
Pre-treatment
Individualized treatment
Post-treatment
Diagnoses (CSR)
3-month F/U
CGII
Diagnoses (CSR)
CGII
Post-treatment
3-month F/U
Diagnoses
(CSR)
Diagnoses
(CSR)
ID
Gender,
age
Diagnoses (CSR)
CGII
CGII
1
M, 12
SoP (5), OCD (4)
SoP (4)
3
SoP (5)
3
SoP (4)
2
OCD (4)
2
2
M, 10
SP (6), GAD (5), SAD
(6), ADHD (4)
SoP (4), SP (4)
2
SoP (6), SP (4),
ADHD (4)
3
–
1
ADHD (4)
1
3
M, 15
SoP (7)
SoP (5)
3
SoP (6)
3
–
1
SoP (4)
2
4
F, 14
SoP (7)
SoP (7)
4
SoP (7), ADHD (4)
3
SoP (5)
3
–
1
5
F, 16
SoP (8), GAD (5)
SoP (7)
3
SoP (7)
3
SoP (5)
3
SoP (5)
3
6
M, 15
SoP (7), DD (6)
4
SoP (7)
4
SoP (7)
3
SoP (7)
4
7
F, 12
SoP (7), GAD (4), DD
(5)
SAD (6), SP (5), GAD
(5), MDD (4)
SAD (6)
4
SAD (6)
3
SoP (5)
3
–
1
8
F, 13
SAD (8), GAD (6), PD
(5), ADHD*
SAD (7), GAD
(6), MDD (6)
5
SAD (6)
3
GAD (4)
1
–
1
9
F, 9
SAD (7)
SAD (6)
3
SAD (7)
3
–
1
–
1
10
F, 16
AP/PD (6), GAD (4)
–
1
SAD (5), GAD (4),
AP/PD (4), DD (4)
3
DD (4)
2
–
1
11
M, 10
SAD (6), SP (5)
SAD (5)
3
SAD (4)
3
–
1
–
1
12
M, 7
OCD (4)
–
1
OCD (7)
3
OCD (4)
2
–
1
13
F, 17
GAD (8), PD (7), SP (7),
DD (6) ADHD*
GAD (5), SP (4)
3
GAD (4), SP (4),
ADHD (5), DD (5)
3
–
1
PD (6)
3
14
F, 9
SP (6), GAD (4), SAD
(4)
SP (6)
3
SP (6), GAD (6), SAD
(4)
3
SP (4)
3
–
1
CSR, clinical severity rating; CGI-I, Clinical Global Impressions-Improvement Scale; SoP, social phobia; SAD, separation anxiety disorder;
OCD, obsessive compulsive disorder; GAD, generalized anxiety disorder; SP, specific phobia; AP/PD, agoraphobia with panic disorder; ADHD,
attention deficit hyperactivity disorder; MDD, major depressive disorder; DD, dysthymic disorder
* Diagnosis given from psychiatric assessment prior to therapy start
mood disorder (ID 10, 13) were encouraged to complete a
positive events diary and arrange more enjoyable activities
in their daily lives. Problems in families that inhibited
youths’ treatment progress, such as an overprotective parenting style (ID 3, 5, 9, 11–14) and parental problems or
conflicts (ID 4, 7, 8, 10), were also addressed. Furthermore,
meetings with the therapist, the family and youths’ teachers
were arranged, when needed (ID 1, 2, 5). Table 2 presents
the most common problem areas identified among the
youths during the manualized treatment, with the respective treatment elements added in the individualized
treatment.
Therapy was most often scheduled to take place in 10
weekly sessions, but in three cases (ID 3, 8, 9) 6 or 8
sessions were offered, often with a 2-week interval
between sessions. The possibility of further sessions was
left open; in two cases (ID 5, 6) 10 additional sessions were
offered. Thus, the number of therapy sessions ranged from
6 to 20 (M = 11.14, SD = 4.13). The individualized
intervention was conducted by one of the Anxiety Clinic’s
psychologists, most commonly the same therapist that had
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led the manualized treatment. All three therapists had prior
experience in CBT for youths with anxiety disorders. The
two therapists with less clinical experience received
weekly supervision by the third therapist, who was specialized in CBT. For educational purposes, a graduate
student attended therapy sessions and sometimes helped
the youth with his/her exposure exercises.
A case vignette of a 13-year old girl with a primary SAD
(ID 8) shown in the Appendix illustrates the course of the
two treatments and is accompanied by the case formulation
as presented at the clinical staff meeting and the family.
Measures
Primary Outcome Measures
Anxiety Disorder Interview Schedule for DSM-IV, Parent
and Child Versions (ADIS-IV-C/P) The ADIS-IV-C/P
(Silverman and Albano 1996) is a semi-structured interview for diagnosing anxiety disorders in youths based on
the Diagnostic and Statistical Manual of Mental Disorders,
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507
Table 2 Problem areas and treatment elements of individualized treatment
Problem areas in manualized treatment
Treatment elements of individualized treatment
Therapy
Therapy
Group format is anxiety provoking
Therapy is conducted in individual setting
Therapy not targeted to youths’ specific symptomatology
Disorder specific treatment (e.g. in case of SoP: in vivo exposures with
video-feedback, social skills training)
Youth
Youth
Cognitive difficulties, youth unable to do cognitive restructuring
Assistance in and simplification of cognitive restructuring, greater
emphasis on exposures
Youth lacks motivation to work on anxiety, seem ambivalent about
engaging in therapy work
Motivation talk (e.g., youth estimates the extent to which they believe in:
1. own capabilities, 2. appropriate timing, 3. treatment)
Avoidance (overt or subtle) of anxiety and use of safety behaviours
Practice relaxation exercises and assist youth in handling anxiety feelings
and staying in situation until anxiety subsides
School refusal or difficulties in attending school systematically
Network meetings with school and planning of a gradual involvement in
school after cooperating with teachers
Depressive symptomatology and difficulties handling pressure
Therapy starts with behavioral activation through very easy exposures
and completion of positive diary
Family
Family
Lack of systematic homework completion
Detailed homework planning and close follow-up, problem solving
Overprotective parenting, parents have difficulties setting demands to
their child
Parents made aware of consequences of behaviors through guided
discovery, instructed in contingency management
Parental psychopathology
Problematic family issues (e.g., lack of resources/time for parents to
practice with youth, parents disagree on how to handle youth’s
symptomatology, parent–child conflicts)
Parents refferred to treatment elsewhere
Closer examination of family issues from which different interventions
may follow, parents are involved in treatment in various ways (e.g.,
individual parent sessions, sessions with both parents and child)
4th edition (DSM-IV; American Psychiatric Association
1994). All disorders are given a clinical severity rating
(CSR) from 0 (no interference) to 8 (extreme interference),
with severity ratings of 4 and above indicating the presence
of a disorder. Psychologists or trained and supervised
graduate students conducted the ADIS-IV interviews (see
Arendt et al. 2015 for further information). The most
impairing diagnosis was considered to be the primary
diagnosis. For the individualized intervention, the primary
diagnosis was considered to be the one that was assessed at
the 3-month follow-up to the manualized treatment. ADIS
assessors were blinded at post and follow-up assessments
to the youths’ prior diagnoses. Previous studies have found
that ADIS-IV-C/P possesses favorable psychometric
properties (Silverman et al. 2001; Wood et al. 2002). A
reliability check was conducted in the RCT (Arendt et al.
2015), where one of two trained assessors watched and
rated a total of 22 (20.2 %) of the video-recorded baseline
interviews. Comparing these new assessments to the original, the interrater reliability (j) for the primary anxiety
diagnosis was .77, and the interclass correlation coefficient
for the CSR of the primary anxiety diagnosis was .69 (twoways mixed for individual raters, consistency).
Clinical Global Impressions-Improvement Scale (CGII) The CGI-I (Guy 1976) is a clinician administered 7-point
Likert-type scale, the seven points correspond to the following descriptions: 1: very much improved, 2: much
improved, 3: minimally improved, 4: no change, 5: minimally worse, 6: much worse, 7: very much worse. A score
of 1 or 2 reflects a substantial, clinically meaningful
decrease in the primary anxiety disorder, while changes in
other disorders are also considered. A score of 2 may be
given in case of the presence of a mild disorder (CSR 0–4).
The CGI-I is commonly used in clinical trials to define
response to treatment and it has been found to be sensitive
to change through treatment (Zaider et al. 2003). The CGI-I
was rated by the therapist of the preceding treatment
(manualized or individualized) after consulting changes on
the ADIS, but without knowledge of youths’ and mothers’
reports on anxiety levels. A substantial agreement was
found (j = .72) between CGI-I non-response and the
presence of the primary diagnosis at the 3-month follow-up
to the manualized treatment, when non-responders were
identified.
Spence Children’s Anxiety Scale (SCAS-C/P) The SCAS
was completed by youths (SCAS-C; Spence 1997) and
mothers (SCAS-P; Nauta et al. 2004). SCAS-C consists of
seven subscales for specific anxiety diagnoses: social
phobia, panic disorder and agoraphobia, generalized anxiety disorder, obsessive–compulsive disorder, separation
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anxiety disorder, and specific phobia (fear of physical
injury). It has 38 items and six positive filler items, rated 0
(never) to 3 (always). In the current study only the total
score of the subscales added together was used (range
0–114). The Danish translation of the SCAS-C has
demonstrated excellent internal consistency for the total
scale (a = .89) in a sample of youths with anxiety disorders and good test–retest reliability after 2 weeks (r = .84)
and 3 months (r = .83) in a community sample (Arendt
et al. 2014). The SCAS-P, completed by mothers, contains
the same items as SCAS-C, except for the six positive filler
items, and is scored in the same way. The Danish translation of SCAS-P has demonstrated good internal consistency for the total scale (a = .87) in a sample of parents of
youths with anxiety disorders and good test–retest reliability after 2 weeks (r = .88) and 3 months (r = .81) in a
community sample (Arendt et al. 2014). Internal consistency in the RCT sample (Arendt et al. 2015) for the total
scale was excellent for SCAS-C (a = .90) and for SCAS-P,
completed by mothers (a = .89).
Secondary Measures
Children’s Life Interference Scale (CALIS-C/P) The
CALIS is designed to measure life interference and
impairment experienced due to anxiety (Lyneham et al.
2013). All items are rated from 0 (not at all) to 4 (a great
deal). The child version (CALIS-C) consists of nine items
that examine the impairment experienced due to anxiety in
several areas (e.g., how much do fears and worries make it
difficult for you to do the following things? a. getting on with
parents; b. getting on with brothers and sisters; c. being with
friends outside of school…). The parents’ version (CALISP), completed by mothers in the study, consists of seven
additional items that examine the extent to which youth’s
anxiety interferes with parents’ own life (e.g., how much do
your child’s fears and worries interfere with your everyday
life in the following areas: a. your relationship with your
partner, or a potential partner; b. your relationship with
extended family; c. your relationship with friends…)
attributed to the youth’s anxiety. In the current study only
ratings for the combined measure of overall interference are
reported (possible score range CALIS-C: R = 0–36,
CALIS-P: R = 0–64). The scale has demonstrated satisfactory internal consistency on subscales for both youth (a
range = .70–.84) and parent ratings (a range = .75–.90)
and moderate stability for a 2-month retest period
(r range = .62–.91; Lyneham et al. 2013). In the RCT study
(Arendt et al. 2015) the Cronbach’s a was .81 for youthreported and .83 for mother- reported overall interference
with the youth’s life and .87 for interference on mothers’ life.
Experience of Service Questionnaire (ESQ) The ESQ is
a measure assessing youths’ and parents’ satisfaction with
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the treatment (Attride-Stirling 2002). The items are rated
with 0 (not true), 1 (partly true), or 2 (true). The youth
version consists of 7 items (e.g., the treatment helped me;
after therapy I felt more like being with family and
friends…), and mothers’ version of 10 items (e.g., the
treatment helped my child; I was able to change my
behavior towards my child in a positive way…), while the
parent version has an additional two questions to comment
on what they liked about the treatment and what could be
improved.
Administration of Measures
Measures of outcome were administered pre- and posttreatment, and at 3-month follow-up in both the manualized
and the individualized intervention (3-month follow-up to
the RCT was pre-treatment for the individualized treatment).
At the 12-month follow-up of the individualized treatment,
families completed only the SCAS and the CALIS, while the
ESQ was administered only at post-treatment in both interventions. All questionnaires were electronically administered. Families were sent an e-mail with a link, and in case of
non-reply, they received 3 weekly reminders, followed by a
phone call after 4 weeks.
Statistical Analysis
Pre-post and pre-3 month comparisons of the individualized treatment were analyzed, examining: (1) the number
of participants responding to treatment (CGI-I \ 3); (2) the
number of participants free of diagnoses (primary and all);
(3) the number of participants showing statistical and
clinical significant change on the SCAS-C/P; and (4) the
degree of change on continuous outcome measures (CSR,
SCAS-C/P, CALIS-C/P). Statistical and clinical significant
change was calculated according to the Jacobson and Truax
(1991) method. In order for youths to show statistical
significant change, the change in scores had to be greater
than the calculated reliable change index (RCI) and in
order to show a clinical significant change, in addition to
the statistical significant change, they also had to score
below the cut-off point between anxious (clinical) and nonanxious (norm) youths after the individualized treatment:
CScut-off = [(SDnorm 9 Mclinical) ? (SDclinical 9 Mnorm)]/
(SDnorm ? SDclinical). The clinical cut-off and RCI values
were calculated using Danish community and clinical
norms split into age groups (7–12 and 13–17 years) and
gender (see Arendt et al. 2014, Table 7, p. 953). Maintenance of treatment outcome from post-treatment to
12-month follow-up was examined on SCAS-C/P and
CALIS-C/P. Outcomes on the continuous measures were
analyzed by t-tests for related samples and Cohen’s d effect
size was computed based on change score values
36.93
(14.36)
13.00 (7.96)
31.93
(15.64)
SCAS-P
CALISC
CALIS-P
26.79 (16.35)
10.00 (8.03)
27.07 (10.55)
24.38 (16.03)
C/P = 13/14
6.43 (3.16)
28.57 (14.57)
10.14 (7.5)
27.14 (10.64)
23.5 (9.54)
N = 14
8.07 (3.40)
16.79 (11.57)
8.17 (6.78)
17.00 (9.75)
15.17 (8.93)
C/P = 12/14
3.93 (2.95)
2.86 (2.21)
N = 14
15.23
(12.15)
5.75 (4.16)
17.77 (8.15)
14.17 (9.52)
C/P = 12/13
3.29 (3.54)
2.36 (2.17)
N = 14
3-month F/U
17.50 (13.35)
4.64 (3.70)
21.29 (13.11)
15.36 (10.23)
C/P = 11/14
–
–
N = 14
12-month
F/U
3-month follow-up to manualized
* p \ .05; ** p \ .005, two tailed
a
t(12) = 4.69**,
d = 1.30
t(11) = 3.12*, d = .90
t(11) = 1.14, d = .33
t(13) = 3.87**,
d = 1.03
t(13) = 3.53**, d = .98
t(11) = 4.17**,
d = 1.20
t(13) = 3.43**, d = .92
t(13) = 6.15**,
d = 1.64
Pre- 3-month F/U
t(13) = 3.02*, d = .81
t(11) = 3.63**,
d = 1.05
t(13) = 3.10*, d = .83
t(13) = 6.64**,
d = 1.78
Pre- post-treatment
Test statistics of individualized treatment
CSR, Clinician Severity Rating (ADIS); SCAS, Spence Children’s Anxiety Scale; CALIS, Child Anxiety Life Interference Scale
C = youth report, P = mother report
41.00
(16.65)
N = 14
12.36 (5.39)
5.93 (1.07)
N = 14
N = 14
4.93 (2.24)
N = 14
6.43 (1.22)
Pretreatmenta
Posttreatment
Pretreatment
SCAS-C
All
CSR
Primary
CSR
Mean (SD)
Mean (SD)
Posttreatment
Individualized treatment
Manualized treatment
Table 3 Continuous outcomes of manualized and individualized treatments
t(13) = -.16, d = -.04
t(10) = 2.38*, d = .72
t(13) = -1.17,
d = -.31
t(10) = .00, d = .00
–
–
Post- 12-month F/U
J Child Fam Stud (2016) 25:503–517
509
123
510
(Borenstein et al. 2009). Statistical analyses were carried
out using the Statistical Package for the Social Sciences
(SPSS) version 21.
Results
Outcomes on a Group Level
Table 1 shows categorical outcomes following the manualized and the individualized treatment. As assessed by the
CGI-I following the individualized treatment, nine youths
(64.3 %) had responded at post-treatment, and eleven
(78.6 %) at the 3-month follow-up. After the individualized treatment, eight (57.1 %) were free of their primary
diagnosis and six (42.9 %) were free of all anxiety diagnoses. At the 3-month follow-up, 11 (78.6 %) were free of
their primary diagnosis and nine (64.3 %) were free of all
diagnoses.
Table 3 shows the means and standard deviations on the
continuous measures, as assessed following both interventions, together with the test statistics for the pre-post,
pre-3 months and post-1 year follow-up comparisons for
the individualized treatment. The CSR for the primary
anxiety diagnosis, the CSR for the sum of all anxiety
diagnoses and the anxiety reported by youths and mothers
(SCAS-C/P) all showed a significant decrease from pre- to
post-treatment and from pre- to 3-month follow-up, with
large effect sizes (d = .81–1.78). A significant reduction
was seen in life interference from pre- to post-treatment
according to mothers, but not youths, while a significant
change from pre-treatment to the 3-month follow-up was
seen on both self-reports (CALIS-C/P). The treatment
gains of the individualized treatment remained stable from
post-treatment to the 1-year follow-up, with no significant
change on the anxiety levels (SCAS-C/P) or the life-interference according to mothers, only youths reporting a
significant decrease on CALIS-C.
Outcomes on an Individual Level
As shown in Table 1, three of the non-responders (ID 4, 7,
14) identified after the individualized treatment, subsequently responded at the 3-month follow-up. At the followup two of the youths (ID 5, 6) had been non-responders
since post-treatment, while the third non-responder (ID 13)
had relapsed in the follow-up period. After the individualized treatment, two of the six youths (ID 2, 3) with a
primary SoP diagnosis had remitted from their primary
diagnosis, while all five youths with a primary SAD
diagnosis (ID 7–11) had. At the 3-month follow-up, the
youths who had not remitted from their primary diagnosis
(ID 1, 2, 4) all had a primary diagnosis of SoP.
123
J Child Fam Stud (2016) 25:503–517
In Fig. 1 the self-reported anxiety levels are illustrated
for the manualized (T1–T3) and the individualized (T3–
T6) treatment for individual participants. Clinical cut-offs
are shown in the figure, along with statistically significant
changes for the individualized treatment. A significant
decrease in anxiety symptoms was found at post- treatment
(T4) for six youths (ID 2, 4, 5, 8, 11, 14) on SCAS-C, all of
them but one (ID 13) also having shown a clinically significant change. Eight youths (ID 2, 4, 5, 8, 9, 11, 13, 14)
showed a statistically and clinically significant change on
SCAS-P. A significant increase in anxiety symptoms was
found for one youth (ID 12) on SCAS-P. At 3-month follow-up (T5), when compared to pre-treatment, a significant
decrease in anxiety symptoms was found for six of the
youths (ID 4, 5, 7, 8, 11, 14) on SCAS-C, all of them but
one (ID 4) also having shown a clinically significant
change. Four youths (ID 4, 6, 8, 14) showed a statistically
and clinically significant change on SCAS-P. A significant
increase in anxiety was found for two youths (ID 7, 14) on
SCAS-P.
Families’ Satisfaction with Treatment
After the individualized treatment, almost all of the youths
(91.7 %) and all mothers indicated it was true that
‘‘treatment helped the youth’’, while the corresponding
percentages after the manualized treatment were lower
(youths 69.2 %, mothers 71.4 %). Youths’ evaluation of
the statement ‘‘I trusted my therapist’’ as true rose from
85 % following the manualized treatment to 100 % following the individualised treatment, while twice as many
youths believed it was true that ‘‘after therapy I felt more
like being with family and friends’’ following the individualized as opposed to the manualized treatment (67 vs.
31 %). Among mothers, the percentage who believed it
was true that ‘‘during therapy I managed being able to
change my behavior towards my child in a positive way’’
rose to 79 from 64 % that was following the manualized
treatment.
Most families described how the manualized treatment
had been helpful, as it offered them some techniques (e.g.,
It was really good to get some techniques that I had been
missing for a long time and was starting to be desperate)
and many families experienced the group setting of therapy
as positive (e.g., It was nice and deliberating not to be the
only ones with problems), but they felt it had not been
sufficient (e.g., It has been too short and there was not
enough time…we are afraid we cannot use the techniques
on our own after so little time), while after the individualized treatment they got the last bit they were missing:
(e.g., It gave us the last bit of understanding of the tools
applicability).
J Child Fam Stud (2016) 25:503–517
Fig. 1 Self-reported changes in
anxiety levels in manualized
and individualized treatments.
Note T1 = pre manualized,
T2 = post manualized, T3 = 3month F/U manualized/pre
individualized, T4 = post
individualized, T5 = 3-month
F/U individualized, T6 = 1year F/U individualized.
*Statistical significant change
on SCAS-C; statistical
significant change on SCAS-P.
– Change in negative direction
511
SCAS-C
SCAS-P
_ _ SCAS-P cut-off
… SCAS-C cut-off
80
70
60
50
40
30
20
10
0
ID. 1
T1
T2
T3
T
T4
T5
T6
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
ID. 3
T1
T2
T3
T4
T5
T6
ID. 5
T1
T2
T3
T4* †
T5*
T6
ID. 7
T1
T2
T3
T4
T5*
T6-†
ID. 9
T1
T2
T3
T
T4†
T5
T6 -†
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
ID. 2
T1
T2
T
T3
T4* †
T6 -* †
T5
ID. 4
T1
T2
T
T3
T4* †
T5* †
T6
T5†
T6
T5* †
T6
ID. 6
T1
T2
T
T3
T4
ID. 8
T1
T2
T3
T
T4* †
ID. 10
T1
T2
T3
T4
T5
T6
123
512
J Child Fam Stud (2016) 25:503–517
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
ID. 11
T1
T2
T3
T4* †
T5*
T6
ID. 13
80
70
60
50
40
30
20
10
0
80
70
60
50
40
30
20
10
0
ID. 12
T1
T2
T3
T4 -†
T5
T6*
ID. 14
Fig. 1 continued
Discussion
The aim of this study was to evaluate the effectiveness of
an individualized case formulation-based CBT intervention
for youths with anxiety disorders, who had not responded
to a manualized group CBT. Nine of the 14 youths
responded to the individualized treatment, as measured on
the CGI-I at post-treatment, and this number increased to
11 at the 3-month follow-up. Youths’ anxiety levels
decreased significantly with large effect sizes after the
individualized treatment (post-treatment and 3-month follow-up). The positive outcomes remained stable until the
1-year follow-up.
Even though all outcome measures showed positive
effects on a group level, when examined on an individual
level, the clinicians’ evaluation (ADIS-IV, CGI-I) was not
always in agreement with the self-reported changes in
anxiety (SCAS-C/P). For instance, three of the non-responders, identified after the individualized treatment (ID
4, 5, 14), reported decreased anxiety levels that were statistically and clinically significant. Youths and mothers
reported similar changes in anxiety symptoms during the
two interventions; the greatest differences often being prior
to the manualized treatment (ID 6, 8, 9) and at the 1-year
follow-up to the individualized intervention (ID 2, 7, 14).
Besides a decrease in anxiety symptoms, the individualized treatment contributed also to a decrease in perceived
life-interference due to anxiety. Usually impairment brings
clients to seek treatment (Angold et al. 1999) and a
decrease in life-interference is likely to have the strongest
impact on satisfaction with the treatment (Lyneham et al.
123
2013). The families were highly satisfied with the individualized treatment, which they believed had helped the
youths. Compared to the manualized treatment, more
youths liked being with family and friends after the individualized treatment, and more mothers believed they had
been able to change their behavior towards their child in a
positive manner.
The participants most commonly had a primary SoP
diagnosis or a SAD diagnosis. Youths with primary SAD
appeared to benefit a lot from the individualized treatment,
since all of them were diagnosis-free at post-treatment and
at the 3-month follow-up. Therapists often involved parents
of youths with SAD to a large extent in treatment, since
youths’ symptomatology was directly related to their parents. Instead of reinforcing and maintaining the youths’
anxiety by reassuring them nothing bad would happen to
them, parents were guided on how to encourage their child
to become more independent. Parents would encourage
their child to use the treatment techniques, while elements
of contingency management and transfer of control were
emphasized. This kind of active parental involvement has
also earlier been associated with better long-term outcomes
(Manassis et al. 2014). In contrast, three of six youths with
a primary SoP diagnosis still had their primary diagnosis at
the 3-month follow-up. SoP has been associated with worse
treatment outcomes after manualized CBT (e.g., Hudson
et al. 2015) and this was also found for the participants of
the manualized treatment from which non-responders were
identified for our study (Arendt et al. 2015). The individual
setting enabled the addition of disorder-specific techniques
for youths with SoP, such as video-feedback (Harvey et al.
J Child Fam Stud (2016) 25:503–517
2000) and social skills training (Beidel et al. 2005). Even
though some of the youths were not diagnosis-free after the
end of the individualized treatment, the severity of their
anxiety had in most cases decreased.
The three non-responders, who even after the individualized treatment and the additional 3 months after therapy
still had not responded to treatment, were among the older
participants in the study and two of them had been offered 20
sessions of individualized CBT each. Following information
collected after the conclusion of therapy, parents of two of
the youths reported that their child had been diagnosed with
another disorder (epilepsy and borderline personality disorder) that could better explain their symptomatology.
Overall, most of the youths classified as non-responders,
who underwent additional individualized treatment, had to
some degree progressed during the manualized treatment
(i.e., most common CGI-I rating was minimally improved).
Families were able to learn techniques in a cost-effective
format and might have profited from non-specific group
factors (Yalom 1975), such as group cohesiveness, interpersonal learning and ‘‘universality’’ (not being alone with
the problems). However, the families experienced the
manualized treatment as too short and felt unable to continue working on their own, while the therapy format had
not allowed the clinician to include additional treatment
elements in case of non-anxiety difficulties, or work more
closely with every family. The individualized therapy
allowed for the flexibility to tailor treatment components to
each family’s specific needs. The focus of the individualized treatment was to enhance the families’ understanding
of the techniques, in order for them to be able to work more
independently, and to increase the amount of time youths
spent on exposures, which is considered the primary active
ingredient of anxiety treatment (Barlow 2002; Crawley
et al. 2012). This was done through in vivo exposures
during sessions, which also allowed clinicians to address
any subtle avoidance behaviors and coach parents on how
to respond to their child when anxious by using the treatment skills. Clinicians also emphasized that youths should
carry out exposures between sessions; following up more
closely on the families’ homework, problem solving with
them when obstacles were encountered, and working on the
parents’ tendency to overprotect their child, thereby creating natural exposures for the youth.
Results of our study must be viewed in the light of
methodological limitations, as there was a lack of a control
group, a small sample size and the CGI-I assessments were
conducted by the therapists. Nevertheless, our study holds
promising findings of clinical relevance, as it showed how
non-responders to manualized CBT may respond to treatment, when offered CBT tailored to their needs. In case of
non-response to CBT, when examining the factors, which
may influence treatment gains, decision-making maps
513
could facilitate this process (see Marder and Chorpita
2009). When clinicians create or revise the individual case
formulations, information from the first treatment can be
integrated, facilitating the planning of the individualized
treatment. This approach encourages a collaborative stance
towards the family, while therapists can flexibly address
difficulties families may encounter during therapy. What
seemed to contribute to the youths’ response to treatment
was the enhancement of their exposure to anxiety-provoking situations. In case of non-response to this extra
intervention, clinicians may need to consider whether
another primary disorder than anxiety better explains the
youths’ difficulties and another treatment form is more
suitable. Further research is needed to develop empirically
supported guidelines on the development of case formulations and to examine their effectiveness among non-responding youths with anxiety disorders.
In conclusion, the individualized case formulation-based
CBT overall proved to be effective in treating youths with
anxiety disorders, who had not profited sufficiently from
manualized CBT. The majority of youths responded to the
treatment and most of the youths were free of their anxiety
diagnoses at post-treatment and the 3-month follow-up.
The self-reported anxiety and life interference showed
significant changes with large effect sizes following the
individualized treatment and the positive outcomes were
maintained at the 1-year follow-up.
Acknowledgments The authors would like to acknowledge the
financial support of this research by TrygFonden (Grant ID No.
10691), who had no further role in the study or in the decision to
submit the article for publication. Furthermore, the authors would like
to thank Lisbeth Jørgensen, Signe Matthiesen, Kristian Bech Arendt,
and Marianne Bjerregaard Madsen for their contributions to the
implementation and evaluation of the study.
Appendix: A Case Vignette
Presenting Picture
Lise (fictitious name; ID 8) was a 13-year old girl, who
attended 6th grade and lived with both of her parents. She
was the youngest of four children, one of which had moved
from home. Her parents contacted the Anxiety Clinic
because Lise was afraid something horrible would happen
to her mother. For instance, she was afraid that her mother
might die in a car accident or from a heart attack. This
negatively affected her school attendance, social activities
with peers and the family life, because she wanted to be
with her mother at all times. From the diagnostic interview,
it was concluded that Lise had a primary SAD diagnosis, a
comorbid generalized anxiety disorder and panic disorder
(see Table 1). Parents reported feeling stressed, since both
123
514
J Child Fam Stud (2016) 25:503–517
A
PERSONAL MAINTAINING FACTORS
Psychological factors
- Avoidance behaviour
- Anxiety escalates fast and youth is
therefore unable to use treatment
techniques
- Cognitive difficulties due to comorbid
ADHD
- Immature defence mechanisms
(idealization of mother and devaluation of
father) contribute to conflicts at home
PERSONAL PROTECTIVE
FACTORS
Psychological factors
- Normal intelligence
Anxiety
CONTEXTUAL PROTECTING
FACTORS
Treatment system factors
- Family accepts there is a problem and
are committed to resolving the problem
- Good relation to therapist in Clinic
CONTEXTUAL MAINTAINING
FACTORS
Family system factors
- Father attends treatment
Family system factors
- Stressed parents, difficult to prioritize
practicing between sessions
- Problematic reinforcement of avoidance
behaviour (lack of school attendance is
rewarded with time with the mother)
- Problematic relation to father
- Overinvolved mother-child relation
Social network factors
- Good relations to the rest of the family
- Good school placement
B
WHAT WE NEED TO DO MORE
- Create realistic plans
- Choose few techniques that work
- Use techniques consistently
- Complete more homework (daily plans)
- Follow-up on homework very closely in
sessions
-Practice staying in situation until anxiety
subsides
- Give rewards consistently
- Spend time with both mum and dad
- Work on becoming more independent
- Dad and mum learn how to let go in a good
way
PROTECTIVE FACTORS
Anxiety
-Smart girl
- Liked by others
- Creative
- Works well with the techniques
- Therapy was successful– it
works
- Parents are supportive
- Good relationship to older
sisters
- Good school that is supportive
- Friends
Parents’ suggestions:
- Mother spends more time outside the house
- Work on Lise’s catastrophic thoughts
Fig. 2 Case formulation of Lise. a Case formulation as presented at clinical staff meeting. b Case formulation as presented to family
were working fulltime, and three of their children had an
ADHD diagnosis that demanded a lot of attention.
History
At the intake interview, parents reported that Lise
showed the first signs of separation anxiety when she
123
had to start at early childhood care. Her difficulties
escalated in the 3rd grade, when she stayed home from
school for half a year. She underwent psychiatric evaluation that resulted in an ADHD diagnosis for which she
received medication. Lise also saw a psychologist for 12
sessions and was helped to gradually attend a new
school. Nevertheless, she was only able to attend the
J Child Fam Stud (2016) 25:503–517
new school occasionally and only if her mother accompanied her.
Manualized Treatment
Lise had difficulties completing tasks in the group, as
because of her ADHD she became easily distracted. Furthermore, the family had difficulties completing exercises
on cognitive restructuring between sessions, because Lise
became irritated with her father, when he posed questions
that were meant to challenge her erroneous attributions, as
she felt he did not take her difficulties seriously. On the
other hand, the mother had a tendency to reassure Lise,
instead of challenging her worries. As the mother herself
reported, sometimes it was easier to stay home from work,
rather than having to deal with an extremely anxious child.
Therefore, the parents had very different ways of handling
her anxiety. Lise idealized the mother and devaluated the
father, demanding for instance that he (not the mother)
should sit behind the wheel, so he would be the one killed
in a possible car accident. The family was introduced to the
principles of graduated exposures, but it was difficult for
them to practice systematically, because of a hectic and
chaotic everyday in the family. The therapist reported
having difficulties to follow-up on the family’s work as
closely as needed, while there was not enough time to
address the problematic family dynamics in a group setting.
Outcome of Manualized Treatment
Lise made some progress during the manualized treatment, as
she started spending more time with her father and on ‘‘good
days’’ she would go to school alone. She and her mother
reported decreased anxiety levels after the end of treatment
(see Fig. 1). Nevertheless, the diagnostic interviews at posttreatment and at the 3-month follow-up indicated she had not
remitted from her anxiety diagnoses and she was classified as a
non-responder (see Table 1), so she was offered further
treatment. Lise’s case formulation, as presented at the clinical
staff meeting and the family, is displayed in Fig. 2.
Individualized Treatment
The treatment consisted of eight sessions, the first four
every week and the remaining every other week. From
therapy start, the family’s homework was closely monitored, the therapist following up at each session the entries
on exposure work made by the family, in the booklet they
were given. Lise had difficulties with completing the
exposure exercises due to her anxiety escalating very
rapidly, making it hard for her to use the techniques.
Interoceptive exposures were practiced in the session and
Lise at first laughed when seeing the therapist
515
hyperventilating, then when she started to hyperventilate,
she felt dizzy, got scared and thought: this will end badly.
She was encouraged to challenge her catastrophizing
thoughts and she got a cue card with the alternative
thought: I have some techniques I can try out. I am sure I
can make this stop. Lise made progress in staying home for
longer intervals and when she would get thoughts such as:
what if they never come home? They could be dead, she
tried to ignore them by focusing on what she was doing.
When she got anxious in school, she reported tackling the
butterflies in the stomach by trying to breathe more calmly,
as she was taught to do in therapy sessions. She commented
on her progress: Now I am a bit more like the others, doing
the same things as them. Nevertheless, Lise would easily
become discouraged and it was hypothesized that the
ADHD contributed to her difficulties in having an overview
of her progress and drawing learning from her experiences,
negatively impacting her motivation. She was therefore
given a success-diary in which she would write down her
success-experiences and what she had learned. The individualized format allowed the therapist to spend some time
with the parents alone during the sessions, where behaviors
that contributed to the maintenance of Lise’s anxiety were
discussed. During those sessions, a trained graduate student
would conduct in vivo exposures with Lise, where she
would practice taking the bus. The parents developed a
more consistent way of handling her anxiety, assisting her
in the implementation of techniques and praising her for
bravery. Instead of creating ‘‘stepladders’’ of graduated
exposures, they were presented with an alternative graphical presentation of behavioral experiments that was more
flexible and easy for them to follow.
Outcome of Individualized Treatment
Lise made great progress during the individualized treatment, as she for instance became able to stay home alone for
2 h and she ended up taking the bus alone to school on a daily
basis. At post-treatment and at the 3-month follow-up, Lise
was classified as a responder (see Table 1). Self-reports
showed that Lise and her mother experienced a significant
decrease in anxiety levels, which remained low at the
3-month and at the 1-year follow-up assessments (see
Fig. 1). The mother evaluated the therapy they had received:
It [the individualized treatment] was intense…but
also good. We had already learned the techniques
and gotten a lot out of being together with the others.
Now we needed to work more intensively and it was
very good that it was always adjusted in order to fit
exactly to what Lise needed. It wouldn’t have helped
being in a group again…We needed this continuous
monitoring in order to get to the bottom of
123
516
things…breathing exercises might for instance not be
something all children need, but we couldn’t get any
further, until Lise learned to tackle the symptoms.
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