Introduction
Autism spectrum disorder (ASD) is a neurodevelopmental disorder characterised by impaired social interaction and communication, repetitive behaviour and restricted interests (DSM-5, American Psychiatric Association, 2013). Furthermore, ASD can entail maladaptive responses due to difficulties with social communication and repetitive behaviours (DSM-5). It is understandable that due to the areas of impairment people with ASD may experience, rates of anxiety are high in this population. A meta-analysis of 31 studies found that approximately 40% of children with ASD met the criteria for anxiety disorders (van Steensel et al., Reference Van Steensel, Bogels and Perrin2011). However, ASD patients who undergo cognitive behaviour therapy (CBT) to reduce anxiety may also find that the problematic characteristics of their disorder interfere with recovery (Murray et al., Reference Murray, Jassi, Mataix-Cols, Barrow and Krebs2015). On the other hand, recent studies have shown that CBT is effective in this context. For instance, a meta-analysis of 14 studies with moderate effect sizes (g = –0.71) suggested that CBT effectively reduced anxiety in ASD patients (Ung et al., Reference Ung, Selles, Small and Storch2015), while a later meta-analyses of 23 studies supported these findings (moderate effect size; g = –0.66) (Perihan et al., Reference Perihan, Burke, Bowman-Perrott, Gallup, Thompson and Sallese2019). In addition, a previous randomised controlled trial (RCT) for CBT for co-morbid obsessive-compulsive disorder (OCD) in high-functioning ASD has reported within-group effect sizes of 1.01 for the CBT group (Russell et al., Reference Russell, Jassi, Fullana, Mack, Johnston, Heyman, Murphy and Mataix-Cols2013). To summarise the results of previous studies, CBT appears to be effective in treating anxiety in patients with ASD (Perihan et al., Reference Perihan, Burke, Bowman-Perrott, Gallup, Thompson and Sallese2019; Ung et al., Reference Ung, Selles, Small and Storch2015), but less effective in patients without ASD (Tsuchiyagaito et al., Reference Tsuchiyagaito, Hirano, Asano, Oshima, Nagaoka, Takebayashi, Matsumoto, Masuda, Iyo, Shimizu and Nakagawa2017).
In addition, cases involving high-intensity CBT lasting 1–2 h per session may be a significant burden to ASD patients, thus leading to high drop-out rates. Here, even 1 h may present a substantial burden for these patients, who have trouble learning new things and/or interacting on an interpersonal level. It is therefore important to address potential barriers to therapy and by doing so reduce drop-out rates while increasing treatment satisfaction. For example, previous research involving patients without ASD found that remote treatments implemented through video conferencing and visual aids to explain complex CBT models resulted in an 81% (24/29) patient satisfaction rate (Matsumoto et al., Reference Matsumoto, Sutoh, Asano, Seki, Urao, Yokoo, Takanashi, Yoshida, Tanaka, Noguchi, Nagata, Oshiro, Numata, Hirose, Yoshimura, Nagai, Sato, Kishimoto, Nakagawa and Shimizu2018). Providing CBT in an acceptable manner for patients with ASD may provide a guide for future treatment. One promising treatment approach is blended CBT, which combines digital aid materials and/or computer programs with built-in human interactions (e.g. psychoeducation) in addition to support for therapists in capturing abstract thought processes (Nakao et al., Reference Nakao, Nakagawa, Oguchi, Mitsuda, Kato, Nakagawa, Tamura, Kudo, Abe, Hiyama, Iwashita, Ono and Mimura2018; van der Vaart et al., Reference van der Vaart, Witting, Riper, Kooistra, Bohlmeijer and van Gemert-Pijnen2014). Blended CBT is a simple, low-intensity application that can be offered as a manualised guided self-help intervention. A meta-analysis of 64 studies on internet-based CBT for anxiety disorders (including panic disorder, PD) found that such applications were equally as effective as face-to-face CBT (Andrews et al., Reference Andrews, Basu, Cuijpers, Craske, McEvoy, English and Newby2018). Blended CBT is also known to significantly improve symptoms in Japanese patients with depression (Nakao et al., Reference Nakao, Nakagawa, Oguchi, Mitsuda, Kato, Nakagawa, Tamura, Kudo, Abe, Hiyama, Iwashita, Ono and Mimura2018). However, none of these studies examined whether blended CBT improved anxiety among adolescent ASD patients with PD, nor have any assessed what adaptations must be made to apply the treatments.
Central coherence, cognitive flexibility and theory of mind have been reported as common cognitive dysfunctions among ASD patients (Velikonja et al., Reference Velikonja, Fett and Velthorst2019; Westwood et al., Reference Westwood, Stahl, Mandy and Tchanturia2016; Yirmiya et al., Reference Yirmiya, Erel, Shaked and Solomonica-Levi1998). ASD is also associated with poor emotional perception (Uljarevic and Hamilton, Reference Uljarevic and Hamilton2013), while many patients find it difficult to express their emotional states and thought processes (Moseley et al., Reference Moseley, Shtyrov, Mohr, Lombardo, Baron-Cohen and Pulvermüller2015). These patients also have problems with ‘belonging’ mental states (e.g. desires and beliefs) regarding the self and/or others (Frith et al., Reference Frith2001). ASD patients may thus have difficulty gaining insight into their own mental health issues. That is, they may not fully convey emotions or cognition to clinicians, thus increasing the possibility of receiving ineffective treatment. It is therefore important for therapists to understand the cognitive traits of ASD when delivering CBT to ASD patients.
PD describes a condition in which unexpected panic attacks repeatedly occur, while agoraphobia induces fear and anxiety over undesired stimuli in certain locations (APA, 2013). Patients with these conditions often avoid public transportation, very large areas, and/or enclosed spaces. However, a previous systematic review (including a meta-analysis of RCTs) showed that CBT was effective for treating PD (Hofmann and Smits, Reference Hofmann and Smits2008). Furthermore, the National Institute for Health and Care Excellence (NICE) guidelines state that CBT is recommended as a first-line treatment in conjunction with selective serotonin re-uptake inhibitors (SSRIs) for PD patients (NICE, 2011). Japanese clinical trials conducted by Seki et al. (Reference Seki, Nagata, Shibuya, Yoshinaga, Yokoo, Ibuki, Minamitani, Kusunoki, Inada, Kawasoe, Adachi, Yoshimura, Nakazato, Iyo, Nakagawa and Shimizu2016) and Matsumoto et al. (Reference Matsumoto, Sutoh, Asano, Seki, Urao, Yokoo, Takanashi, Yoshida, Tanaka, Noguchi, Nagata, Oshiro, Numata, Hirose, Yoshimura, Nagai, Sato, Kishimoto, Nakagawa and Shimizu2018) also reported that 80% (16/20) and 50% (5/10) of PD cases were in remission, respectively. However, no patients in these studies had developmental disorders such as ASD. In addition, related systematic reviews on the effectiveness of internet-based cognitive behavioural therapy (ICBT) for PD were solely conducted in Western contexts (Andrews et al., Reference Andrews, Basu, Cuijpers, Craske, McEvoy, English and Newby2018). This makes it even more important to examine the feasibility of blended CBT for PD among East Asian patients with ASD. This case study thus implemented blended CBT treatments with a Japanese ASD patient who had both PD and agoraphobia.
Method
Presenting problem
The patient examined in this study was a girl named Lucy, who was 17 years of age at the time of analysis. Lucy lived with her mother and younger brother, as her father and mother divorced when she was a baby. Lucy had experienced difficulty interacting with others since childhood, and did not have any friends. She willingly chose to attend a correspondence-based high school. In X year, she was unable to go out or use public transportation due to the onset of panic attacks while eating out. She later visited a general practitioner and was offered medication (Diazepam 2.00 mg), but strongly disliked taking the medicine and did not attend any follow-up visits. She then underwent a psychological examination at another hospital. Lucy also received counselling at two different times (non-CBT), which she interrupted due to the lack of results. In fact, her PD symptoms failed to improve, while her agoraphobia actually worsened. Lucy eventually became withdrawn. It was thus essential for her mother to accompany her whenever leaving the home. Lucy was eventually referred to our hospital in X+1 year.
Prior to implementing CBT, the first author conducted interviews with Lucy during an assessment session based on the Mini-International Neuropsychiatric Interview (MINI) (Muramatsu et al., Reference Muramatsu, Miyaoka, Kamijima, Muramatsu, Yoshida, Otsubo and Gejyo2007; Sheehan et al., Reference Sheehan, Lecrubier, Sheehan, Amorim, Janavs, Weiller, Hergueta, Baker and Dunbar1998; Sheehan et al., Reference Sheehan, Lecrubier and Otsubo2010;). At that time, Lucy sufficiently met the criteria for both PD and agoraphobia diagnoses as described in the DSM-5. The severity of her PD was specifically diagnosed as ‘markedly ill’ based on a score of 18 on the Panic Disorder Severity Scale (PDSS) (Furukawa et al., Reference Furukawa, Katherine, Barlow, Gorman, Woods, Money, Etschel, Engel and Leucht2009). She was assessed using the Wechsler Adult Intelligence Scale-III (WAIS-III) (Wechsler, Reference Wechsler1997; Fujita et al., Reference Fujita, Maekawa, Dairoku and Yamanaka2006) to ascertain her Intelligence Quotient (IQ) prior to treatment. Her Full-Scale IQ was 81, with a low average on the WAIS-III (Table 1). Furthermore, her cognitive functions were evaluated using the Rey Complex Figure Test for central coherence, Wisconsin Card Sorting Test for cognitive flexibility, and a social cognition screening questionnaire (Table 1). With an ADOS-2 clinical qualification, the first author then conducted the Autism Diagnostic Observation Schedule, Second Edition (Lord et al., Reference Lord, Rutter, DiLavore, Risi, Gotham and Bishop2012) to assess Lucy for ASD; her score reached the cut-off of 9 points. Furthermore, her Autism-Spectrum Quotient (AQ) (Baron-Cohen et al., Reference Baron-Cohen, Wheelwright, Skinner, Martin and Clubley2001; Wakabayashi et al., Reference Wakabayashi, Tojo, Baron-Cohen and Wheelwright2004) score was 39, thus exceeding the cut-off point of 33. However, her Liebowitz Social Anxiety Scale (LSAS) score was 49, which did not meet the cut-off score of 60 (Mennin et al., Reference Mennin, Fresco, Heimberg, Schneier, Davies and Liebowitz2002).
Table 1. Summary of Lucy’s scores
WAIS-III, Wechsler Adult Intelligence Scale; Rey, Rey Complex Figure Test; SCSQ, Social Cognition Screening Questionnaire; AQ, Autism-Spectrum Quotient; LSAS, Liebowitz Social Anxiety Scale.
a Median of healthy Japanese (Hamatani et al., Reference Hamatani, Tomotake, Takeda, Kameoka, Kawabata, Kubo, Ohta, Tada, Tomioka, Watanabe, Inoshita, Kinoshita and Ohmori2018);
b median of healthy Japanese from 15 to 19 years old (Okazaki et al., Reference Okazaki, Saeki and Hachisuka2013);
c average value of healthy Japanese (Hamatani et al., Reference Hamatani, Tomotake, Takeda, Kameoka, Kawabata, Kubo, Tada, Tomioka, Watanabe and Ohmori2016).
Thus, Lucy’s primary diagnoses based on the DSM-5 were ASD, PD and agoraphobia. Lucy preferred to receive CBT alone (without medication) during the course of the sessions. We therefore agreed to implement CBT for panic disorder and agoraphobia.
Measures
Lucy completed assessments designed to investigate her level of panic during daily life at each CBT session to provide treatment effectiveness data. The main results were then measured using the PDSS (Houck et al., Reference Houck, Spiegel, Shear and Rucci2002; Katagami, Reference Katagami2007).
Treatment goals
Lucy’s short-term treatment goals were to be able to watch movies, shop, and enter public vehicles (e.g. trains) while alone. She stated the following as a medium-term goal: ‘I want to be able to go to restaurants and go to the theme park with my mother’. She also set a long-term goal of completing domestic travel. Finally, Lucy’s therapeutic goal was primarily involved with improving her agoraphobia. This is probably because she was not experiencing daily panic attacks and was not aware of her self-anticipatory anxiety. Through psychoeducation and a case formulation process guided by the therapist, Lucy eventually became aware of the difficulties related to panic (e.g. catastrophic misconceptions about bodily sensations and safety behaviours).
Case formulation
CBT models for treating PD show that patients with PD misunderstand normal somatic sensations, such as breathing and dizziness; this can lead to panic attacks (Clark, Reference Clark1986). The CBT model is intended to help patients with PD understand that central to their difficulties is the catastrophic misunderstanding of bodily sensations and to challenge this through a range of behavioural and cognitive methods. Therefore, a simplified case formulation was collaboratively developed based on the Seki and Shimizu model (Matsumoto et al., Reference Matsumoto, Sato, Hamatani, Shirayama and Shimizu2019) during session 3 (Fig. 1). Figure 1 shows a schematic diagram of the first panic attack, which occurred while on a train. At that time, Lucy felt ‘unreal’ and short of breath. She seemed to have catastrophically interpreted these bodily sensations as evidence of feeling faint. As such, the image of her fainting was taken as fact, thus increasing her anxiety symptoms. Lucy has avoided the train since. The therapist and Lucy confirmed that the more Lucy pays attention to her internal senses, the more Lucy notices anxiety. It seemed that avoiding places or situations in which Lucy had occurred panic attack was actually maintaining Lucy’s agoraphobia.
Figure 1. Case formulation.
Course of therapy
Lucy resisted typical CBT treatments consisting of weekly 50 min sessions, but agreed to 20 min CBT sessions every other week. Blended treatment entails that face-to-face contacts may be added to internet interventions, or vice versa. Here, an internet-based portion may be arranged as an adjunct to existing face-to-face programmes (Erbe et al., Reference Erbe, Eichert, Riper and Ebert2017). After completing the treatment session, the therapist saved the treatment programme as a visual slide on Lucy’s USB drive so that Lucy could continue her learning by using the internet-based materials even while offline. This also meant Lucy could re-watch the same materials at any time. The first and second authors (clinical psychologists who were familiar with CBT) conducted peer supervision at each session.
As mentioned earlier, this study’s cognitive behavioural model was based on the Seki and Shimizu model (Matsumoto et al., Reference Matsumoto, Sato, Hamatani, Shirayama and Shimizu2019); the therapist also innovatively conducted CBT, taking into consideration the cognitive nature of autism (Tables 1 and 2). As far as the authors know, there are no previous practical reports on the Seki and Shimizu model for use in treating panic disorder and agoraphobia in children, adolescents or ASD patients. The therapist thus facilitated Lucy’s understanding, expressions and ability to change her behaviour using various strategies including visual aids, modelling, adjusted progress speed (e.g. through repetition) and a detailed plan of exposure and response prevention (ERP). The therapist role played what Lucy did at those times or presented relevant slides to help her recognise her thoughts during panic attacks. Lucy and the therapist did not include the public opinion survey because Lucy was not interested in how others would assess her condition.
Table 2. Treatment components
People with ASD sometimes have difficulties identifying their own and others’ emotional state (Uljarevic and Hamilton 2013) and to express their own thought processes and emotions (Moseley et al., Reference Moseley, Shtyrov, Mohr, Lombardo, Baron-Cohen and Pulvermüller2015). In addition, people with ASD are not good at belonging mental states such as desires and beliefs to the self (or others) (Frith, Reference Frith2001). This feature may be affected by cognitive impairments entailing poor metacognition and theory of mind. In other words, it may be difficult to gain insight into mental health issues, which may not fully convey emotions and cognitions to the clinician and may not receive appropriate treatment. Lucy did not initially mention self-focused images, but the therapist later provided descriptions of images that many patients experience during episodes of panic (e.g. ‘die, or collapse with a heart attack’). Lucy chose the image of ‘faint’, saying that she had experienced a related self-image. With regard to making catastrophic interpretations of body sensations, Lucy said, ‘I don’t think so far’. This made it seem like Lucy did not make catastrophic interpretations at all. The therapist thus presented many closed-ended questions to detect her catastrophic misinterpretation of bodily sensations. Lucy relayed the following: ‘If my head is hard, I might faint’. The therapist understood these words to describe a feeling of losing consciousness. When actually presented with a hard situation, Lucy was more confused by her fear of ‘fainting’ than ‘I might be in pain and faint’. Lucy and the therapist thus shared what Lucy would usually say such things (i.e. ‘I don’t think so much’).
During regular attention training, patients focus on (1) external information (e.g. colour, sound, smell and tactile sensation) and (2) then alternate between internal information (e.g. breathlessness and palpitations) and external information (Matsumoto et al., Reference Matsumoto, Sato, Hamatani, Shirayama and Shimizu2019). Lucy’s poor imagination was remarkable during attention training. Lucy would not conduct attention training after seeing the visual slide, even when she was instructed to do this in front of the therapist. However, Lucy was able to do this when the therapist first showed her the model. In general, only one attention training session is conducted. However, Lucy refused to enter the next stage of the behavioural experiment by saying ‘still early’. This may have been caused by her ASD characteristics, which made it difficult to accept minor changes. Lucy’s poor cognitive flexibility may have also been an influencing factor in this regard. The therapist thus conducted three attention training sessions as a way of adjusting progress speed.
Outcome
The results of PDSS outcome were measures at pre- to 8 months of follow-up (see Fig. 2). Psychological education and attention training were conducted in the first half of the intervention, and during the middle of the treatment, Lucy and the therapist went up the stairs in the hospital repeatedly for behavioural experiments against the catastrophic misinterpretation of bodily sensations. In the second half of the treatment, Lucy and the therapist created the anxiety hierarchy table, and Lucy was gradually exposed to agoraphobia-causing situations, in which Lucy exhibited less anxiety. After intervention, Lucy’s score on the PDSS was 2; a decrease of 16 points from the baseline; PD severity was normal (Furukawa et al., Reference Furukawa, Katherine, Barlow, Gorman, Woods, Money, Etschel, Engel and Leucht2009). In the 8 months of follow-up after the intervention, the PDSS score was 0. This PDSS total scores indicates that the symptoms of panic and agoraphobia were completely remitted. With the reduction of agoraphobia, Lucy can now go out to the following places and situations: after treatment, Lucy was able to use public transport and go out alone; use trains, buses and elevators; be in narrow places, restaurants, movie theatres, grandparent’s house, live shows, supermarkets, department stores and shopping streets.
Figure 2. PDSS outcome measures from pre-study to 8-month follow-up.
Discussion
This study found that it was feasible to conduct blended CBT for PD symptoms in ASD patients. Specifically, this case study used the Seki and Shimizu model (16 weekly sessions; 50 min each) to conduct blended CBT. The therapist modified this by specifically conducting a total of sixteen 20-min sessions every other week. This was done because the patient (Lucy) indicated difficulty when conducting 50-min weekly sessions in the hospital setting. That is, Lucy said she was unable to visit the hospital alone and that long treatments were burdensome. As such, blended CBT was the only successful treatment for Lucy. Blended CBT should help patients learn how to use online modules in a stimulating way; this entails developing therapies to reflect individual patient needs (van der Vaart et al., Reference van der Vaart, Witting, Riper, Kooistra, Bohlmeijer and van Gemert-Pijnen2014). The therapist in this study helped Lucy learn and develop her skills based on attentive cognitive behavioural science (e.g. carefully choosing which modules to use and considering cognitive characteristics that are unique to ASD patients). Typical individual CBT sessions last 45–90 min. However, Lucy’s treatment times were reduced to between 20 and 30 min each. The ICBT further suggests that therapists can help with PD and agoraphobia through short-term assistance when intervening in patients with cognitive handicaps. This study found that blended CBT was practical for treating PD and agoraphobia in an ASD patient.
A systematic review of 27 RCTs for patients with mental disorders showed that blended CBT was both feasible and effective when compared with a no-treatment control (Erbe et al., Reference Erbe, Eichert, Riper and Ebert2017). Blended CBT has also been reported to establish therapeutic alliances (Vernmark et al., Reference Vernmark, Hesser, Topooco, Berger, Riper, Luuk, Backlund, Carlbring and Andersson2019). In addition, although a previous study examined the treatment elements of CBT for anxiety in ASD patients (Ung et al., Reference Ung, Selles, Small and Storch2015), we know of no studies that have investigated how to treat (i.e. how treatment elements and innovations apply) ASD-specific cognitive disabilities in PD patients. Our results first suggest that blended CBT should be feasible for treating anxiety and ASD around the world. Reports have shown that ASD patients can successfully use social media to reduce interpersonal anxiety (van Schalkwyk et al., Reference van Schalkwyk, Marin, Ortiz, Rolison, Qayyum, McPartland, Lebowitz, Volkmar and Silverman2017). Notably, blended CBT integrates fewer interpersonal factors than face-to-face CBT, thus allowing ASD patients to address issues in a way that promotes therapeutic effects.
Cognitive function tests indicated that Lucy had weak central coherence (Table 1). Here, the central integration feature is weak when integrating whole stations, but emphasises details. It is thus necessary to encourage these patients to consider threats not only from negative aspects, but also based on total events. For example, when evaluating Lucy’s fear of everything based on partial anxiety, the therapist asked, ‘How do you see from various aspects?’; Lucy only answered ‘all are the same’. However, checking the facts one by one, the therapist asked ‘How about this part?’ and Lucy thereby realised that the horrible event was not the full nature of the experience, thus decreasing her anxiety. This shows that it is practical to perform blended CBT while intervening in cognition disabilities that are specific to ASD.
From a cost-effective perspective, blended CBT may also reduce the per-treatment direct medical costs when compared with regular CBT. This is because face-to-face sessions are replaced with a self-help programme (Kooistra et al., Reference Kooistra, Wiersma, Ruwaard, van, Smit and Lokkerbol2014). In short, by reducing the time or number of required one-to-one sessions, blended CBT enables therapists to accept greater numbers of patients. In this study, one 20-min session was provided every other week. Simply calculated, this required about one-third the time of a regular 45- to 90-min CBT session. Previous RCTs among Japanese patients with major depressive disorders have indicated that blended CBT was the most effective and promising for trainee therapists; using web-based support, therapists can more confidently implement CBT, even with relatively less experience (Nakao et al., Reference Nakao, Nakagawa, Oguchi, Mitsuda, Kato, Nakagawa, Tamura, Kudo, Abe, Hiyama, Iwashita, Ono and Mimura2018). Hence, greater numbers of patients may be treated as it becomes easier for doctors to request treatment sessions with less-experienced therapists.
Summary
Lucy was an ASD patient with both PD and agoraphobia. We conducted blended CBT using digital teaching materials following the Seki and Shimizu CBT model (Matsumoto et al., Reference Matsumoto, Sato, Hamatani, Shirayama and Shimizu2019). The leading author as the therapist was able to support Lucy in real time. Lucy was able to improve her panic symptoms both pre- and post-intervention and did not report any adverse events. Previous findings support the effectiveness of CBT for treating anxiety in ASD patients (Zaboski et al., Reference Zaboski and Storch2018). This study’s results further show the viability of blended CBT for PD patients with ASD. Japanese clinicians and certified psychologists should not hesitate to provide CBT to these patients.
Acknowledgements
The authors would like to sincerely thank Lucy for her consent to publish information about her treatment.
Financial support
This work was supported by JSPS KAKENHI grant numbers 18K17313 and 19J00227. The funding source had no role in the design or conduct of the study.
Conflicts of interest
The authors have no conflicts to declare.
Ethical statement
The authors abided by the Ethical Principles of Psychologists and Code of Conduct, as stipulated by the APA. The authors did not seek ethical approval as this was a single case study. However, informed written consent was obtained from the patient along with permission to publish this case.
Disclaimers
We assert that the views expressed in this article are our own and thus not official positions of the institution or funder.
Key practice points
(1) Cognitive behavioural therapy (CBT) can be used for patients with autism spectrum disorder, even if they have cognitive dysfunction.
(2) Clinicians and certified psychologists can use visual aids to deepen patient understanding when autism spectrum disorder is combined with panic symptoms or agoraphobia in patients. As a specific example, blended CBT is practical.
(3) Blended CBT uses visual aids, allowing the therapist to focus more on the patient’s statements and intentions. Therefore, blended CBT is a promising treatment for those who are not good at expressing cognitive processes verbally.
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