Case Report: ASI intervention on a child with autism in Saudi Arabia

Participant

Several exclusion criteria were applied to identify suitable participants. From the families with ASD children that came to OT consultations, we first selected a ten of patients that would be suitable for ASI intervention with sensory issues. Secondly, children with other medical conditions were excluded. Thirdly, children that were not enrolled in mainstream preschool were excluded. We contacted all the remaining families, explained to them the scope and purpose of the research, and invited them to take part in the study. Only one family agreed to participate and committed to the lengthy intervention. The other six families we contacted did not agree to be recorded or were not willing to commit to such a long intervention.

L, a Saudi girl aged 4 years and 7 months, was diagnosed with level-2 ASD at 2 years and 3 months by a multidisciplinary team based on DSM-5 ASD criteria. Her parents provided voluntary consent to participate in the study at Riyadh’s Centre for Autism Research (CFAR) in October 2018. The study was presented to CFAR at King Faisal Hospital and ethical approval was granted for it to proceed.

Her mother provided a detailed history, revealing that the girl was born after a full-term pregnancy with no birth complications. L’s medical reports showed no sign of early infancy complications either and her overall health was good. There was no other relevant family medical or social history.

A semi-structured interview, using occupational profiles, was conducted with L’s mother to identify how L interacts with her environment (AOTA, 2017) and to identify her participation strengths and challenges (Schaaf & Mailloux, 2015). These profiles were translated into Arabic so the mother could understand them.

L had participated in a 2-month home-based speech intervention, completing 3-6 sessions weekly. While this appeared to improve her communication skills, L’s interactions with her peers were limited, and she exhibited frequent tantrums during transitions or changes to routine. Despite her poor communication skills, her cognitive development was typical. In the semi-structured interview, her mother reported that L can follow simplified instructions, especially when given visual cues. She described L as a fast learner, who loves to sing and play with building blocks. The mother was concerned since L found it difficult to make choices and was fussy, always saying no. She was unable to put on socks and shoes and seemed unsure of how to play with other children. She also had difficulties with drawing and with linking it to writing.

L attended school full-time in Riyadh, where she participated in small groups of students with special educational needs. Her teachers had not undertaken ASD-specific training, and she was not receiving ASD-specific interventions. The teacher reported that she used to bump into peers excessively on the playground, avoided activities involving motor skills, and presented difficulties in holding or using crayons or other objects. L’s teacher reported that L had a limited attention span and struggled to accept rules. All these challenges were reported to negatively impacted her social relationships. participation in the classroom, and learning development.

Assessment

To assess impairment related to SI and praxis, we used the Sensory Integration and Praxis Tests (SIPTs) and the Arabic Sensory Processing Measure-Preschool (SPM-P). The SIPTs comprise 17 individual sub-tests for children aged 4-8 years, measuring neurological ability to integrate the sensory inputs required for coordination, motor planning, visual-spatial actions, and perception (Schaaf & Lane, 2015). The SIPTs discriminate between normal and dysfunctional children in the United States at a statistically significant level (Schaaf & Mailloux, 2015). Considering that an acceptable reliability score for research purposes is .70, completing 13 out of 17 test items means that the reliability threshold could be reached around that figure. Each SIPT results in a standard score that reflects the child’s performance compared to age-matched norms; the average score for a group of given age is 0 (Schaaf & Mailloux, 2015). Scores below -1 standard deviation are considered evidence of dysfunction.

The Arabic SPM-P consists of home and classroom forms, which are suitable for children aged 2-5 years (Alkhalifah et al., 2020). The home form has 75 items, and the classroom form has 62. The home form has excellent internal consistency when used with ASD (α = .93) and typically developing (α = .95) children (Alkhalifah et al., 2020). The Arabic classroom form has not yet been assessed, though the English version has excellent internal consistency when used with clinical (α = .93) and typical (α = .94) samples (Parham & Ecker, 2007). Both forms use Likert scales to measure sensory behavior frequency in sensory processing, praxis ability, and social participation (Parham & Ecker, 2007). Both were used in this study because assessing children with ASD in different environments gives a comprehensive understanding of SI function (Parham & Ecker, 2007).

Additionally, we used the Peabody Developmental Motor Scale-2 (PDMS-2), a task-observation test designed to evaluate fine and gross motor skills in children aged 0–5 years (Folio & Fewell, 2000). We selected the PDMS-2 because L’s challenges were associated with impaired fine and gross motor skills, which are common in ASD (Provost et al., 2007). Using a motor skills measure that assesses visual-motor integrations was important as visuomotor connections seem to be aberrant in ASD (Sharer et al., 2015).

Unstructured clinical observations had been gathered during the assessment period. L's overall performance revealed that she had poor equilibrium reaction. She was using her whole body rather than stabilising her body when playing with a ball. She could not grade the force or time to release of the ball. L demonstrated very poor grasp of objects and it was difficult for her to manipulate them around the playground. Additionally, L was unable to maintain a flexion position for more than 10 seconds, while prone extension was achieved for 3 seconds. She showed difficulty with sequential finger touching and ramped arm movements.

Her emotional stability was dependent on the type of tasks she was performing. For example, during the SIPT and PDMS-2 assessments, she refused or cried when confronted with unfamiliar tasks. Her anxiety generated impulsive behavior and affected her overall attitude towards changes or encounters with unfamiliar objects. Whenever she felt overwhelmed or struggled to overcome an obstacle, she started crying. When the therapist urged her to jump, she was unable to and immediately showed signs of utter frustration. Any changes to rules were perceived as frustrating and she threw tantrums during the assessment whenever she felt uncomfortable with something.

Her reactions to new toys and spontaneous behavior were also poor. The playground overwhelmed her instead of provoking joy and excitement. For instance, when she was presented with a space hopper, which was unfamiliar to her, she did not know what to do, nor did she try to explore it. She had limited coping strategies when things felt out of control, or the activities were perceived as too hard. She avoided or ran away from tasks that seemed unfamiliar, such as swinging back and forth. She would say no to any new attempt and it took some time for the therapist to change her mood and bring her back. She also had difficulties in imitation or copying a new behavior and following simple directions. The therapist’s hypothesis was underlying praxis associated with poor imagination, ideation, imitation.

Assessment findings

Results from pre-test SIPT showed that L attempted 13 of the 17 SIPTs, completing 12 (see Figure 1). Scores indicated difficulty with tactile and kinaesthetic processing, especially manual form perception, finger identification, and graphesthesia. L struggled with motor planning as measured by postural praxis, oral praxis, sequencing praxis, design copy, and bilateral motor coordination. Similarly, she scored low for space visualization. L was unable to participate in the localization of tactile stimuli, kinesthesia, and constructional praxis tests, and was unable to fully participate in motor accuracy and post-rotary nystagmus tests. L performed very poorly on tests of both fine and gross motor skills on the PDMS-2, and her total motor quotient score was very poor (Table 1).

Figure 1.

L’s Sensory Integration and Praxis Tests scores.

The findings from the Arabic SPM-P classroom and home forms are illustrated in Table 2 and Table 3, respectively. The environmental difference score demonstrated consistency across environments. Both measures indicated that L struggled with balance and motion, social participation, hearing, and touch. The home test suggested definite dysfunction for almost all scales, including social participation, hearing, body awareness, balance and motion, planning and ideation. Except for vision which was typical, the remaining two scales, touch and total sensory system, also ranked high, indicating that L had difficulties in those areas as well. In comparison, the school form suggested less dysfunction, with only the hearing scale ranked as definitive dysfunction, while the rest fell between typical and problematic. The lack of significant correlation between the home and classroom results might originate from the divergence between adult expectations and daily routines at home as opposed to school (Parham et al., 2007). Her mother’s expectations were definitely higher for how she acted at home, such as her clumsy movement, her unwillingness to wear or put on shoes, her utilise and grade appropriate force when she holds writing and other tools, and her tendency to bump into others and the results supported that since they were almost entirely in the definitive dysfunction ranking. On the other hand, the lack of correlation could relate to the Saudi culture, as parents tend to overprotect their children. This attitude could limit ASD children’s opportunities to be stimulated physically and, thus, to process proprioceptive and vestibular information (Al-Heizan et al., 2015).

Hypotheses and individual goals

In line with the theory of ASI and the practice guidelines for OT, the goals were based on the assessment findings and concentrated on the underlying conditions that impacted L’s development. Overall, the findings suggested L’s participation challenges were due to impairments related to SI and praxis. Specifically, it was hypothesized that L had somatodyspraxia (Schaaf & Mailloux, 2015).

The first author worked with L’s mother to identify specific and measurable goals reflecting L’s functional challenges (Schaaf & Mailloux, 2015). L’s mother knew about her difficulties in school and could contribute to setting both school- and home-based goals. As recommended by Ruble et al. (2012), goals were quality checked by the second author.

The goals were established using the Goal Attainment Scale (GAS; King’s College London) (Ruble et al., 2012). The GAS is both systematic and sensitive to changes in functioning associated with ASI. It entails describing the individual’s present level of functioning for a given goal and then scaling it for the anticipated level of function over the intervention period. GAS helps assess outcomes that are challenging to measure with traditional instruments (Mailloux et al., 2007), and is a valid and reliable tool (Steenbeek et al., 2007) for individuals with ASD (Ruble et al., 2012). GAS uses a five-point scale, with values ranging from -2 to 2, scaled with equally spaced probability intervals (Kiresuk et al., 2014). A score of 0 reflects the anticipated level of function, while -1 and -2 suggest a level of attainment less/much less than anticipated, respectively, while +1 and +2 suggest a level of attainment better/much better than anticipated, respectively. Scores for each goal are used to calculate T-scores, representing the extent to which functioning improved to the degree anticipated.

L’s goals were:

These goals were based on the premise that improving sensory processing would increase participation in everyday activities (Schaaf & Mailloux, 2015).

The authors determined the proximal and distal outcomes to use to track progress.

Distal Outcome Measure: Changes in L’s goals were measured by GAS, enabling the evaluation of progress toward specific, measurable, and time-dependent goals.

Proximal Outcome Measures: The Arabic SPM-P and the PDMS were used as secondary outcome measures. The former was used to measure sensory reactivity, praxis, and social participation, the latter to measure fine and gross motor skills.

Intervention

The intervention was designed with the approval of L’s mother. L would participate in 1-hour ASI sessions twice weekly for 10 weeks. Meanwhile, the author would meet her mother in person and have discussions with her teacher over the phone for 30 minutes every week. Each session would provide sensory-rich experiences to elicit changes in L’s behavior. Accordingly, L’s sessions were designed to provide a challenge level that was “just right” for her sensory systems (Schaaf & Lane, 2015).

For therapy room sessions, the intervention was divided into three phases: early, intermediate, and final sessions (Schaaf & Davies, 2010). Schaaf and Davies (2010) emphasized that ASI entails attention to meaningful activity, requiring adaptive responses and active participation from the child. L had numerous opportunities to play with tactile-rich apparatus, to improve her proprioceptive and tactile perception, and praxis. Moreover, through pulling, pushing, and hanging activities, she was encouraged to stretch and engage her muscles. She participated in various active sensory–motor activities. She used a scooter board to experience proprioceptive and vestibular sensations and increase body awareness. She also engaged in jumping into a ball pit, climbing, rolling, and crawling. There were opportunities to change the apparatus and the rhythm, duration, frequency, and/or intensity of sensory experiences, based on L’s responses. In line with ASI principles, the first author and child cultivated an active, trusting relationship, with the former monitoring activity demands to ensure a just-right level of challenge (Schaaf & Mailloux, 2015).

The use of the ASI Fidelity Measure also ensured the intervention was in line with ASI principles (Parham et al., 2011). This measure defines the structure of the intervention and process elements (Parham et al., 2011; Schaaf & Mailloux, 2015). A score of 80 is a tentative cut-off point for adherence to ASI (Parham et al., 2011). The measure has high interrater reliability for both total fidelity scores (.98) and individual items (.94–.99). The validity of the measure is strong as raters can distinguish ASI sessions from other interventions with 92% accuracy (Parham et al., 2011).

All 20 sessions were videotaped, and the second author, trained in the application of the measure and with 30 years of experience working with children with ASD, assessed four randomly selected tapes. Additionally, the second author provided weekly consultations to the first author and was available throughout to discuss intervention challenges. Only one session produced an unacceptable fidelity score, after which the authors collaborated to ensure acceptable fidelity in all other sessions. The mean fidelity score across the four sessions assessed was 85 (SD = 9).

The early sessions focused on getting L accustomed to the environment, therapist, and equipment on the playground. The author worked with L to help her understand that she could make her own choices. L also learned that she had her own space that she could explore and enjoy. Engaging her in a funny way also provided the therapist more possibilities to gauge her moving abilities. The first sessions gave L many opportunities to develop and use proprioception and vestibular and tactile sensations. The OT notes revealed that L would often choose a familiar object and avoid motor activities. She would always rely on the therapist to initiate activities and presented a low level of adaptive response. Transitions proved to be a real challenge and she needed a lot of positive feedback from the therapist to proceed with new sensory experiences. The therapist learned how to scaffold the activities so that L would get the right challenge and she moved the equipment around to adjust it to L’s needs.

During the intermediate sessions, L became more self-confident and able to express herself, although she needed continuous feedback and encouragement. She was also more confident in her therapist and engaged in the tasks with much more ease than before. The therapist learned to be more specific about the feedback and presented more sensory opportunities, such as tactile and challenge praxis. Under careful guidance and encouragement from the first author, L finally got on the swing, which was acknowledged and celebrated by L’s mother, teacher, and therapist. L was provided with additional tasks that would involve the rotary vestibular eye tracking and crash-bang-or-jump activities.

Over the last sessions, the therapist strategically created specific themes for each session to develop L’s ideation and planning. She also continued to provide L with proprioceptive, vestibular, and tactile sensations for successful integration. L started making choices, initiating, and participating with far fewer prompts. She was able to sustain and develop individual activities for a much longer period. Moreover, she did not feel the urge to withdraw when new activities were introduced to her. She was much more confident in her abilities to engage with the tasks. During the last sessions, L was slightly fatigued with a virus and then a sandstorm hit the region. Her improvements were especially significant in this last phase since the virus and the weather could not stop her from enjoying the sessions and showing obvious progress.

Preliminary evidence for the efficacy of ASI in SA

Given the lack of research describing ASI in SA, this case study examined the feasibility and effectiveness of this intervention. The study suggests ASI is feasible within SA and can lead to improvements in individualized functional goals in ADL and performance on sensory and motor tasks. Not only is the ASI intervention feasible, but the measuring instruments also proved reliable in this context.

However, not all tests measuring SI are suitable in the SA context. The SIPTs had not previously been applied in an Arab nation and they fared questionably in the first exploration of their suitability. In the current study, L could not complete all of the SIPTs; therefore, additional measures were utilized, with GAS completed post-intervention. GAS provided more accurate results and was more effective for quantifying L’s progress during the intervention. SPM-P and PDMS-2 scales could also be implemented to measure ASI interventions in SA and the Fidelity scores demonstrated that therapists could use such instruments with confidence in this socio-cultural context.

The therapist’s unstructured notes and clinical observations as well as feedback from L’s mother and teacher were discussed at length to offer more evidence for ASI implementation challenges and opportunities in SA. The detailed presentation and analysis of data offer support for efficient ASI implementation in ASD children from SA in the future. The authors ensured that the experiment was fully video-recorded and the tools available were rigorously assessed.

Our recommendation for future related studies is to utilize the Evaluation of Ayres Sensory Integration (EASI) instrument, released in 2020. Suitably qualified therapists have open access to the EASI, which incorporates normative data collected internationally. The EASI meets the demand for the assessment of ASI constructs with psychometrically validated and internationally applicable measurement tools (Mailloux et al., 2018).

Limitations

The limitations of the study are divided into two categories: (1) research-related challenges, and (2) cultural context. The authors did not have access to data regarding previous ADOS diagnosis. L was diagnosed with ASD level 2, at 2 years and 3 months by a multidisciplinary team, but the data were not available. The study adopted an experimental single case report of one Saudi child; therefore, the extent to which the results can be generalized is limited, even in the context of SA. Future studies in SA could replicate this study using the measuring instruments used here. Other tools may render different results because ASI interventions have not been regulated yet in Saudi Arabia. The lack of resources translated into Arabic plays a very significant role in the limitations of this study, as discussed below. Another limitation is that the evaluation was completed by the therapist delivering the treatment, not independent assessors, as that was not possible.

Neuroscientific evidence suggests OT-ASI interventions elicit increased neurological adaptations if they simultaneously target multiple sensory systems (Lane & Schaaf, 2010). However, acquiring apparatus suitable for stimulating L’s various sensory systems was challenging; few items were available in SA, so they had to be ordered internationally. The ASI fidelity tool requires a suitable room, points of suspension, and equipment; negotiating additional space and financing for this was time-consuming.

However, most limitations were connected to the lack of resources in Arabic and the shortage of teachers and qualified therapists trained in ASD interventions. A few Arabic language resources are available, such as a translation of the SPM-P (Alkhalifah et al., 2020). It is necessary to translate more resources into Arabic to increase understanding of ASD and SI among teachers in SA, to inform their practice and help them identify pupils who might benefit from OT interventions. While OT-ASI in SA appears feasible, its delivery was not straightforward. Since OT-ASI has only recently been introduced to SA, few parents were aware of its potential, making it challenging to find willing participants. This was partially addressed by translating materials that explained what ASI is and why it is valuable, which was a labour-intensive process.

Teachers in SA rarely participate in interventions and have limited knowledge of ASD and sensory issues (Haimour & Obaidat, 2013). Therefore, the first author spent time explaining to L’s teacher how to complete the Arabic SPM-P and was available to answer questions. Additionally, the first author was available to provide L’s mother and teacher with guidance to understand the intervention. However, therapists in SA working with individuals with ASD and their families often lack time to model interventions or address families’ complex medical and behavioral needs (Alnemary et al., 2016).

Some Saudi professionals do not share alternative treatments with families because of the limited hands-on support available for ASD interventions (Alshehri et al., 2019). This shortage of qualified trainers could reduce the quality of services in SA for ASD (Alnemary et al., 2016). The first author undertook training abroad for over one year. To complete this study, the therapist required confidence and experience in navigating the local protocols and ethical consent processes (Schaaf & Nightlinger, 2007).

Key points for occupational therapy

Ethics and consent

The study was presented to the Centre for Autism Research (CFAR) at King Faisal Hospital and ethical approval was granted for it to proceed. L’s mother gave written informed consent for L to be involved in the study and for L’s information to be published in this manuscript. L’s mother also gave permission for L to be recorded during her sessions. All the recorded sessions are stored in the CFAR’s system, in a separate, secure (locked) folder to ensure data protection. Further, in accordance with the research data management policy, all recorded sessions have been secured in password protected files only accessible to the researchers. The videos will be retained for 10 years, before being securely disposed.