Table 4: Treatment setting and intervention characteristics

Study	Brown et al¹³	Byars et al¹⁴	Clawson et al¹⁵	Cornwell et al¹⁶	Greer et al¹⁷	Kindermann et al¹⁸	Hartdorff et al²⁴	Sharp et al²⁵	Silverman et al¹⁹	Trabi et al²⁰	Williams et al²¹
Setting Inpatient Day treatment	x	x	x	x	x	x	x	x	x	x	x
Treatment duration, d Mean (SD) Range	19	11.4	29	3	46.8	17	14.4	5	10.9	21.6	24
Contributing disciplines Gastroenterologist/physician	x	xX	X	X	X	X	X	X	X	X	X
Nursing/nurse practitioner	X	X	X	X	X	X	X	X	X	X	X
Nutrition/dietician	X	X	X	X	X	X	X	X	X	X	X
Occupational therapist	X	X	X	X	X	X	X	X	X	X	X
Psychologist	X	xX	xX	X	X	Xx	X	X	X	X	X
Speech-language pathologist	X	X	X	X	X	X	X	X	X	X	X
Social worker	X	X	X	X	X	X	X	X	X	X	X
Intervention mechanism(s) Behavioral intervention	x	xX	X	X	X	X	X	X	X	X	8 (73)
Nutrition education	X	X	X	X	X	X	X	X	X	X	2 (18)
Oral-motor exercises	X	X	X	X	X	X	X	X	X	X	5 (45)
Tube weaning	xX	xX	X	X	X	X	X	X	X	X	6 (55)
Behavioral elements Contingency contacting/extinction	X	xX	X	X	X	X	X	X	X	X	7 (64)
Differential attention	xX	X	X	X	X	X	X	X	X	X	2 (18)
Negative reinforcement	X	X	X	X	X	X	X	X	X	X	3 (27)
Positive reinforcement	X	xX	X	X	X†	X†	X	X	X	X	9 (82)
Response cost	X	X	X	X	X	X	X	X	X	X	3 (27)
Shaping/fading	x	xX	X	X	X	X	X	X	X	X	5 (45)
Not specified/used	Xt	x	X	X	X†	X‡	X§	X	X	X	2 (18)
Caregiver training	KX	x	X	X	X	X	X	X	X	X	11 (100)

One further study which investigated intensive multi-disciplinary behavioural treatment for feeding disorders has been published since the systematic review by Sharp et al (2017) [28] (Table 5).

Table 5: Additional published intensive multi-disciplinary treatments for feeding disorders

Author (year) and country	Study aim	Methodology and sample	Symptoms/pres entation	Treatment	Outcome
Seiverling etal. [29] 2019 USA	Examine the effects of intensive interdisciplinary behavioural treatment on 11 feeding outcomes	Retrospective chart review	52 children (ASD = 16, other special needs = 19, NAD = 17)	All children exhibited problem behaviours during mealtimes which prevented advancement in diet variety and/or consumption. All cleared of feeding safety concerns	-Attended day treatment facility between 8.15-3.00 Mon-Fri - positive reinforcement for acceptance of target foods -stimulus fading to increase bite sizes -escape extinction (non-removal of spoon) contingent upon inappropriate mealtime behaviour - dietitian, paediatric nurse practitioner, and gastroenterologist provided nutritional and medical monitoring

Discussion points

There are positive outcomes associated with day treatment and inpatient programs which utilise a multi-disciplinary approach to severe paediatric feeding problems. All studies reported improvements in consumption following interventions.

The below considerations should be taken into account when utilising this systematic review as evidence for the treatment of ARFID.

9. 9/11 included studies were published before the introduction of ARFID as a diagnosis in the DSM-5

• Dependence on enteral feeding or oral nutrition was used as a substitute for an ARFID diagnosis.

-This means results cannot be generalised to the broader ARFID population as we

cannot be certain how many included participants will clinically have an ARFID diagnosis. 10) Majority of included studies were of low quality (non-randomised) 11) 82% tube dependence – more severe form of feeding disorder 12) Considerable heterogeneity

• Outcome measures highly variable. Only tube weaning could be included in meta- analysis -Variable primary feeding and medical concerns (25% with ASD/developmental delay/neurologic)
• Majority of settings were inpatient (8/11)

13. Variable treatment duration Mean = 22.3 days (SD 13.7)
14. Every study included a gastroenterologist/physician, nutritionist/dietician and psychologist
15. Behavioural intervention was most commonly used (73%), however, only two studies used the intervention in isolation.
16. Of those studies that utilised behavioural intervention, 82% used positive reinforcement, 64% contingency contacting/extinction and 45% used fading/shaping. The authors note that “available evidence suggests intensive multidisciplinary treatment likely holds benefits for children with severe feeding difficulties, particularly in cases involving complex medical histories that cannot be effectively managed in an outpatient setting.” Current literature involves notable differences in the sequence, timing, and volume of tube feed reduction. Greater specificity regarding the target(s) of intervention and discharge criteria is recommended. More consistent reporting of follow-up data also is needed to assess the durability of treatment over time. Improved measurement also should entail better characterisation of patients at baseline, including clarity regarding medical and/or behavioural barriers to

Individual Behavioural and Sensory Interventions for Children with Feeding Difficulties

Despite the high prevalence of feeding difficulties in children with ASD, and the implications for short- and long-term health, research regarding intervention for feeding difficulties in this group is scant. It has been shown that clinicians most commonly use therapy approaches based on either operant conditioning (behavioural intervention) or systematic desensitization (sensory intervention) in their treatment for children with ASD and feeding difficulties [30].

Across therapy interventions, those based on operant conditioning currently have the strongest evidence base. However, the majority of existing behavioural research depicting effective specific feeding treatment protocols consist of single case studies or small sample sizes. Operant conditioning interventions use an externally driven ‘top-down’ approach to prompt the child to perform a desired behaviour, often in conjunction with chaining and/or shaping, and then provide a response contingent on that behaviour. Systematic desensitization is an internally driven ‘bottom-up’ approach that involves exposure to a feared stimulus (i.e., food) in the presence of relaxation or play activities. Systematic desensitization is also commonly used in the treatment of feeding difficulties but seldom reported in the literature. Table 6 summarises recent systematic reviews that investigate the efficacy of behavioural and sensory interventions for feeding disorders (primarily in those with ASD). Additionally, several recently published RCT’s relating to the comparison of operant conditioning and systematic desensitisation are also presented.

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Table 6: Summary of literature investigating behavioural and sensory interventions for feeding disorders

Author (year) and country	Study aim	Methodology and sample	Symptoms/ presentation	Treatment	Outcome
Behavioural techniques
Silbaugh et al. 2017 [31]	Evaluate the certainty of the evidence to guide the evidence-based practice of ABA in the treatment of packing	Systematic review of single-subject designs	7 included studies (6 clinical settings and 1 school)	Reflux, failure to thrive, autism, development delay, gastronomy tube	5/7 studies didn’t report patient symptoms. One child packed new or non-preferred foods and one held foods until they dissolved

| Ledford et al. 2018 [32] | (a) What types of interventions have researchers evaluated for individuals with ASD related to mealtime behaviours, and what types of dependent variables have they addressed? Who implemented study procedures, and in what settings were the studies | Systematic review All study type included if there was a comparison condition included | All ASD participants Sixty-five articles or manuscripts with 202 designs | Highly selective eating (i.e., eating fewer than 15 foods; 46%) Problematic mealtime behaviours such as aggression or disruption (38%) Unspecified selectivity (29%) | Contingent rewards (n = 145) Non-removal of spoon (n = 68) Stimulus shaping or fading (n = 63) Re-presentation (n = 62) Response prompting (n = 60) Non-contingent rewards (n = 38) Response shaping (n = 41) Simultaneous presentation (n = 23) Scheduling or restricting food or liquid (n = 17) Behavioural momentum (n = 13) Visual supports (n = 9) Provision of negative consequences (n = 10) | Average of 2.87 components per study - Clinics (outpatient and inpatient) = 88; Homes = 71; Schools = 24; Residential settings = 9; Unspecified = 9

• Total success rate was 75% for studies addressing acceptance, 45% for problematic mealtime behaviour, and 54% for rumination or vomiting.

• Interventions lasted between 2 and 220 sessions (mean = 31)

• 50 studies included a maintenance measure, only 4 (8%) reported that outcomes were not maintained. |

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| Conducted? (c) What were the outcomes, and are they different across independent and dependent variable types, settings, or implementers? | -Rumination or vomiting (18%). | Choice (n = 8) Sensory-based Antecedents (n = 5). | More research is needed to determine for whom and under what conditions feeding interventions are effective, especially for problematic mealtime behaviours. Little information is available regarding the generalization and maintenance of treatment outcomes. | Silbaugh et al. 2016 [33] | (a) Summarise study and participant characteristics of behaviour analytic treatments for food selectivity in children with ASD; (b) evaluate methodological rigor and evidence quality using current standards for evidence-based practice in special education | Systematic review Inclusion criteria: at least 1 participant with ASD, Asperger’s disorder, pervasive developmental disorder. (b) Evaluated a behavioural intervention of food sensitivity; and (c) used a single-subject design including graphed data to allow for visual analysis of treatment effects and outcomes. | -Disordered Feeding Mealtime challenging behaviour | 27 studies (96%) evaluated a treatment consisting of two or more components. One study (4%) evaluated a treatment component (simultaneous presentation) in isolation.

• Differential reinforcements of target feeding behaviour with high preferred food (n = 14, 45%)
• Escape extinction (EE) including non-removal of the spoon (n = 12, 39%)
• Contingent praise (n = 27, 87%)
• Rules (n = 10; 32%)
• Simultaneous presentation (n = 7; 23%), Stimulus fading (n = 7; 23%) Demand fading (n = 7; 23%)
• Differential reinforcement of feeding responses with non-food reinforcers (n = 9; 29%).

Most studies (86%) combined two or more treatment components, including praise, making it difficult to conclude with certainty in many cases precisely which treatment components were responsible for changes in target behaviours. Standards to determine evidence-based practice found that behaviour analytic treatments of food selectivity for children with ASD were classified as having insufficient evidence.

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Marshall etal. 2014 [34]	To assist clinicians in decision-making regarding early intervention for children with ASD and feeding difficulties, and to direct further research.	Systematic Review -experimental design was used to investigate treatment outcomes (control group, within group designs, or single-case based)	Children with ASD aged 0-6 years	Unclear. Inclusion criteria states ‘difficulties relating to eating’ ‘food selectivity;	Intervention was predominantly provided in an intensive format (multiple times daily) (n =10, 43%), parents were the therapy agents in at least one treatment stage in nearly half of the studies (n = 11, 48%), and some component of treatment was completed in the child’s home in 61% of the studies (n = 14).	Intervention feature -Antecedent -Response -Consequence -Reinforcement -Punishment -Non-removal of spoon -Thinning reinforcement -Non-contingent reinforcement -Escape as a negative punishment

Comparison of behavioural and sensory techniques

| Chawner etal. 2019 [35] | Identify interventions used with developmentally disordered populations and to assess their effectiveness in promoting healthy eating behaviours including increasing dietary variety | Systematic review 30 case studies, 3 pre-post intervention design, 1 cross-sectional, 1 retrospective chart review Excluded all eating disorders including ARFID | Symptoms/prese ntations of included participants not reported. | Operant conditioning — escape extinction, non-removal of spoon, physical guidance, differential reinforcement or alternative behaviour, non-contingent reinforcement, lag schedules Based on exposure — systematic desensitisation, stimulus/texture and fading, simultaneous presentation, modelling, high probability sequences, choice of foods, access to preferred food Familial and environmental methods — psychoeducation, parental training, | 34/36 reported positive or effective results Techniques from all groups have been reported to be effective (although environmental interventions were only effective when combined with family interventions) for increasing healthy eating of an individual, Case-by-case basis, by increasing the number of new foods eaten, the percentage of bites accepted during a meal and the amount (weight) of new foods that have been consumed. Authors state “Although escape extinction techniques have been consistently reported as most effective, exposure and reinforcement |

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| Reinoso et al. (2018) [36] | What is the evidence of the effectiveness of Sequential Oral Sensory (SOS), Sensory Integration (SI), and (Differential Reinforcement of Alternative Behaviour) DRA interventions for food selectivity and sensitivity in children with ASD? | Systematic review (cohort studies to case series) | Unclear – can only assume all included studies only investigated ASD | Ages ranged from 3 months to 14 years | Symptoms/prese ntations of included participants not reported | Outcomes measured SOS: progression in feeding developmental milestones, increased repertoire of foods, mealtime behaviour and positive sensory responses, self-feeding, food rejection SI: mealtime behaviour, increased repertoire of foods DRA: self-feeding, mealtime behaviours, intake of non-preferred foods, food refusal, destructive behaviour | techniques should be tried before escape extinction and physical guidance strategies due to ethical reasons and to avoid the possibility of adverse side effects -No follow up to determine long term effectiveness - Overall, the evidence was not sufficiently robust to determine the effectiveness of these strategies on a population level. | OFFICIAL

19 clinical, 9 home setting, 5 school Majority ASD, ID, pervasive development delay, down syndrome, ADHD mealtime plans, positive behaviour support, environmental interventions SOS: Several studies have demonstrated promising results. One included study reported no statistically significant improvements, however, it was a crossover design that may have confounded results due to SOS’s impact being exponentially greater with longer duration of treatment. SI: Results were mixed and inconclusive. Possibly best as an adjunct intervention. DRA: far more research published on DRA as compared to SOS and SI. Research confirms the short-term benefits of this approach, with limited long-term validity. DRA is supported for food selectivity. DRA has the most consistent findings in support of its use for food selectivity. SOS is highly recommended because it addresses sensory-based and behaviour-based aversions; whereas SI addresses sensory-based and DRA addresses behaviour-based. Further research is required in the field of SOS to improve its evidence base. OFFICIAL Page |32

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Marshall etal. (2014) [37]

Determine whether intervention across 2 therapy arms (Operant conditioning vs Systematic Desensitisation) had an impact on increasing dietary variety and quality and decreasing the frequency of undesirable mealtime behaviours in children with feeding difficulties.

RCT

Feeding difficulties in children with an ASD diagnosis and those considered non-medically complex (never received treatment for a medical condition).

78 eligible participants.

Food selectivity by type (<10 foods across each food group: fruits/vegetables, proteins, carbohydrates). Food selectivity by texture (e.g., only consuming purees).

Mealtimes averaging >30 minutes, and/or clinically significant difficult mealtime behaviours that were having an impact on parental stress.

• 10 sessions consisting of 30-60 minutes (either 10 in one week or weekly over 10 weeks)

• Systematic desensitisation (SysD): “Bottom up” modelling and play based therapy

• Operant conditioning (OC): Top-down prompt and reward therapy

Number of foods offered, short and long term goals, parent involvement and generalisability were the same across both treatment interventions.

No different in efficacy of interventions. Total number of foods consumed by OC group was clinically greater but not statistically significant. No differences observed between etiological groups or intensity (weekly vs intensive intervention).

3 month follow up showed continued improvements however treatment groups were not separated.

Official Document

| Marshall et al. (2018) [38] | To examine the outcomes of therapy intervention for medically complex (MC) versus Non MC participants overall, OC versus SysD intervention, and intensive versus weekly therapy intensity dose; and to examine the parent satisfaction following access to a feeding therapy program. | RCT | As above | As above | Statistically and clinically significant favourable changes to outcome measures for children receiving either intervention were observed. When delivered to a protocol, with consideration of the sensory motor skills of the child, and with the inclusion of parent training, OC or SysD approaches can be successful forms of treatment for feeding difficulties. Parents of children in the MC arm were significantly more likely to elect for intensive intervention than weekly (P 0.02).

| Galpin et al. (2018) [39] | To examine the impact of a sensory based intervention to address food selectivity in autistic pupils that could be delivered in a school setting by teaching staff. | Repeated-measures within-subject design | No specific eating or feeding difficulties noted All children had the requisite oral-motor skills to eat table food and had no physical complications, such as dysphagia. | “Sensory Snack Time”: systematic desensitization through the sequential presentation of foods A range of 52 different foods, three liquids and five sauces categorized based upon their texture and food group was made available to pupils during the 12 weeks of Sensory Snack Time sessions, with 4–8 foods available during each session | There were significant improvements in food selectivity score (P <0.001), food refusal (P 0.005) and number of foods tried (P 0.003) post-intervention. Results indicated that pupils ate a wider variety of foods and displayed significantly reduced food selectivity, distressed mealtime behaviours, and food refusal following the 12-week intervention Further research is necessary to qualify the precise impact the intervention had and to examine the potential for the intervention to be generalized to main meals and different settings, such as pupils’ homes.

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The below article was not included in this synthesis as there was significant overlap of included studies with more recent reviews conducted by Silbaugh et al (2016), (2017), Marshall et al (2014), Ledford et al (2018) and Chawner et al (2019)

Sharp WG, Jaquess DL, Morton JF, Herzinger CV. Pediatric feeding disorders: A quantitative synthesis of treatment outcomes. Clinical child and family psychology review. 2010 Dec 1;13(4):348-65.

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Discussion points

All systematic reviews investigating behavioural interventions concluded that the level of evidence was low or ‘suggestive’. This is due to small sample sizes, case study designs and inconsistent outcome measures.

Silbaugh et al. 2016 [33] concluded that “standards to determine evidence based practice found that behaviour analytic treatments of food selectivity for children with ASD were classified as having insufficient evidence.”

There was little information available regarding the generalisation and maintenance/follow up of treatment outcomes.

The intensity of intervention provided (e.g. multiple times per day) appeared to have no impact. There was a trends towards more successful intervention outcomes where parents undertaking intervention in their home environments

Further research using standardised protocols and randomised study designs are required to enable the evaluation of the certainty of the evidence. This will enable researchers and clinicians to determine for whom and under what conditions feeding interventions are effective, especially for problematic mealtime behaviours.

Systematic reviews comparing sensory and behaviour interventions found that techniques from all groups have been reported to be effective (although environmental interventions were only effective when combined with family interventions) for increasing healthy eating of an individual (increasing the number of new foods eaten, the percentage of bites accepted during a meal and the amount (weight) of new foods).

Chawner et al. (2019) [35] concluded that “Although escape extinction techniques have been consistently reported as most effective, exposure and reinforcement techniques should be tried before escape extinction and physical guidance strategies due to ethical reasons and to avoid the possibility of adverse side effects

This was reiterated by Reinoso et al. (2018) [36] who stated that Sensory interventions are highly recommended because they address sensory-based and behaviour-based aversions

Ethical Concerns with Applied Behavioural Analysis for Autism Spectrum Disorder

Autism advocates have raised concerns about the use of ABA for many years, citing bioethical concerns about the rights of autistic children and their parents which are regularly infringed upon [40]. The question of the ethicality of ABA is of critical societal importance especially as it is often referred to as the “gold standard” of care for ASD [40].

ABA is a form of behaviour modification that relies heavily on external reinforcement, both positive and negative (operant conditioning) [41]. ABA is intended to modify or diminish behaviours, as well as increase language, communication, social skills, attention, etc., in children with ASD [41]. While operant conditioning may be effective for teaching specific tasks in certain situations, in nearly all other circumstances it is not typically used to the extreme extent that it has been applied with for the treatment of many children with ASD [41].

ABA therapy has been viewed as the gold standard for treating children with ASD because various meta-analyses have found it to be very efficacious [41]. However, research indicates

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efficacy only with those who have a measurable Intelligence Quotient (IQ), typically at 70 or above [41]. Much of the research has excluded children who are non-verbal, particularly those who are “lower functioning’ and ‘untestable’ [41]. Unsurprisingly, this is the population that tends to receive continuous ABA therapy over a longer period of time due to their reduced ability to meet the criteria needed to master a task [41].

ABA has been described as “an encroachment on the autonomy of children forced to receive it. Even granting that parents have the authority to decide in favour of ABA, doing so runs two very serious risks. First, it can alter children’s identities by preventing them from forming and pursuing their own passions. Second— and more problematically—it can teach them that there is something wrong with who they are, teaching them how to blend in rather than exercise their own unique capacities.” [40]

A lifetime or punishment and reward without an understanding of the task that is being asked, can create individuals who are compliant and conditioned to obey others, independent of a task. Research [42] has indicated numerous problems with the underlying theory of ABA, specifically unintended consequences such as; (1) compliance, (2) low intrinsic motivation, (3) prompt dependency (4) low self-confidence, or self-esteem to successfully engage in any task and (5) lack of independent functioning—the latter of which is the presumed goal of ABA therapy in the first place.

Sandoval-Norton et al. 2019 [41] stated that “being punished for certain movements, and being forced to engage in eye contact despite the physiological pain and discomfort of doing so, is psychological and physical abuse. A lifetime of being forced to sit still with no regard for actual cognitive abilities can create further emotional and psychological harm.”

ABA neglects current research and data on children with Autism. Some of this research would include the autistic brain, access to MRI studies, or comorbid psychopathology associated with autism such as;

1. Anxiety
2. Attention-Deficit/Hyperactivity Disorder (ADHD)
3. Obsessive Compulsive Disorder OFFICIAL

This knowledge is neglected by ABA therapists who implement behaviourist principles that are inappropriate to treat these comorbid disorders.

Sandoval-Norton et al. 2019 [41] notes that ABA therapists “…are essentially practicing out of their scope and without a license, with the hopes that ABA will somehow address both maladaptive behaviours and comorbid disorders….ABA is never prescribed to rid someone of anxiety but it can in fact create more anxiety along with a myriad of other issues previously discussed.”

It should also be noted that most ABA practitioners are unregulated and unlicensed paraprofessionals and care givers, with neither the discipline of psychology nor related fields nor government establishing any real oversight or review procedures [43].

• ABA is not regulated in Australia.
• Griffith University and Monash University are the only two institutions that offer a BCBA qualification.

A recent online survey by Kupferstein (2018) [44] investigated what percentage of individuals exposed to ABA met criteria for PTSD based on responses from both caregivers and adults with ASD. This survey was further analysed using qualitative techniques [45]. The findings of this survey are summarised in Table 7. This is the only study to date which has investigated this interaction.

Table 7: Research into Post Traumatic Stress Disorder caused by ABA

Author (year) and country	Study aim	Methodology and sample	Data collection	Results/Outcome
Kupferstein (2018) [44]	(a) To investigate whether autistic individuals exposed to ABA intervention would meet the PTSD criteria. (b) Test for correlations between the severity of PTSS and the length of time exposed to the intervention.	Online survey Professional diagnosis of ASD Age over 18 (autistic adults and caregivers) Recruited via social media, support	-Basic demographics-Type of ASD intervention received as a child-Length of intervention-26 questions relating to PTSD using Likert Scale	-46% of ABA exposed respondents met the threshold for PTSD-Within that group, 47% recorded extreme levels of severity-Adults and children without ABA exposure had a 72% chance of reporting no PTSS

Kupferstein (2019) [45]

To explore why autistic people and their caregivers choose interventions other than ABA, and how their decision impacts them over their lifespan.

Online survey

• Thematic analysis of comments section of previous survey by Kupferstein.
• Secondary analysis of initial survey responses

As above

Communication-based intervention group experienced less PTSS (30%) than their ABA-exposed peers (42%). Only 17% of those with no treatment met the criteria for PTSD (p <0.001)

Qualitative analysis

• Those exposed to ABA more likely to use psychologically abnormal language that were indicative of desensitisation.
• Those who opted out of the survey did so around the questions pertaining to self-harm and injurious behaviour.
• Those who abandoned the survey were less likely to have been exposed to ABA.

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