Research Request — Avoidant Restrictive Food Intake Disorder (ARFID)

Brief

Review the research behind;

• ARFID (diagnosis and prevalence in the population and those with ASD)
• Intensive feeding programs – including any systematic reviews of the different intensive feeding programs available
• Best practice treatments for ARFID
• Negative effect of Applied Behavioural Analysis interventions on those with ASD

Date: 26/05/2020

Requester: redacted Researcher: redacted Cleared by: redacted

Please note:

The research and literature reviews collated by our TAT Research Team are not to be shared external to the Branch. These are for internal TAT use only and are intended to assist our advisors with their reasonable and necessary decision making. Delegates have access to a wide variety of comprehensive guidance material. If Delegates require further information on access or planning matters they are to call the TAPS line for advice.

The contents of this document are OFFICIAL

Table of Contents

• Summary of Research Request …3
• Treatments for ARFID …3
• Intensive Multi-disciplinary intervention for paediatric feeding disorders…3
• Individual Behavioural and Sensory Interventions for Children with Feeding Difficulties…4
• Ethical Concerns with Applied Behavioural Analysis for Autism Spectrum Disorder…5
• Overview of Avoidant Restrictive Food Intake Disorder …6
  • What is Avoidant Restrictive Food Intake Disorder?…6
  • How should ARFID be assessed?…7
  • Prevalence…8
  • Clinical Characteristics…9
• Treatment Interventions for Avoidant Restrictive Food Intake Disorder…10
  • Pharmacological treatment …10
  • Psychological treatment …11
  • Multi-modal approach …12
  • Discussion points …21
  • Limitations …21
• Intensive Multidisciplinary Intervention for Paediatric Feeding Disorders …22
  • Summary of results…22
    • Treatment settings and approach to intervention …22
    • Treatment outcomes …23
  • Discussion points …26
• Individual Behavioural and Sensory Interventions for Children with Feeding Difficulties
  • Discussion points …36
• Ethical Concerns with Applied Behavioural Analysis for Autism Spectrum Disorder …37
• Reference List …41

Summary of Research Request

Treatments for ARFID

There are no well-established treatments for ARFID, with a limited number of randomized clinical trials among patients with ARFID. Studies investigating the treatment of ARFID were:

• Low quality (mainly case studies) with small sample sizes. Further research will need to focus on larger RCT’s which use consistent population characteristics and outcome measures.

This literature review evidences several promising treatment avenues which warrant further study:

• FBT, CBT and adjunctive pharmacological intervention appear to be the methods with the best evidence.
• A multi-modal approach is also endorsed, particularly for those with severe feeding difficulties.

Overall consensus is that this must be individualised, depending on the main concern and degree of severity.

Evidence to guide treatment for this heterogeneous population is needed. An expert consortium [Eddy] agreed that not all individuals with ARFID would require a multidisciplinary treatment team. The expert consensus was that all patients generally require a minimum of a primary care practitioner and/or paediatrician to monitor physical health. The need for multidisciplinary involvement increases at younger ages and with higher levels of severity and medical complexity. Patients who are older or less severe may manage treatment with a single practitioner whose expertise is most relevant to the case.

Intensive Multi-disciplinary intervention for paediatric feeding disorders

There are positive outcomes associated with day treatment and inpatient programs which utilise a multi-disciplinary approach to severe paediatric feeding problems.

The below considerations should be taken into account when utilising the systematic review by Sharp et al [28] as evidence for the treatment of ARFID:

1. 82% of 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.

2. Majority of included studies were of non-randomised and of low quality
3. 82% of studies included participants who are tube dependent (severe form of feeding disorder)
4. Considerable heterogeneity between studies

• Outcome measures highly variable

-Variable primary feeding and medical concerns (25% with ASD/developmental delay/neurologic)

• Majority of settings were inpatient (8/11)

5. No consistency in treatment duration Mean =22.3 days (SD 13.7), range 5-46.8 days
6. Every study included a gastroenterologist/physician, nutritionist/dietician and psychologist
7. Behavioural intervention was most commonly used (73%), however, only two studies used the intervention in isolation.
8. Of those studies that utilised behavioural intervention:

• 82% used positive reinforcement
• 64% contingency contacting/extinction
• 45% 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.”

The 11 studies included in the systematic review by Sharp et al [28] prohibit definitive conclusions regarding optimal models of care due to poor patient characterisation, heterogeneity in outcome measures and lack of follow up to determine treatment durability. More systematic evaluation of different treatment approaches and adjuncts to behavioural intervention and/or tube weaning is warranted.

1.3 Individual Behavioural and Sensory Interventions for Children with Feeding Difficulties 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 [13] 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 Systematic reviews comparing sensory and behaviour interventions found that techniques from all groups have been reported to be effective for increasing healthy eating of an individual. Chawner et al. (2019) [35] concluded that “Although escape extinction techniques have been consistently reported as most effective, exposure and reinforcement techniques should be OFFICIAL Page | 4

tried before escape extinction and physical guidance strategies due to ethical reasons and

to avoid the possibility of adverse side effects

Sensory interventions have been recommended because they address sensory-based and behaviour-based aversions (food selectivity and sensitivity); whereas Differential Reinforcement of Alternative Behaviour only addresses behaviour-based. However, further research is required in the field of sensory interventions such as sequential oral sensory (SOS) to improve its evidence base.

Recent high quality RCT’s by Marshall et al. (2015, 2018) [37, 38] have compared operant conditioning to sensory desensitisation and found that:

• No differences in efficacy of both interventions
• No differences observed between etiological groups or intensity (weekly vs intensive intervention)
• 3 month follow up showed continued improvements

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. 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.

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]

Practitioners that often deliver ABA are unregulated and unlicensed paraprofessionals and care givers. Neither of which have experience in the discipline of psychology nor related fields [39].

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

The link between ABA and PTSD has recently been investigated by Kupferstein (2018) [44] using an online survey format. The survey found that 46% of ABA exposed respondents met the threshold for PTSD. Within that group, 47% recorded extreme levels of severity. This is the only study to date which has investigated this causal link, therefore, further research is required to confirm results.

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Overview of Avoidant Restrictive Food Intake Disorder

What is Avoidant Restrictive Food Intake Disorder?

Avoidant/restrictive food intake disorder (ARFID) was introduced in 2013 as a formal diagnostic category in the ‘Feeding and Eating Disorders’ section of the Diagnostic and Statistical Manual, Fifth Edition (DSM-5) and more recently in the 11th Revision of the World Health Organisation’s International Classification for Diseases (ICD-11). ARFID provides a diagnostic label for a heterogeneous group of children, adolescents or adults who engage in avoidant or restrictive eating behaviours without weight or body image concerns [1, 2].

It is defined as a persistent disturbance in feeding or eating behaviour resulting in the individuals energy needs, their nutritional need, or both, failing to be met. In other words the person fails to eat enough in terms of variety, overall amount, or both. If energy needs are not met, normal weight gain in childhood will falter or weight will drop. Insufficient energy intake can also have a negative impact on growth, which can falter. If nutritional needs are not met through a limited diet, the individual will be at risk of developing nutritional deficiencies, with related medical and physical consequences such as reliance on tube feeding and oral nutritional supplements. It is important to be clear that the avoidance and restrictions of food intake characteristic of ARFID does not necessarily result in weight loss or low weight. Some individuals may only accept a very restricted range of foods, but if these foods have a high energy content (potato chips, chocolate biscuits or soft drinks etc.), weight may be normal or high, yet the individual is likely to present with significant nutritional deficiencies. However, some children do present with extremely low weight, and some with nutritionally related stunting. The important point here is to recognise that ARFID is not a low weight disorder per se.

Clinical observations and scientific reports have demonstrated considerable variability in the presentation of ARFID. There is a lack of consensus in the medical community as to whether ARFID is an ‘eating’ or ‘feeding’ disorder. ARFID resembles a feeding disorder in demographic features, comorbidity, source of presentation, and greater acceptance of invasive treatments [3]. Conversely, ARFID resembles anorexia nervosa (AN) in children in

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terms of management and treatment of the illness and shares similar presentations with non-fat phobic AN in some cases [3].

The DSM-5 diagnostic criteria [1] currently lists three examples of features that may be driving disturbances in eating behaviours:

• 1. An apparent lack of interest in eating
• 2. An avoidance based on the sensory characteristics of food
• 3. A concern about the aversive consequences of eating

It is important to note that this list is not mutually exclusive and not intended to be exhaustive, with the diagnostic manuals acknowledging that other causal processes can underpin restrictive eating in ARFID. Instead, they are intended as a first step towards parsing variability in ARFID and understanding its underlying causes.

2.2 How should ARFID be assessed?

A multi-disciplinary group of international experts in feeding disorder and eating disorder clinical practice and research convened as the Radcliffe ARFID workgroup to operationalise ARFID and to guide research [4]. Individuals with ARFID often present to settings other than mental health clinics. The group achieved clear consensus that screening of possible ARFID can be made by any healthcare professional including, but not limited to, a mental health provider, dietitian, paediatrician, family physician, internist, nurse practitioner, endocrinologist, gastroenterologist, speech and language pathologist, or occupational therapist.

It is recommended that the evaluation and diagnosis (medical and nutritional assessment) of ARFID be performed by a medical professional (e.g., primary care physician, paediatrician) [4]. Such evaluation should include a physical assessment to ascertain growth, eating history, and the assessment of acute and potential long-term medical and nutritional complications of avoidant/restrictive eating such as sequelae of low weight (e.g., hypogonadism, bone loss) or obesity, as well as malnutrition (e.g., insufficient vitamin and mineral consumption), which can occur in individuals with ARFID across the weight spectrum. Medical assessment should also explore presence of underlying systemic or

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gastrointestinal disorders which may contribute to the onset or persistence of ARFID, such as celiac disease, peptic or allergic gastrointestinal disease (including eosinophilic esophagitis), Crohn’s disease, and functional gastrointestinal disorders including constipation and irritable bowel syndrome. Nutritional/dietary assessment should determine the adequacy of dietary diversity, and caloric needs to maintain growth and development. Additional opinion and input from specialists may be needed for more complex ARFID presentations [4]. A mental health clinician (e.g., psychologist, psychiatrist, social worker) should complete the diagnostic interviews and assessment of psychosocial impairment and functioning [4].

2.3 Prevalence

A systematic scoping literature review of ARFID identified significant variation in prevalence estimates, with preliminary estimates among clinical eating disorder populations ranging from 1.5% to 64% and <1% to 15.5% in non-clinical cohorts [5].

Although ARFID comprises multiple aetiologies, clinical populations are found to display some demographic similarities. The literature consistently reports that ARFID patients are younger than non-ARFID ED patients, more likely to be male and report a longer duration of illness, on average, compared to AN or bulimia nervosa (BN). A recent study which retrospectively reviewed clinical data from an eating disorder day program found a significantly higher comorbidity of anxiety disorders in patients with ARFID (72%) than the other eating disorder groups (31%) (P< 0.0001). Autism spectrum disorder (P= 0.001), learning disorders (P < 0.0001), and cognitive impairment (p < 0.0001) were also seen more frequently in the patients with ARFID, based on past history reported at initial assessment [6]. It is important to note that much of the current understanding is based on the study of relatively small, clinical samples, particularly those who have presented to an eating disorder programme or sought help from a physician specialising in eating disorders.

Two sequential population based surveys were conducted in South Australia to investigate the prevalence and burden of ARFID of individuals aged 15 years and older [7]. The authors reported a very similar three-month prevalence of ARFID in 2014 and 2015 (0.3% Cl 0.1-0.5

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Clinical Characteristics

Current literature states that ARFID commonly presents alongside various medical and psychiatric comorbidities, including attention deficit hyperactivity disorder (ADHD), ASD and internet gaming disorder [7]. Although associated with a high degree of co-morbid anxiety disorders ARFID patients are found to be less prone to mood disorders than those with other eating disorders [7].

The current literature supports the existence of different ARFID presentations which vary according to the main driver of food avoidance. This has prompted efforts to investigate the validity of the three examples of features included in the DSM diagnostic criteria [1]. Though presentations characterised by one of each of these three features have been observed and reported, individuals often present with multiple characteristics which overlap and co-occur [7].

The systematic scoping literature review conducted by Bourne et al [7] yielded nine studies which compared the medical and psychological profile of patients with ARFID and other restrictive eating disorders. Whilst similar levels of dietary restriction were observed in the cohorts studied, patients with ARFID were found to display clinically-distinct presentations compared to those with other eating disorders, including a history of abdominal pain, a longer length of illness and a distinct absence of any cognitions relating to weight or body image. Several case studies (n=6) also reported that ARFID can develop in the context of various secondary medical or psychiatric illnesses, including food avoidance associated with drug use, dietary restriction due to gastrointestinal discomfort following surgery and two cases of ARFID occurring alongside psychosis [7].

Treatment Interventions for Avoidant Restrictive Food Intake Disorder

To date, only one review (Level ///-2) exists which systematically assesses the ARFID literature relating to current treatment options [5]. The review was conducted in 2019 and identified various RCTs, case and cohort studies that delivered treatment to patients with ARFID. These were separated into three broad groups; (1) pharmacological treatment; (2) psychological treatment and (3) multi-modal treatment. These studies are summarised in Table 1 below.

Pharmacological treatment

Five studies have reported on the pharmacological treatment of ARFID and in particular, the use of medication as an adjunct to therapeutic intervention, which is recognised as an increasingly common treatment approach. Owing to its success in treating anorexia nervosa (AN), Olanzapine was presented as a potential treatment strategy for relieving related symptoms of anxiety and promoting appetite [8]. Several other medications, including Mirtazapine and Buspirone, have surfaced as pharmacological candidates in the treatment of ARFID, both of which were found to relieve anxiety associated with choking and/or vomiting [9, 10]. Gray et al. [11] also reported on the use of Mirtazapine to increase appetite and facilitate weight gain, but in contrast to Tanidir and Herguner [10], the authors noted heightened anxiety associated with an increased dosage. Thus, varying results have been observed.

The only double-blind, placebo-controlled study which reports on the efficacy of using medication to treat chronic food refusal took 15 children with ARFID and randomly assigned them to one of two conditions [12]. While both groups participated in daily intensive behavioural intervention, eight were administered D-cycloserine (DCS) as an adjunct to therapy, and remaining participants given a placebo. The behavioural intervention treatment consisted of manual incorporated escape extinction and reinforcement procedures. Though a substantial improvement in mealtime behaviours was observed in both groups, DCS was found to enhance response to the behavioural intervention.

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Preliminary findings are a promising indicator that DCS is an effective adjunct to behavioural intervention, although larger clinical trials are warranted to fully verify this.

Psychological treatment

Five case studies were found to report on the use of cognitive behavioural therapy (CBT) to treat ARFID. In four studies, the interventions used CBT approaches to formulate and address eating-associated anxiety and fears about food consumption, without the focus on weight and shape concerns used in CBT methods for other eating disorders such as AN [13-16]. A fifth study employed a novel 4-week, exposure-based CBT intervention, developed to target other drivers of food avoidance and/or restriction (i.e., disgust sensitivity, dysfunctional cognitions about feared foods, the aversive consequences of eating) [17]. This method, which has been designed specifically for adolescents with ARFID and integrates inhibitory learning principles has demonstrated preliminary success in treating a number of ARFID presentations.

Cognitive Behavioural Therapy (CBT)

Cognitive Behavioural Therapy (CBT): is a short-term, goal-oriented psychotherapy treatment that takes a hands-on, practical approach to problem-solving. Its goal is to change patterns of thinking or behaviour that are behind people’s difficulties, and so change the way they feel. CBT works by changing people’s attitudes and their behaviour by focusing on the thoughts, images, beliefs and attitudes that are held (a person’s cognitive processes) and how these processes relate to the way a person behaves, as a way of dealing with emotional problems Examples: Learning how to manage stress and anxiety (e.g., learning relaxation techniques such as deep breathing, coping self-talk such as “I’ve done this before, just take deep breaths,” and distraction) identifying situations that are often avoided and gradually approaching feared situations.

Two case series and one feasibility study were found to report on the use of family-based therapy (FBT) to treat ARFID [18-20]. FBT, which is designed to empower caregivers, reduce familial guilt and support recovery at home, is often used in the treatment of eating disorders. Although FBT-ARFID is similar in this respect, and employs the main principles of FBT, it has been adapted to address the needs of patients with different ARFID presentations, targeting those with sensory sensitivities, fear-based concerns and little interest in eating [18]. Though limited by small sample sizes and lack of a long-term follow-up, the evidence suggests that FBT may prove to be a feasible treatment approach. Ina

similar manner, a small number of parent training curricula have been trialled which aim to

coach caregivers in implementing at-home behavioural feeding interventions. Initial findings indicate that both parent teleconsultation and attendance at group education sessions can adequately prepare caregivers to support children who engage in severe selective eating but do not require treatment in a hospital setting [21, 22].

Family-based therapy (FBT) for eating disorders is commonly known as The Maudsley Model

and was originally developed to treat adolescents with Anorexia Nervosa and Bulimia Nervosa. FBT aims to assist the family, namely the parents, to bring about recovery in their child with an eating disorder. The core principles of are:

1. No one is to blame for the development of the eating disorder
2. The eating disorder is externalised or separated from the sufferer and the eating disorder is targeted to reduce blame and criticism
3. The family are viewed as the best resource to bring about recovery
4. Hospitalisation is a short term solution for the problem
5. Each family member is assigned a specific role

3.3 Multi-modal approach

Intervention-focused papers commonly endorse a multi-modal approach, characterised by input from a multidisciplinary team and incorporating a wide range of interventions [23, 24, 20]. The efficacy of such an approach was supported by an RCT investigating the treatment of chronic food refusal in a day treatment programme [25]. The researchers randomly assigned twenty children aged 13-72 months to either a waiting list or a five-day intensive behavioural intervention with treatment input from a multidisciplinary team. Despite a small sample, the intervention group displayed significantly greater improvements (p < .05) on all primary outcomes compared to no treatment, suggesting that a collaborative approach to treatment can safely and effectively address the challenging nature of food refusal.

| Author (year) and country | Study aim Methodology Symptoms/presentation | Treatment Outcome | — | — | — | — | | Brewerton & D’Agostino 2017 [8] USA | To document the clinical progress of ARFID patients treated with low doses of adjunctive olanzapine | Retrospective chart review of 9 patients (8 females and 1 male) (9–19 years) - Mean admission BMI 15.6 ± 1.8 kg/m2 | Participants diagnosed with ARFID using DSM-5 criteria | - Adjunctive low-dose olanzapine (alongside meal behaviour therapy and other treatment modalities offered to ED patients)

• Mean number of days on olanzapine 53.4 ± 22.4 | - Mean change in BMI 3.1 ± 1.34 kg/m2
• Mean change in BMI index for age percentile 11.0 ± 14.7 to 35.9 ± 27.5 Olanzapine promoted weight gain in all patients and relieved symptoms of anxiety, depression and cognitive impairment | | Okereke 2018 [9] USA | To describe the successful treatment of anxiety using Buspirone in an individual with ARFID | Case study 14-year-old female BMI 20.3 kg/m2 (58th percentile) | Complaints of anxiety, abdominal pain and vomiting resulting in food restriction (later diagnosed with ARFID as well as irritable bowel syndrome) | - Individual and family therapy
• Sertraline at 50 mg/day (discontinued when patient experienced agitation and thoughts of suicide) Buspirone 5 mg twice daily increased to 7.5 mg twice daily at 1 month follow up and 10 mg twice daily at 6-month follow-up
• Follow-up 1, 2, 4, 6, and 8- months post-treatment |
• BMI at 8-month follow up was 22.0 kg/m2 (73rd percentile) SSRIs can be used to treat eating-related anxiety but may cause adverse side effects, particularly in children and adolescents Buspirone successfully treated anxiety symptoms associated with eating (patient denied any significant side effects) |

| Tanidir & Herguner 2015 [10] Turkey | To present a case of ARFID successfully treated with mirtazapine | Case study 10-year-old female | Refusal to eat solid food after choking incident at 4 years old | - Initial behavioural approach: 10 mg/day fluoxetine increased over time to 30 mg/day for 2 months with no success. - 15 mg/day mirtazapine for 6 months | - Weight increased to 34 kg (25—50th percentile). - Mirtazapine well tolerated marked and rapid improvement in symptoms relating to choking phobia. - Within 2 weeks, the patient reported less anxiety during mealtimes and experienced an increase in appetite. - No re-emergence of complaints at 6-month follow up.

| Gray 2018 [11] USA | To evaluate the use of mirtazapine in treating patients with ARFID | Retrospective chart review: 6 females, 8 males (7–23 years) who received treatment at a San Diego eating Disorders clinic from 2015 to 2016. Mean BMI at intake 16.8 ± kg/m² | Difficulty eating related to low appetite cues, taste, or texture sensitivity, anxiety of an adverse event (e.g., choking), or significant functional gastrointestinal distress | - Six patients treated with mirtazapine as monotherapy and 8 on additional medications. - Average dose of mirtazapine: 25.5 mg. - Follow-up 6-months post treatment and monthly follow ups thereafter | - Average change in BMI without mirtazapine = 0.10 BMI point per week.

• Average change in BMI with mirtazapine = 0.23 BMI point per week (t13 = −3.11, p < .05).
• Overall, mirtazapine was safe, well tolerated and encouraged greater weight gain than treatment-as-usual programme.

| Sharp 2017 [12] USA | To examine the feasibility and preliminary efficacy of combining D-cycloserine with a behavioural intervention in treating young children with chronic food refusal | Double-blind, placebo controlled study: 16 children (37.5% female) 18 months – 6 years | Active and persistent food refusal which severely restricted the volume of food consumed | - Randomisation to intensive Behavioural intervention + D-cycloserine OR intensive behavioural intervention + placebo over 5 days (15 meals in total). - Follow-up 1-month post treatment | Mealtime behaviours improved significantly in both groups, but D-cycloserine further enhanced response to intervention, rapidly increased food acceptance and reduced disruptive behaviours.

Psychological treatment

| Fischer 2015 [13] USA | To evaluate the effects of an intervention for chronic food selectivity in an adolescent with ARFID | Case study 16-year-old-male | History of extreme food selectivity, associated feeding anxiety and some acute sensory aversion to certain foods | - Intervention incorporating both a clinic (behavioural treatment and CBT) and concurrent in-home component (enforced by the patient’s mother) | - Follow-up 1- and 3-month post treatment | - Greater consumption of foods (both quantity and variety) | - Reduced anxiety and ability to eat out in a social environment | - Daily bowel movements and increased energy (findings maintained post-treatment) | King 2015 [14] USA | To present a case of ARFID successfully treated with CBT | Case study 41-year-old female, BMI 15.5 kg/m² | Patient had Crohn’s disease as a child and developed severe illness anxiety following acute gastroenteritis which caused her to limit food intake | - Inpatient treatment - 8 sessions of CBT including psychoeducation, systematic desensitisation (in vivo exposure) and cognitive restructuring | - Follow-up 8-months post treatment | - At discharge, patient was consuming 1650 calories daily and BMI 16.5 kg/m², and reported reduced anxiety and increased energy | - At 8 months post-discharge, patient BMI was 19.4 kg/m² | Aloi 2015 [15] Italy | To present a case of ARFID successfully treated with CBT and family involvement | Case study 24-year-old male, slightly overweight with BMI 25.5 kg/m² | - Dysfunctional eating behaviours dating back to the age of 2 | - Avoidance based on an unpleasant sensory experience | - Complaints of anxiety relating to shared meals, resulting in social withdrawal | - Psychotherapeutic intervention once a week for one hour over six months

• Phase 1 (session 1–4) psychoeducation
• Phase 2 (session 5–7) family Therapy
• Phase 3 (session 8–18) CBT
• Phase 4 (session 19–20) relapse prevention Follow up 6 months post treatment | - Many new foods introduced to the patient’s diet
• Improved social relationships and willingness to engage in shared meals | Gormez 2018 [16] Turkey | To present a case of ARFID successfully treated with CBT | Case study 27-year-old female BMI 16 kg/m² (lost 6 kg) | Nausea, retching, vomiting and unable to tolerate the sight and smell of food | - 12 40-minute weekly CBT sessions as an inpatient and 8 sessions as an outpatient as | OFFICIAL Page | 15

| Dumont 2019 | Netherlands | To test a new 4-week exposure-based CBT day treatment for adolescents with ARFID | Case series Patients referred to SeysCentra, a Specialised treatment facility for children with feeding disorders (n = 11), 36% female, 10–18 years | Various presentations including anxiety driven (phobia), lack of interest in food, driven by disgust or aversion | - Exposure based CBT treatment designed to address a variety of ARFID presentations (i.e., disgust sensitivity, distorted cognitions about the consequences of eating feared foods)

• A non-concurrent multiple baseline design followed by 4-week CBT
• Various measures taken at baseline and throughout including measurement of DSM-5 ARFID diagnosis, food neophobia, body weight and anxiety
• Follow-up 3-months post treatment | months post discharge • Improvement on cognitive domains, energy levels and anxiety
• At follow up, 10 of the 11 patients were at a healthy weight and had an age adequate nutritional intake
• For most, food neophobia scores decreased to a nonclinical range
• Dysfunctional cognitions about food intake/eating and anxiety decreased
• Tube feeding eliminated in 6 Patients
• All 11 patients demonstrated a more varied food repertoire
• Demonstrates a CBT approach which has the potential to treat various issues which drive restrictive/avoidant eating behaviours in ARFID | Lock 2018 [18] | USA | To illustrate the use of FBT in treating preadolescents with ARFID | Case study (1) 8-year-old female (2) 9-year-old female (3) 11-year-old female | 3 different ARFID presentations: (1) Low appetite and lack of interest in eating (2) Sensory aversion to food (3) Fear of eating and extreme fear of vomiting | Family Based Therapy | (1) No major changes in interest in food but capable of eating sufficient quantities and eating-related family conflicts decreased (2) Greatly increased range of food, increased flexibility in social situations (3) Coping strategies used to manage fears, steady weight

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| Bloomefield 2019 [21] USA | To examine the use of teleconsultation in treating a patient with ARFID | Case study 8-year-old-male Frequent refusal of non-preferred foods resulting in tantrum behaviour (whining, crying, gagging) upon sight or smell | - Parent teleconsultation (behavioural feeding intervention to increase food variety)- Follow-up 1- and 4-months post-treatment | Increase in the frequency of bites of non-preferred foods

| Dahlsgaard & Bodie 2019 [22] USA | To report the acceptability, feasibility and initial outcomes of the Picky Eaters Clinic | Pilot trial 21 children with a diagnosis of ARFID (4—11 years) and their Parents Picky eaters (eating less than 20 foods, difficulty socialising, refusal to eat non-preferred foods) | -7 sessions (90 min each) of parent-led behavioural intervention- Follow-up 3-months post treatment | Reduction in picky eating and negative mealtime behaviours

| Zucker 2018 [23] USA | To present an acceptance-based interoceptive exposure treatment for young people with ARFID and demonstrate its success in treating a | Case study 4-year-old female - Patient had percutaneous endoscopic gastrostomy (PEG tube) since 14 months of age- Indifference to food, lack of awareness of | 8 weekly sessions followed by 4 bimonthly sessions of acceptance based interoceptive exposure treatment - Feeling and Body Investigators (FBI)-ARFID Division (also mirtazapine for a month prior to exposure treatment) | - Patient no longer met criteria for ARFID- Notable improvement in capacity to cope with change, unknown internal sensations no longer viewed as a threat - Increase in quantity of food

Multi-modal approach

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One further study which investigated FBT for the treatment of ARFID has been published since the systematic review by Bourne et al (2020) [5] (Table 2).

Table 2: Additional published ARFID treatments

| #3: general disinterest in food and eating, as well as limited variety. Anxiety and depression | - 19 treatment days | #3: Psychologist encouraged mother to increase food variety, calories, and consistency in her interactions during meal times | -19 days in PHP | - 12 days in Intensive outpatient program

Discussion points

There are no well-established treatments for ARFID, with a limited number of randomized clinical trials among patients with ARFID. This literature review evidences several promising treatment avenues which warrant further study:

• FBT, CBT and adjunctive pharmacological intervention appear to be the methods with the best evidence.
• A multi-modal approach is also endorsed, particularly for those with severe feeding difficulties.
  • Overall consensus is that this must be individualised, depending on the main concern and degree of severity.

Despite the phenotypically heterogeneous nature of ARFID, there is currently no direct evidence that different presentations warrant diverse interventions. Indeed, Dumont et al. (2019) [17], have demonstrated that a flexible CBT approach can be used to treat ARFID with several presentations. Of course, we will only be able to recognise whether different methods are necessary when we know more about the nature of this heterogeneity and begin to test patient responses.

There are several other worthwhile directions for further research including an investigation into ARFID’s psychiatric comorbidity, since it has been found to co-occur with various other diagnoses such as generalised anxiety disorder, obsessive compulsive disorder and autism.

Limitations

1. Included studies were of low quality (mainly case studies) with small sample sizes. Further research will need to focus on larger RCT’s which use consistent population characteristics and outcome measures.
2. There is a wealth of literature relating to sub-clinical restrictive eating behaviours which are symptomatically similar to ARFID, as well as studies pre-dating the introduction of ARFID which would likely provide valuable treatment options for the disorder.

Intensive Multidisciplinary Intervention for Paediatric Feeding Disorders

A systematic review conducted by Sharp et al (2017) [28] investigated the medical literature regarding treatment of paediatric feeding disorders at inpatient and day treatment programs. The authors summarise treatment models and outcome measures, and evaluate the evidence with the use of both descriptive and meta-analytic procedures. The sample characteristics (Table 3) and treatment settings and interventions characteristics (Table 4) are summarised below.

Summary of results

Treatment settings and approach to intervention

• 11 included studies (2 RCT and 9 Non Randomised Studies)
• Collectively the studies include 593 participants (age range 15.7–48 months; 314 boys and 279 girls)
• Treatment for feeding tube dependence (n = 535; 90.2%), liquid formula to meet nutritional needs (n= 22; 3.7%), remaining 36 (6.1%) subjects had various feeding problems but were not tube or formula dependent.
• 8 studies delivered treatment in inpatient facility and 3 in day treatment program and 1 within both settings
• Multiple treatment interventions:
  • Behavioural intervention: positive reinforcement of appropriate mealtime behaviours, bite persistence (aka, contingency contacting, escape extinction), and/or stimulus fading—represented the most common treatment approach
  • Oral motor exercises aimed to decrease tactile hypersensitivity and/or increasing the range, strength and control of the lips, cheeks, jaw and tongue
  • Tube weaning: restriction and then reduction
  • Nutritional intervention: calculation of energy needs, monitored hydration, adjust tube feeds, tracking of advances
• All studies involved care givers in treatment

• No study, however, provided specific data on caregivers’ acceptance, mastery, and adoption of treatment strategies

4.1.2 Treatment outcomes

• 43% to 100% (Mean 69.8% [SD 21.6%]) of patients were weaned from enteral feeding tubes across the 8 studies that reported this outcome.

• Six studies reported improvement in oral consumption during meals, ranging from 38% to 100% (Mean 74.5 [SD 21.5]) following intervention.

• 36% of studies reported additional gains at follow up, however, 27% reported resumption of tube feeding

• Four studies that included behavioural intervention without tube weaning reported stabilization or improvement in weight.

• The 6 studies that involved tube weaning as a primary treatment component reported weight loss at discharge. Of these, 4 reported on the percentage of weight loss, which ranged from 4% to 9.2%.

• Dependence on enteral feeds was eliminated in 71% of children at discharge. When documented, these benefits appear to persist, with 80% of patients tube-free at follow-up.

Table 3: Summary of sample characteristics