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Received: 12 December 2019    Revised: 12 May 2020    Accepted: 18 May 2020

DOI: 10.1111/apa.15372

REGULAR ARTICLE

Controlled case series demonstrates how parents can be


trained to treat paediatric feeding disorders at home

Tessa Taylor1,2  | Neville Blampied2  | Nikolas Roglić1

1
Paediatric Feeding International, Sydney,
NSW, Australia Abstract
2
Department of Psychology, and the Aim: Paediatric feeding disorders are normally managed by specialist clinics. We ex-
School of Health Sciences, University of
amined whether treatment gains were maintained when trained parents continued
Canterbury/Te Whare Wānanga o Waitaha,
Christchurch, New Zealand the programme at home and during meals out.
Methods: This controlled consecutive case series recruited 26 children (22 boys) with
Correspondence
Tessa Taylor, Department of Psychology, avoidant/restrictive food intake disorder, from a private paediatric feeding disorders
Speech & Hearing, and the School of Health
practice in New South Wales, Australia. Their mean age was six (2-13) years. All had
Sciences, University of Canterbury/Te
Whare Wānanga o Waitaha, Private Bag severe feeding problems and mealtime skill deficits, and most had autism and de-
4800, Christchurch 8140, New Zealand.
velopmental delays or intellectual disabilities. The children received intensive, indi-
Email: DrTaylor@PaediatricFeedingIntl.com
vidualised, behaviour-analytic treatment for 11 (6-21.5) days, and the parents were
trained to continue it at home. The primary treatment outcomes included the range
and amount of food eaten and mealtime behaviour.
Results: The children met all of the therapeutic goals agreed at the treatment out-
set. They ate a mean of 92 different foods and improved how they ate, drank and
behaved during mealtimes. The mean differences before and after treatment were
clinically and statistically significant, and the gains were maintained during follow-up
at a mean of 2.3 years. Parental satisfaction and treatment acceptability were high.
Conclusion: Specially trained parents successfully continued paediatric eating disor-
der treatment at home and maintained treatment gains.

KEYWORDS
avoidant/restrictive food intake disorder, home treatment, inappropriate mealtime behaviour
paediatric feeding disorders, parental training

1 |  I NTRO D U C TI O N disabilities. 2,3 Many meet the criteria for avoidant/restrictive food
intake disorder (ARFID), which includes failure to thrive, nutritional
Chronic and severe paediatric feeding disorders can include being deficiencies, dependence on artificial feeding methods and, or, di-
dependent on a gastrostomy tube or liquids, refusing food and only etary supplements and marked impairment in psychosocial function-
eating certain items. Feeding problems occur in wide-ranging popu- ing.4 When this occurs during critical neuro-developmental periods,
lations, including children with typical development, medical com- numerous adverse effects on behaviour, cognition, emotion and
plications, such as food allergies and reflux,1 and developmental health may occur and persist into adulthood.3-7
Over the past 40 years, treatment for paediatric feeding disor-
Abbreviations: ARFID, avoidant/restrictive food intake disorder; ES, effect size; CI, ders has been based on behaviour analysis principles and research
confidence interval.

© 2020 Foundation Acta Pædiatrica. Published by John Wiley & Sons Ltd

Acta Paediatrica. 2020;00:1–9.  |


wileyonlinelibrary.com/journal/apa     1
|
2       TAYLOR et al.

has delivered a range of empirically supported, effective treatments.


However, these have only been generally available from specialised
Key Notes
multidisciplinary, hospital-based research clinics served by staff with
• This study examined whether treatment gains were
high levels of expertise in the treatment methods.4,8-11 Most children
maintained when specially trained parents treated pae-
do not have access to this treatment.7,12 In general, conventional
diatric feeding disorders at home.
treatment methods do not have a high-quality evidence base world-
• We recruited 26 children aged 2-13 years with avoidant/
wide.4,12-17 Thus, lack of awareness of, and access to, effective treat-
restrictive food intake disorder, and most had autism
ment may lead to continuing suffering and distress for children and
and developmental delays or intellectual disabilities.
their families. Repeated treatment failure can also waste healthcare
• Some 2 years after the programme started, the chil-
resources and time.7 Translational research 18 is urgently needed to
dren were eating a mean of 92 different foods and
demonstrate the feasibility of adapting specialist, in-hospital treat-
12,19,20 had improved how they ate, drank and behaved during
ment so that it can be delivered at home and in the community.
mealtimes.
We also need research to evaluate treatment outcomes and show
that gains can be maintained over time and in different settings.
Our primary aim was to demonstrate what could be achieved
when effective behaviour-analytic treatment was translated from experienced unsuccessful treatment for a mean of 3.5 ± 3.0 years, and
specialist hospital settings and adapted so that specially trained par- the maximum was 11 years. Multiple treatment experiences were com-
ents could use it at home and during meals out. It presents the first mon, with a mean of 5 ± 4 treatments and a maximum of 12. Most chil-
analysis of the overall outcomes achieved with a consecutive, con- dren were receiving ongoing services and monitoring, but these were
trolled case series that received in-home, behaviour-analytic feeding paused during the intensive behavioural treatment programme and the
treatment without any additional food-related therapy. previous feeding targets did not continue after the programme. The
therapists and parents agreed on a mean of 11 treatment goals (range
7-17), which were used to provide individualised treatment. The ses-
2 |  M E TH O DS sions were held in settings with the necessary seating, equipment and
supplies. The primary setting was at home, and the other settings were
The intensive, individualised treatments used in this translational cafes, restaurants, day care centres and schools.
effectiveness trial study were adapted from Taylor et al19 The
study took place from May 2016 to March 2019 at a private clini-
cal practice specialising in treating paediatric feeding disorders in 2.2 | Measurement, reliability, integrity and
New South Wales, Australia. It was performed in accordance with acceptability
the Declaration of Helsinki and the relevant ethical standards for
psychologists and behavioural analysts. The parent provided writ- A trained observer computer-coded the children and therapists'
ten, informed consent for the therapy and the subsequent use of the behaviours using specialised real-time data collection software
de-identified data. (BDataPro, Kennedy Krieger Institute; 21). The frequency keys
recorded the frequency of responses, including acceptance,

2.1 | Patients TA B L E 1   Characteristics of the 26 children in the study cohort

n (%)
We reviewed 29 consecutive cases and reported the outcomes re-
ported for 26 children who were diagnosed with ARFID by a registered Male 22 (85%)
clinical psychologist and doctoral-level certified behavioural analyst Autism, developmental delay 20 (77%)
with specialist training in paediatric feeding disorders. Three cases Growth impairments 14 (54%)
were excluded because they were receiving other therapies. Each Constipation 11 (42%)
child's physician approved their participation and provided medical in- Nutritional deficiencies 8 (31%)
formation, such as prior screening, growth and medical history, food Formula or baby bottle dependence 8 (31%)
allergies and any multidisciplinary specialist and testing referrals. The
Genetic/chromosomal abnormalities 5 (19%)
detailed patient characteristics can be found in Table 1, and the meth-
History of nasogastric tube 3 (12%)
odology is summarised here and described in more detail in Appendix
Eczema 3 (12%)
S1. The average age of the 26 children was six (range 2-13) years: 15
Gastrostomy tube 2 (8%)
were Caucasian Australian and the others were of Asian, Arabic and
Parenteral nutrition 1 (4%)
European ethnicities and nationalities. Most of the families had high so-
cio-economic status. At their initial assessment, the children displayed a Coeliac disease 1 (4%)

range of severe feeding problems and mealtime skill deficits.7 They had Note: Other factors not detailed here were prematurity and reflux.
TAYLOR et al. |
      3

swallowing, inappropriate mealtime behaviour and expulsion. The control, using within-subjects, single-case experimental designs,
duration keys measured delays in seconds before acceptance and where each case served as its own control. 23 The most frequently
swallowing occurred and the duration of negative vocalisation. used design, for more than 80% of the children, was a reversal, or
Frequency of swallowing was divided by the number of therapist- withdrawal, design. This involved taking the treatment out, namely
programmed bite presentations during the session to provide an baseline two, and putting it back in when needed, namely treat-
overall measure of the percentage of food consumed. The last ment two. This was because replication showed that the treatment
three data points in the initial phases, namely baseline one and was needed and was the reason for the improvement in eating.
treatment one, were averaged, as were the repeated baseline two The outcomes were examined using a combination of graphic tech-
and treatment two phases. The plates were photographed to re- niques24 and effect size estimation, using Cohen's dav, where the
cord the variety of the food eaten, and the food was measured standardiser was the average of the standard deviations before
before and after eating to determine the volume consumed. All and after treatment. 25 The graphs were predominantly modified
19
parents were provided with a questionnaire at the end of the Brinley plots (Figure 1;26). These are scatterplots that display indi-
programme, and the responses to the five-point Likert scale items vidual changes over time, with each data point representing coor-
were averaged to assess the 23 programme satisfaction items and dinates of an individual's score at time one on the x-axis and time
the 16 items on social acceptability of the treatment. All the ses- two on the y-axis.
sions were video-recorded to assess the interobserver agreement
and reliability, which was 96%. 21 The mean responses per minute
for lapses in procedural integrity for each minute were 0.1 lapses 2.4 | Procedure
for the therapist and 0.2 for the parents, and the mean interob-
server agreement was 99.5%. The mean waiting time for treatment was 3.5 (range 1-10) months.
Once it had started, the children received intensive continuous treat-
ment, which was provided for about 6 days per week for 2-4 weeks
2.3 | Experimental design and data analysis during which they received treatment for an average of 11 (6-21.5)
days. The first author was the principal therapist and conducted the
The study included all cases that met the inclusion criteria, regard- sessions for approximately 7-8 hours per day with a trained assistant.
22
less of outcome, to reduce selection and publication bias. This The involvement of the therapist reduced as the parents developed
was consistent with the consecutive, controlled case series experi- the skills they needed and the responsibility for the intervention was
mental design. All the 26 cases also demonstrated experimental gradually transferred to them.

Modified Brinley plot interpretaon

Reducon = improvement Increase = improvement

Deterioriation Improvement
Final Treatment

ge
ch f
o
an
ne
Li
no

Improvement
Deterioriation

Baseline Baseline

F I G U R E 1   Illustrates the key features of modified Brinley plots and the interpretation of the graph space when a reduction in score
indicated clinical improvement (left panel) and when an increase indicated improvement (right panel). The + shows the coordinates of the
baseline and final treatment means for the lowest data points. Data points that fall near, or on, the 45° diagonal line of no change represent
individuals who showed little or no change between time one and time two. The magnitude of individual change is displayed by points lying
at greater distances above and below the line. Depending on the direction of the clinical and desired change for the measure, any change can
be classified as an improvement or deterioration. To aid interpretation, 26 the group mean coordinate is shown by a plus sign and the Cohen's
dav effect size 25 is displayed along with the 95% confidence Interval (95% CI), calculated using ESCII software developed by Cumming, La
Trobe University, Melbourne, Australia 28
4       | TAYLOR et al.

Percentage consumpon from baseline to final treatment


Baseline 1 vs Treatment 1 Baseline 1 vs Baseline 2 Baseline 2 vs Treatment 2

100

Treatment 2
Perc ent of bites c ons um ed

80 Treatment 1

Baseline 2 Baseline 2
60

40

20
Baseline 1

0
0 3 6 9 12 15 18 21 24 27 0 3 6 9 12 15 18 21 24 27 0 3 6 9 12 15 18 21 24 27
Parcipants (in order of case number)

F I G U R E 2   Horizontal dot plots show the percentage of bites consumed by each child in initial baseline one compared to initial treatment
one (left panel), replication of baseline two compared to initial baseline one (middle panel) and replication of final treatment two compared
to replicated baseline two (right panel). Pairs of data points are shown for each child in consistent case-number order from child one on the
extreme left of the x-axis to child 26 on the extreme right of each panel. Pairs of percentages (one above the other) are displayed for each
case sequentially for case one to case 26 in the same order over the three figure panels

2.5 | Assessment and treatment end of the programme, they were asked to offer their child the foods
on the discharge food list, record mealtime data, take photographs
The review of the intake assessment included the physician's report of the plates and video the meals. The data were returned to the
and reports from other disciplines and service providers, meal obser- first author for review and follow-up. The parents were contacted
vations, intake questionnaires, parental interviews, three-day diet 2-3 years after treatment, at a mean of 2.3 years, and asked to com-
records, functional assessment, case-conceptualisation and therapy plete a five-point Likert satisfaction survey, which ranged from one
goal specification. We directly assessed each child's food and reward for worse than pre-treatment to five for resolved.
preferences.19,27 Foods from each food group were targeted and the
spoon size and food texture matched individual needs. Treatments
were individualised and data-driven, with components selected from 3 | R E S U LT S
8-10,12
well-established, empirically supported treatments. Treatment
involved changing the mealtime environment, such as providing Figure 2 presents the primary outcome data, which was consump-
incentives for swallowing and praising appropriate mealtime be- tion, and demonstrates the experimental control as horizontal dot
haviour. Treatment also involved setting achievable mealtime re- plots. Food consumption was very low at the initial baseline meas-
quirements, such as continuing to present food and liquids despite ure, baseline one, with most children not consuming any of the food
rejection, replacing food or liquid that was spat out and providing presented. At the end of the first treatment phase, treatment one,
prompting instructions and guidance. We also taught the skills children were completing 100% of the programmed bites consist-
needed to achieve goals, such as chewing, using utensils, self-feed- ently, within a mean of 46 (2-134) minutes of treatment starting.
ing and drinking, drinking with an open cup, biting from whole food When treatment was withdrawn as a replication procedure, at base-
portions rather than pre-cut bite sizes and taking medicine. As the line two, consumption decreased for all children. When treatment
treatment progressed, intensive parental behavioural skills training resumed in the second treatment phase, namely treatment two, con-
was provided and we varied the cutlery and crockery provided, how sumption increased again for all children, with no overlap of points
the children were placed at family meals and the setting where they with the baseline, thus replicating the treatment effect. At the end
ate19 to ensure generalisation of therapy gains. of the programme, the children were consuming an average of 158
(52-292) grams of solids per meal.
The number of foods the children ate increased from an av-
2.6 | Maintenance and follow-up erage of six, mainly starch, to a mean of 92 from all food groups,
which equated to a very large Cohen dav effect size of about four
To help the children maintain their treatment gains, the parents were (Figure 3). One child could only eat 30 different foods due to aller-
given specific maintenance instructions. For two weeks after the gies and was awaiting tests because of vomiting (Figure 3). Examples
TAYLOR et al. |
      5

of the meals consumed are shown in Figure 4. Figure 5 shows that goal levels at baseline. Other reductions were 99% for food refusal,
for all children, inappropriate mealtime behaviour, expulsion and 97% for food expulsion and 97% for negative vocalisation. All the
negative vocalisation at the end of treatment were low or reduced. children met 100% of the treatment goals, including learning to take
The overall effects sizes for all three measures were large (Cohen's medication, chewing, feeding themselves with utensils, drinking
dav −1.4 to −2.35). Before treatment, there were long delays to tak- from a cup and sitting for meals.
ing and swallowing bites which decreased after treatment (Figure 6). At discharge, 20 parents (77%) completed the questionnaire.
Individual differences in the nature and texture of the food and how They reported high satisfaction (mean 4.9, range 4.6-5, maximum
skilled the children were at chewing and swallowing accounted for 5.0) and social acceptability of the treatment (mean 4.9, range
the considerably lower Cohen's dav effect size for swallowing, of 4.6-5, maximum 5.0). During the immediate follow-up period of
about −1, than for accepting food when presented (−2.6). two weeks, 17 parents reported on a mean of 25 (4-135) meals.
The Cohen's dav effect sizes were large to very large (range They reported high and stable consumption of the food presented
1.4-4) across the different measures and in no instance did the 95% (mean 99%, range 90%-100%). Anecdotally, families reported im-
confidence interval (95% CI) indicate that the effect size was close provements in other areas of life and well-being, for example in
to zero, as the associated mean change was always statistically sig- height, weight and body mass index, bloodwork for nutritional
nificant (P < .05). When we measured the treatment effect, based on deficiencies, hair quality and growth, toothbrushing, sleeping
the average percentage reduction from baseline to the end of the and toileting. They also reported improvements in their child's
treatment, it was 98% for non-consumption. Delays in acceptance behaviour, such as being able to change between activities and
fell by 97% and 75% for delays to an empty mouth, but the figure follow instructions. Improvements in learning were also reported,
was 89% when we excluded the five children who had reached these such as progress in therapy and school. Other benefits were

Variety of foods consumed from before treatment to final treatment


160
150 Number of Foods Consumed
Fi nal Treatment - Total Number of Foods

140
130
120
110
100
90
80
70
60
50
40
dav = 4.2 [3.1, 5.7]
30
20
10
0
0 10 20 30 40 50 60 70 80 90 100 110 120 130 140 150 160

Baseline - Total Number of Foods

45
Protein Starch Veges Fruit Combo
40
Final Treatment - Number consumed

35

30

25

F I G U R E 3   Modified Brinley plots 20

showing total number of foods consumed


15
before and at the end of treatment (top
figure) and truncated modified Brinley 10
plots showing the range of different
food groups consumed after treatment 5

compared to before treatment (bottom


0
figure). The diagonal line in every plot 0123456789 0123456789
0123456789 0123456789 0123456789
indicates the line of no change from
before treatment to the end of treatment Baseline - Number consumed
|
6       TAYLOR et al.

Sample pictures of plates children consumed

F I G U R E 4   Sample pictures of meals the children consumed

related to energy, sports and social participation, travel, immunity, that their children were eating from all food groups. The exception
skin colour and decreased dependence on items like supplements, was a child who had similar before and after results as moving home
formulas and laxatives. One example of growth improvement was meant their parents did not maintain the protocol.
an 11-year-old child who was overweight and had stunted growth
with a blood analysis that revealed 18 significant deficiencies and
abnormalities. After he was discharged, his parents reported that 4 | D I S CU S S I O N
he had rapidly grown 25 cm in height and this was confirmed by
his therapist. The data analysis used in this study showed both individual and over-
At follow-up, 21 parents reported that their child's feeding prob- all group patterns of change following therapy. The consistently large
lem was much better than before treatment and five said it had com- size of change seen in the figures was confirmed by the standardised
pletely resolved. The mean Likert score was 4.12 (2-5). No parents mean difference effect size and 95% CI estimation, with Cohen's dav
reported the problem was worse. All but one of the parents reported values that were large to very large and statistically significant. This
TAYLOR et al. |
      7

Inappropriate mealme behaviour, expulsion, F I G U R E 5   Modified Brinley plots comparing before treatment
and negave vocalisaons from before treatment with the end of treatment. These show changes in the frequency
to final treatment of inappropriate mealtime behaviour (top panel), the rate at which
food was expelled by each child (middle panel) and the percentage
of sessions with negative vocalisations (bottom panel). The diagonal
Inappropriate Mealtime
35 line in every plot indicates the line of no change from before
Final Treatment - Refusals/minute

Behaviour
treatment to the end of treatment
(IMB)
30

dav = –2.1 highly positive outcome of the treatment was firstly the result of
25
matching the treatment to each child and, second, continuing with
[–2.85, –1.275]
20 treatment until all target goals were met. Confidence in conclusions
was enhanced because the interobserver agreement and procedural
15
integrity measurements showed that the therapy data were accu-

10 rately recorded and that the therapists accurately followed the pro-
tocol, as the data quality and treatment fidelity were high.
5 It was also a strength of this study that the parents were trained
to a high standard so that they could continue to use the meal pro-
0
tocols independently, rather than needing long-term professional
0 5 10 15 20 25 30 35 services. Treatment was tailored by directly working with each
Baseline - Refusals/minute child rather than adopting a generic approach. Parents were then
thoroughly trained on precisely established procedures that were
11 Rate of Food Expels specifically designed for their child and had already been shown
10
Final Treatment - Expels/minute

to work. The treatment was safe, ethical and posed no risks to the
9 dav = –1.44 [–2.1, –.79] child's health, as none of the therapy involved depriving children of
8 preferred foods, drinks or formula. It also encouraged children to
7 eat various nutritious foods from all food groups. Furthermore, the
6 children were taught the mealtime skills they needed for age-appro-
5 priate independence and so that they would accept different food
4 textures.

3 A limitation of this study was the lack of observational and sys-

2 tematic, standardised data during the follow-up period and the lack
of intake data while children were on the waiting list for treatment.
1
Another limitation was that their body mass index could not be cal-
0
culated during these two periods. However, the children increased
0 1 2 3 4 5 6 7 8 9 10 11 their consumption quickly, on the first day, so there were no con-
Baseline - Expels/minute cerns about any further weight loss during treatment, except for one
child with malabsorption and diarrhoea due to a genetic condition
100
Negative Vocalisations and vomiting. Future studies should include multi-method, multi-
90 trait measures that are comparable from the time of intake through
Final Treatment - % of Sessions

80 dav = –2.35 [–3.1, –1.5] to follow-up periods. More research is also needed on younger age

70 groups and early intervention and prevention methods. The current


study was carried out with mostly high socio-economic status fami-
60
lies, due to lack of government funding in this treatment, so it is not
50 clear whether the training and follow-up results would be the same
40 for low status families.

30 It should be noted that most of the children in our study were


not dependent on tube feeding. The severity of typical presenta-
20
tions would probably be higher in hospital settings than in our pri-
10 vate practice, because such clinics may have more patients with
0 long-term gastrostomy tubes and higher levels of more severe med-
0 10 20 30 40 50 60 70 80 90 100 ical complications and developmental issues. Future studies might
Baseline - % of Sessions replicate this treatment model with children who depend on tube
feeding, as they may need longer treatment, especially to teach
them to chew and drink from a cup and to accept different food
|
8       TAYLOR et al.

Time taken (seconds) to accept and swallow bites comparing before treatment to final treatment
Latency to Acceptance Latency to Swallow (Mouth Clean)

Final Treatment
180

Latency (sec)
120 dav = –2.59 [–3.5, –1.7] dav = –1.3 [–1.95, –.65]
60

0
0 60 120 180 240 300 360 420 480 540 600 0 60 120 180 240 300 360 420 480 540 600
Baseline Latency (sec)

F I G U R E 6   Truncated modified Brinley plots showing latency, in seconds, to food acceptance into the mouth upon presentation and
latency to showing a clean mouth, namely swallowing, on inspection following acceptance before treatment and at the end of treatment.
The diagonal line represents the line of no change

textures following tube weaning. Also, certain children may require help many more families coping with severe and chronic paediatric
hospitalisation and multidisciplinary teams if they have swallowing feeding disorders.
problems, severe food allergies, breathing issues, seizures and blood
sugar issues, such as glycogen storage disease. Treatment intensity C O N FL I C T O F I N T E R E S T
and duration needs to be individually calibrated to the severity of The authors have no conflicts of interest to declare.
cases and decisions about treatment should be data-driven and
based on progress towards goals. If not, there is a risk of iatrogenic ORCID
worsening of the feeding disorder.4,11,19 It is important to note that Tessa Taylor  https://orcid.org/0000-0001-8643-3955
the current study was not conducted in the same way as a conven- Neville Blampied  https://orcid.org/0000-0002-0158-4904
tional outpatient service, as we provided continuous sessions for full
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