Boston University

OpenBU http://open.bu.edu
Theses & Dissertations Boston University Theses & Dissertations

2017

Understanding ARFID: clinical

characteristics of patients who
meet avoidant/restrictive food
intake disorder criteria in a
multidisciplinary pediatric growth
and nutrition clinic

https://hdl.handle.net/2144/23696
Boston University
 BOSTON UNIVERSITY

SCHOOL OF MEDICINE

Thesis

UNDERSTANDING ARFID: CLINICAL CHARACTERISTICS OF PATIENTS

WHO MEET AVOIDANT/RESTRICTIVE FOOD INTAKE DISORDER

CRITERIA IN A MULTIDISCIPLINARY PEDIATRIC GROWTH AND

NUTRITION CLINIC

by

ROSHEN T. JOHN

B.A., Dartmouth College, 2015

Submitted in partial fulfillment of the

requirements for the degree of

Master of Science

2017
© 2017 by
ROSHEN T. JOHN
All rights reserved
 Approved by

First Reader
Karen Symes, Ph.D.
Associate Professor of Biochemistry

Second Reader
Susanna Huh, M.D., M.P.H.
Associate Director, Center for Nutrition
Boston Children’s Hospital;
Assistant Professor of Pediatrics
Harvard Medical School
 ACKNOWLEDGMENTS

I would like to sincerely thank Dr. Susanna Huh for her tireless teaching, patience, and

mentorship over the past few months. Her dedication and guidance made this work

possible, and I am so grateful to have been provided such an incredible opportunity.

iv
 UNDERSTANDING ARFID: CLINICAL CHARACTERISTICS OF PATIENTS

WHO MEET AVOIDANT/RESTRICTIVE FOOD INTAKE DISORDER

CRITERIA IN A MULTIDISCIPLINARY PEDIATRIC GROWTH AND

NUTRITION CLINIC

ROSHEN T. JOHN

ABSTRACT

Introduction: Feeding difficulties are commonly multifactorial in nature, and no

uniformly agreed-upon classification system for feeding difficulties currently exists. The

Diagnostic and Statistical Manual of Mental Disorders (DSM)-V included a new

diagnosis called Avoidant/Restrictive Food Intake Disorder (ARFID), created in order to

address the weaknesses of the DSM-IV-text revision (TR) classification system by better

capturing the range of feeding difficulties typically found in clinical practice. Little is

known about the clinical characteristics associated with meeting the ARFID criteria, and

no studies have investigated ARFID prevalence and associated clinical characteristics in

patients below the age of 8 years.

Aim: To describe the clinical characteristics of a sample of patients referred to

Boston Children Hospital’s Growth and Nutrition Program, including the prevalence of

ARFID, and identify clinical characteristics associated with meeting the criteria for

ARFID.

Methods: We examined prospectively collected data from 69 subjects, age 9

months to 7 years, referred to the Growth and Nutrition Program for feeding difficulties

and/or malnutrition between November 2013 and April 2016. Data was collected from

v
caregiver-completed questionnaires, including the Behavioral Pediatrics Assessment

Scale (BPFAS), and each patient’s electronic medical record.

Results: Premature birth (32.3%), digestive conditions (69.2%), developmental

conditions (56.9%), food allergy (20.3%), and meal duration of over 30 minutes (36.2%)

were common. Problematic feeding behaviors such as refusing to eat (62.1%) and

gagging or vomiting when given new foods (29.2%) were also common. Strategies

caregivers used to increase food and liquid consumption included offering only foods the

child likes (60.9%) and feeding in front of the television or electronic devices (30.4%).

90.8% had a BPFAS score above threshold. 83.1% of the sample met criteria for ARFID.

No statistically significant relationship was found between meeting ARFID criteria and

having a BPFAS score above threshold, and there was no statistically significant

relationship between meeting ARFID criteria and having a food allergy, having a first-

degree relative with a food allergy, or with any of the feeding behaviors or strategies we

investigated.

Conclusion: This study suggests that the majority of patients between the ages of

9 months to 7 years with feeding difficulties referred to the Growth and Nutrition

Program meet the criteria for ARFID. While no statistically significant relationship was

found between ARFID and the investigated clinical characteristics, further analysis

involving a larger sample of patients will be useful for better understanding the clinical

characteristics associated with ARFID, and assessing ARFID’s clinical utility.

vi
 TABLE OF CONTENTS

TITLE……………………………………………………………………………………...i

COPYRIGHT PAGE……………………………………………………………………...ii

READER APPROVAL PAGE…………………………………………………………..iii

ACKNOWLEDGMENTS ................................................................................................. iv

ABSTRACT ........................................................................................................................ v

TABLE OF CONTENTS .................................................................................................. vii

LIST OF TABLES ........................................................................................................... viii

LIST OF ABBREVIATIONS ............................................................................................ ix

BACKGROUND ................................................................................................................ 1

1. Feeding Difficulties ............................................................................................... 1

2. Classification of Feeding Difficulties………………………………....…....……9

SPECIFIC AIMS………………………………………………………………………...21

METHODS ....................................................................................................................... 22

RESULTS ......................................................................................................................... 26

DISCUSSION ................................................................................................................... 36

REFERENCES ................................................................................................................. 40

CURRICULUM VITAE ................................................................................................... 46

vii
 LIST OF TABLES

Table Title Page

1 Etiologic mechanisms of feeding difficulties and selected 2

examples

2 BPFAS threshold scores 13

3 Classification of feeding difficulties 14

4 Avoidant/Restrictive Food Intake Disorder Criteria 16

5 Sample demographics 26

6 Birth history 27

7 Child medical history 28

8 Feeding behaviors in the past month 29

9 Feeding strategies used by caregivers in the past month 30

10 BPFAS sub-scores 31

11 ARFID prevalence 31

12 No statistically significant association between ARFID 32

and BPFAS score above threshold

13 ARFID and meal behaviors in the last month 33

14 ARFID and meal strategies to increase food or liquid 35

consumption in the last month

viii
 LIST OF ABBREVIATIONS

ARFID............................................................... Avoidant/Restrictive Food Intake Disorder

ADHD…………………………………………..Attention-Deficit/Hyperactivity Disorder

ASD............................................................................................ Autism Spectrum Disorder

ASD........................................................................................................Atrial Septal Defect

BPFAS………………………………..….Behavioral Pediatric Feeding Assessment Scale

CF…………………………………………………..…Child Frequency BPFAS sub-score

CP………………………………………………………..Child Problem BPFAS sub-score

CPAP………………………………………………..Continuous Positive Airway Pressure

CPSP…………………………………………..Canadian Paediatric Surveillance Program

DSM………………………………Diagnostic and Statistical Manual of Mental Disorders

GERD…………………………………………………...Gastroesophageal Reflux Disease

PDD…………………………………………………....Pervasive Developmental Disorder

PF………………………………………………….…Parent Frequency BPFAS sub-score

PP……………………………………………………….Parent Problem BPFAS sub-score

RAST……………………………………………………………..Radioallergosorbent test

ROC………………………………………………….…Receiver Operating Characteristic

SAS…………………………………………………………..Statistical Analysis Software

VSD…………………………………………………………...…Ventricular Septal Defect

ix
 BACKGROUND

1. Feeding difficulties

a. Definition

Learning to independently self-feed sufficient quantity and diversity of food and

beverages to meet nutritional and hydration requirements is an essential life skill acquired

during the typical development of infants and children. The feeding process is an

inherently interactive experience that plays an important role in promoting social and

psychological development. Feeding problems are associated with a wide range of

problematic developmental effects, including later deficits in cognitive development,

eating disorders and behavior problems (Sullivan, 2016; Marchi & Cohen, 1990; Dahl,

1987). There is no agreed-upon nomenclature uniformly used to describe feeding

problems. Terms such as mild “picky eating” and more severe “feeding disorders” do not

have precise definitions, but are used in the literature in an effort to differentiate among

the wide range of severity and etiology that categorizes various feeding problems. The

term “feeding difficulties” is often used as an umbrella term to mean any type of feeding

problem, regardless of severity or etiology (Kerzner et al., 2015).

b. Epidemiology

The lack of universally accepted definitions makes it challenging to precisely

estimate the prevalence of feeding difficulties. However, previous studies suggest feeding

difficulties to be common, affecting approximately 25% of otherwise normally-

1
developing infants and young children and up to 80% of infants and young children with

developmental disabilities (Chatoor, 2009; Benjasuwantep, Chaithirayanon, &

Eiamudomkan, 2013; Carruth, et al., 2004). Feeding difficulties are often a source of

great concern for parents. Carruth et al., 2004 found the percentage of children in a

national random sample perceived by their caregivers as being “picky eaters” was 19% at

4 months of age, and 50% at 2 years of age.

c. Etiology

Adequate feeding depends on a wide range of anatomical, physiological,

metabolic, and behavioral factors. Therefore, feeding difficulties can arise from a several

different underlying mechanisms. Table 1 details common mechanisms underlying

feeding difficulties and selected examples.

Table 1. Etiologic mechanisms of feeding difficulties and selected examples.

Altered appetite
• Abnormal feeding patterns
• Supplemental feedings
• Inflammatory bowel disease
Diet
• Food allergy
• Inappropriate foods
Dysphagia
• Anatomic:
o Macroglossia
o Cleft lip and palate
o Submucous cleft palate
o Pierre Robin sequence
o Laryngeal cleft
o Tracheoesophageal fistula
o Esophageal stricture
o Retropharyngeal mass

2
 o Vascular ring
o Foreign body
• Neuromuscular:
o Prematurity
o Cerebral palsy
o Bulbar palsy
o Rett syndrome
o Infant botulism
o Muscular dystrophy
o Pseudo-obstruction
o Connective tissue disease
o Repaired tracheoesophageal fistula
• Inflammatory:
o Viral stomatitis
o Candida stomatitis, pharyngitis
o Peptic esophagitis (gastroesophageal reflux)
o Crohn’s Disease
o Mucositis (graft-versus-host disease)
• Systemic:
o Cardiac disease
o Pulmonary disease
Behavioral
• Poor parent-infant interaction
• Autonomy struggles
• Picky eater
• Delayed introduction of solids
Oral aversion
• Conditioned dysphagia
• Post-traumatic eating disorder
Vulnerable child
• Parental responses
Source: Adapted from Sullivan, 2016

While any of these mechanisms alone could result in feeding difficulties, rarely

can one single reason or cause be isolated or identified (Burklow et al., 1998). More

commonly, multiple factors interact and contribute to the development of feeding

difficulties (Budd et al., 1992). For example, a child’s gastroesophageal reflux might

cause esophagitis, in turn causing dysphagia. If the child is not eating, the parent might

3
exhibit stress during mealtimes that is perceived by the child, leading to adverse familial

interactions that could further deter the child from eating. In this example, the feeding

difficulty is multifactorial in etiology—dysphagia and behavioral factors are both

contributing to its development and may also be perpetuating factors.

d. Associated clinical characteristics

Several studies have investigated the clinical characteristics associated with

feeding difficulties in young children.

Rommel et al, 2003 characterized the etiologies and associated characteristics of

feeding difficulties in 700 children referred to a multidisciplinary feeding clinic for

assessment of severe feeding difficulty. 86.1% of these patients had a medical disorder,

61% had an oropharyngeal dysfunction, and 18.1% had a behavioral problem. The

majority of patients qualified for more than one of these categories, further demonstrating

the multifactorial nature of feeding difficulties. Gastrointestinal pathologies were found

to be the most common medical problems (42.45% of medical problems), with the

second most common being neurologic pathologies (11.44%). 33% of all the subjects had

gastroesophageal reflux disease (GERD), while 11.8% had a food allergy. They found

that the prevalence of medical disorders and oropharyngeal dysfunction was inversely

associated with age (with significantly higher prevalence of each condition in children

under the age of two than over the age of two), while behavioral problems were directly

associated with age (with significantly higher prevalence in children over the age of two

than under the age of two). Neurologic disorders were found to be directly associated

4
with enteral feeding, and gastrointestinal disorders were found to be directly associated

with nasogastric and parenteral nutrition and gastrostomy. Subjects with feeding

disorders had a significantly lower birthweight for gestational age when compared to the

typical prevalence of the city in which the study took place. Preterm births were similarly

overrepresented. This suggested a connection between feeding problems and intrauterine

growth retardation.

Field, Garland, & Williams, 2003 studied the clinical characteristics of 349

children, age one month to 12 years, referred to an interdisciplinary feeding program for

evaluation of a feeding difficulty. Each feeding difficulty was categorized as food refusal

(34%), selectivity by food type (21%), selectivity by food texture (26%), oral motor delay

(44%), and/or dysphagia (23%). 64% of the population had a developmental disability.

Those with Autism Spectrum Disorder (ASD) most commonly had selectivity by type or

texture. In contrast, those with Down syndrome or cerebral palsy most commonly had

food refusal, oral motor delays, and dysphagia due to the psychomotor delays which

characterize the disorders. Gastroesophageal reflux was strongly correlated with food

refusal and dysphagia among all three developmental disorders. Cardiopulmonary,

neurological, renal, and anatomic conditions were also studied. When they studied the

relationship between each of these types of conditions and the resulting feeding

difficulties, they found these types of conditions could be attributed to the presence of

gastroesophageal reflux. In fact, those without gastroesophageal reflux showed less food

refusal, oral motor problems, and dysphagia than those with gastroesophageal reflux.

Typically, up to 7% of those in the first two years of life show adverse reactions to food

5
(which often includes chronic vomiting, abdominal pain, and disinterest in eating).

However, 21% of the subjects in this population showed these adverse reactions to food.

These results suggest the possibility that gastroesophageal reflux and conditions like food

allergies can play a role in predisposing infants and children to feeding difficulties.

Benjasuwantep et al., 2013 took a slightly different approach by determining

prevalence and characteristics related to feeding difficulties among a typically

developing, healthy population rather than a referral population. They sought to compare

the feeding practices between those with and without feeding difficulties. 402 children

age one to four years participated in the study. 26.9% were found to have feeding

difficulties based on the feeding difficulty descriptions detailed by Kerzner et al., 2015.

First-born children were significantly more likely to be among those with feeding

difficulties. Those with feeding difficulties were significantly more likely to be fed less

frequently, significantly less likely to be fed at their family table, and significantly more

likely to have mealtimes longer than 30 minutes when compared to those without feeding

difficulties.

Yeung et al., 2015 determined the prevalence of food allergies in a

multidisciplinary feeding program. A retrospective chart review of 302 patients found

that 18% of the sample had a possible food allergy (reported a food reaction), 6% had a

likely food allergy (history of reaction to the food and a positive radioallergosorbent

(RAST) or skin-prick of that food), and 16% very likely had a food allergy (endoscopic

evidence of eosinophilic esophagitis or eosinophilic gastroenteritis). This is much higher

6
than the general population’s 8% food allergy prevalence. Dairy (20%), soy (9.2%), and

egg (5%) were the most common food allergies observed in the sample.

e. Assessment and treatment

Because feeding difficulties are frequently multifactorial in nature, a

multidisciplinary team is often required to effectively assess and treat each patient. These

multidisciplinary teams will often comprise a primary care pediatrician or pediatric

subspecialist, registered dietitian, psychologist, speech and language therapist,

occupational therapist and/or social worker. The team will assess the patient in their

various areas of expertise and then work with the parents to formulate a treatment plan

(Silverman, 2010).

An effective assessment will include a comprehensive clinical history and feeding

history (including a child’s feeding since birth, formula use, introduction of solids,

current diet, meal duration, mealtime behaviors, allergies and food aversions) to help

identify the specific causes of the feeding difficulty and any relevant associated

conditions. A nutrition assessment determines if the patient’s diet has sufficient caloric

intake and variety (Sullivan, 2016). Psychological assessment identifies behaviors

exhibited by the patient and the parents that may contribute to the feeding difficulty.

Clinicians will often observe a feeding episode to better assess the child’s response to

food, the interactions between the parent and child, and any problematic behaviors.

Clinicians will often recommend a combination of mealtime scheduling, meal duration,

and mealtime transition modifications (Silverman, 2015). A videofluoroscopic barium

7
swallowing study may be used to help assess a patient’s ability to swallow safely

(Sullivan, 2016). Basic laboratory evaluation, such as a complete blood count, metabolic

panel, sedimentation rate, or C-reactive protein and urine analysis, can be useful in

patients suspected of organic disease (Kerzner et al., 2015).

From a psychological perspective, feeding difficulties involving problematic

feeding behaviors often result from either classical or operant conditioning. This

distinction is clinically relevant; behaviors caused by classical conditioning, such as food

phobias, are most likely to be treated with methods like systematic desensitization, which

provides slow, gradual exposure to the feared food, and flooding, which provides rapid

and intense exposure to the feared food. Feeding behaviors resulting from operant

conditioning are dependent on the stimuli preceding the behavior and/or the responses

after the behavior. These feeding behaviors are likely best addressed by reinforcement

and/or punishment (Sullivan, 2016).

f. Prognosis and long-term effects

Dahl et al. investigated 50 infants in Sweden with feeding difficulties, and

followed up these infants at two and four years of age to better understand the prognosis

of feeding difficulties (Dahl & Kristiansson, 1987; Dahl & Sundelin, 1992). These

children met strict diagnostic criteria confirming that they experienced feeding

difficulties without any obvious medical reason during the first year of life. Infections

and behavior problems were present in significantly higher frequencies at age two years

compared to controls. 50% of the 42 participants had feeding problems that persisted at

8
two years of age. Severe persistent feeding problems at age two were significantly

correlated with recurrent infections, behavior problems, and psychosocial problems (Dahl

& Kristiansson, 1987). At age four, the 24 participants who remained in the study were

compared to 38 controls from the same health care districts. Parents of 71% of the

children with early refusal to eat reported that their child still had feeding problems.

While their heights and weights were still significantly lower than those of the controls,

these children had “caught up” and all fell within the normal ranges for height and

weight. No statistically significant difference was found in the rate of infections when

compared to the controls. 42% were reported as hyperactive, which could either be due to

a true relationship between early feeding difficulties and later hyperactivity, or the effect

of selection bias (Dahl & Sundelin, 1992).

2. Classification of feeding difficulties

Historically, feeding difficulties have been either characterized by organic

(medical), or nonorganic (behavioral and/or environmental) etiologies (Frank & Zeisel,

1988). More recently, the organic-nonorganic dichotomy has been replaced by a

biopsychosocial approach, originally proposed by Engel in 1977, in which biological and

behavioral factors interact and contribute to the development of feeding difficulties

(Bryant-Waugh et al., 2010). Given this complex, multifactorial nature of feeding

difficulties, as well as the wide range in feeding difficulty severity, researchers have

struggled to present a single unified conceptualization of feeding difficulties that is useful

in the clinical and research settings.

9
 a. A multifactorial approach

Burklow et al., 1998 developed a classification scheme for complex pediatric

feeding problems that includes five categories:

• Structural abnormalities
• Neurological conditions
• Behavioral and psychosocial issues
• Cardio-respiratory problems
• Metabolic dysfunction

These categories are not mutually exclusive, allowing feeding difficulties to be

categorized by several contributing factors. Behavioral issues, in this case, were defined

as psychosocial difficulties, negative feeding behavior shaped and maintained by

reinforcement, or emotional difficulties like phobias or depression. Their study of 103

referred patients aged 4 months to 17 years, with two thirds less than 3 years old, found

that that those with structural-neurological-behavioral etiologies made up 30% of the

sample, neurological-behavioral was 27%, behavioral alone was 12%, and structural-

behavioral was 9%. Behavioral problems were the most frequently coded category, and

were present in 85% of the subjects. This suggested that a multifactorial approach in

which biological and behavioral factors interact and contribute to feeding difficulties is

relevant, even with those with severe “organic etiologies.”

b. Relational dimension of feeding difficulties

Davies, et al., 2006 emphasized a ‘relational’ dimension to the modern

conceptualization of feeding difficulties. They proposed that primary problems, such as

medical problems, might impair a child’s ability to feed, which in turn could negatively
10
affect child-caregiver interactions during feeding, resulting in heightened caregiver

concern and anxiety. This concern and anxiety may then further impair the child’s ability

to feed. Therefore, an effective treatment plan should address the environment in which

the child is feeding, including any inappropriate caregiver behaviors, such as overly rigid

behavior with regards to the child’s growth and eating, failure to recognize satiety cues,

chaotic behavior, limitations in problem solving skills, or lack of understanding about

adequate food.

c. Mealtime behaviors and the BPFAS

Crist & Napier-Phillips, 2001 developed the Behavioral Pediatric Feeding

Assessment (BPFAS) to measure the severity of feeding difficulties through assessment

of problematic child and parent mealtime behaviors. This parent-reported measure

consists of 35 items. The first 25 items are descriptions of the child’s behavior while the

last 10 items are descriptions of the parent’s feelings and strategies regarding eating

problems. Each item consists of a description to which the parent rates how often the

behavior occurs on a five-point Likert scale (from never to always). The parent is then

asked whether or not the behavior is a problem for them by circling “yes” or “no.” Items

are phrased both positively and negatively. This measure yields four sub-scores:

• Child Frequency
• Child Problem
• Parent Frequency
• Parent Problem

11
 The Frequency scores assess the frequency of mealtime behaviors of the child or

the parent, while the Problem scores assess whether or not these behaviors are perceived

as a problem by the parent.

The BPFAS is a valid and reliable measure that has assessed mealtime behavior

problems in a range of patient populations, including typically developing children,

children referred to clinic for feeding problems (Crist & Napier-Phillips, 2001), children

with cystic fibrosis (Mitchell et al., 2004), children with type 1 diabetes mellitus (Patton,

Dolan, & Powers, 2006), and children with ADHD (Sha’ari, et al., 2016).

The BPFAS can be used to help differentiate between those without a feeding

difficulty and those with a feeding difficulty. Dovey et al., 2013 used ROC analysis to

establish statistically derived threshold values for each of the BPFAS’s sub-scores (see

Table 2). Each score was optimized for both sensitivity and specificity in discriminating

between those with and those without feeding problems. If at least one of a patient’s four

sub-scores is above the threshold score, the patient likely has a feeding difficulty. The

threshold scores were used to separate a group with feeding difficulties from a non-

clinical group with 87% accuracy in a UK population sample. (Marshall, et al., 2015)

similarly used these threshold scores to discriminate between typically developing

children and children with feeding difficulties in an Australian population sample, and

found the threshold scores displayed high specificity and high negative predictive value

(>85%).

12
 Table 2. BPFAS threshold scores for children with feeding problems.
Sub-score Threshold score
Child Frequency >61
Parent Frequency >20
Child Problem >6
Parent Problem >2
Source: Dovey et al., 2013

Crist and Napier-Philips originally used the BPFAS to compare a sample of

patients referred for feeding problems without related medical issues, a sample of patients

referred for feeding problems associated with related medical issues, and a control

sample of healthy patients. In this study, factor analysis identified five common patterns

of behavior that the clinical samples exhibited in greater frequency than the control

sample. These behavior patterns were:

• Picky eaters
• Toddler refusal—General
• Toddler refusal—Textured food
• Older children refusal—General
• Stallers

However, these specific behavior patterns only account for 55% of the cumulative

variance between the control and clinical groups, and lack further investigation by other

researchers (Kreipe & Palomaki, 2012).

d. A practical approach

Kerzner et al., 2015 combined aspects of these various approaches to

conceptualizing feeding difficulties. In their model, children are categorized under 3

13
principal feeding behaviors—limited appetite, selective intake, and fear of feeding. These

behaviors and their potential causes are summarized in Table 3.

Table 3. Classification of feeding difficulties.

Limited appetite (not eating enough)
• Misperceived:
o Excessive parental concern despite normal growth
o Misperception can be the basis of a feeding difficulty if anxious parents
adopt inappropriate feeding practices
• Energetic:
o Active children not interested in eating, also described as “infantile
anorexia”
o Often associated with conflict between parent and child
• Apathetic:
o Inactive, disinterested both in eating and other activities, poor
communication with parent
o Malnutrition is evident, and may contribute to depression and anorexia
• Organic disease:
o Structural, gastrointestinal, cardiorespiratory, neural, and/or metabolic
conditions
Selective intake (eating an inadequate variety of foods)
• Misperceived:
o Normal neophobia that begins at the end of the first year of life and peaks
between 18 to 24 months is perceived by parents as inappropriate
selectivity
• Mild selectivity:
o Tried the same number of foods as nonproblem eaters, but liked far fewer
of them
o Will grow and develop normally, but ‘picky eating’ causes family
discord centered around coercive feeding and behavioral consequences
• Highly selective:
o Children limit their diet to <10-15 foods
o Often have sensory food aversions, common in children with autism
• Organic:
o Developmental delay, dysphagia
Fear of feeding (being afraid to eat)
• Misperceived pain:
o Receiving adequate amounts of food, but cry during mealtimes
o Could be due to disordered state regulation or colic
• Infant pattern:
o Painful feeding, which over time will cause fear of feeding

14
 • Older child:
o Functional dysphagia, child chokes, gags, or vomits on food and then
ceases to eat
o Sometimes caused by parent forcefully feeding the child
• Organic:
o Any condition that could make feeding painful (esophagitis, disordered
motility, visceral hyperalgesia, tube-feeding)
Source: Adapted from Kerzner et al., 2015

The child’s history, anthropometric measurements, and physical exam are to be

used to help identify “organic red flags,” such as dysphagia or aspiration, as well as

“behavioral red flags,” such as “responsive, controlling, indulgent, or neglectful caregiver

feeding styles,” that can help the clinician identify, classify, and treat a feeding difficulty

(Kerzner et al., 2015).

e. The DSM-V & Avoidant/Restrictive Food Intake Disorder

The most recent edition of the Diagnostic Statistical Manual of Mental Disorders

(DSM-V) also offers standard diagnostic criteria for the classification of feeding and

eating disorders. The category “Feeding Disorder of Infancy or Early Childhood” in

DSM-IV-TR was replaced in DSM-V with a new diagnosis, “Avoidant/Restrictive Food

Intake Disorder.” (ARFID) The DSM-V’s criteria for ARFID diagnosis is described in

Table 4.

15
Table 4. Avoidant/Restrictive Food Intake Disorder Criteria.
Inclusion criteria Exclusion criteria
An eating or feeding disturbance (such as Eating or feeding disturbance must not be
apparent lack of interest in eating or food, better explained by:
avoidance based on the sensory • Lack of available food
characteristics of food, or concern about • An associated culturally-endorsed
aversive consequences of eating) as practice
manifested by persistent failure to meet • Another concurrent medical
appropriate nutritional and/or energy needs condition
associated with one or more of the
following: It must not occur exclusively during the
course of anorexia nervosa or bulimia
1. Significant weight loss (or failure to nervosa.
gain weight or faltering growth in
children) There must be no evidence of a body
2. Significant nutritional deficiency image disturbance.
3. Dependence on enteral feeding or
oral nutritional supplements
4. Marked interference with
psychosocial functioning
Source: Adapted from the Diagnostic and Statistical Manual of Mental Disorders, 5th
Edition, American Psychiatric Association, 2013.

ARFID was created in part because the DSM-IV-TR’s feeding and eating disorder

categories did not fully capture the range of eating disturbances typically found in clinical

practice. For example, the category “Eating Disorder Not Otherwise Specified,” intended

to include those who did not meet the full criteria for any of the other diagnoses, was in

reality used to describe the majority of treatment-seeking individuals with feeding/eating

difficulties. Additionally, the diagnosis “Feeding Disorder of Infancy or Early

Childhood” was thought to be too broad and non-specific to have significant clinical

utility (Bryant-Waugh, 2013). It was rarely used in clinical or research settings, and

limited information was available on the characteristics, course, and outcomes of children

with the diagnosis. ARFID provides detailed, inclusive criteria that are intended to
16
capture a range of presentations that were not adequately captured in the DSM-IV-TR

(Varley, 2013). ARFID also follows the DSM-V’s theme of developing a more “lifespan”

approach, giving additional consideration to how mental disorders can manifest

themselves across ages and stages of development. The new diagnosis establishes a wider

set of criteria that can be applied across ages (Bryant-Waugh, 2013).

Many clinicians are unaware of ARFID, or have not embraced it. Results from the

2014 annual Canadian Pediatric Surveillance Program (CPSP) one-time survey showed

that 63% of 664 surveyed pediatricians and pediatric subspecialties were unfamiliar with

ARFID (Katzman, Norris & Stevens, 2014). Of those who suspected a diagnosis of

ARFID, 30% inappropriately applied the exclusion criteria, resulting in a misdiagnosis.

Some authors have criticized such changes to the DSM, stating that they will

“significantly add to, not correct, the already existing problems of over-diagnosis and

over-treatment,” and that ARFID “lacks the empirical literature, seems mainly to describe

picky eaters, and may only encourage parents who already worry too much about how

their children are eating to be even more concerned” (Frances, 2013; Paris, 2013). Given

these criticisms, investigating the utility of ARFID and the characteristics of those who

meet ARFID criteria is essential.

f. Prevalence and characteristics of patients with ARFID: prior data

Because this is a new diagnosis, research concerning patients who specifically

meet ARFID criteria is limited. Some studies have examined the prevalence of ARFID

among adolescent patients referred to eating disorder programs, which has been found to

17
range from 5 to 14%, and as high as 22.5% in a pediatric day eating disorder treatment

program (Fisher et al., 2014; Nicely et al., 2014; Norris et al., 2014; Ornstein et al.,

2013). The finding that significant populations of patients are meeting ARFID criteria

suggests its possible clinical utility. These studies also found that those within eating

disorder patient populations who meet ARFID criteria are consistently more likely to be

male than those diagnosed with anorexia nervosa or bulimia nervosa, and have a high rate

of comorbid medical or psychiatric symptoms (Norris & Katzman, 2015). These studies

all used eating disorder population samples, and children or adolescents at least eight

years of age or older, limiting their generalizability to younger populations. These studies

compared the clinical characteristics of those identified as meeting ARFID criteria to

samples presenting with eating disorders such as anorexia nervosa or bulimia nervosa. As

a result, the findings cannot be generalized to a population of patients with a wider

variety of feeding problems. In the study by Nicely et al., 2014, the clinicians were not

blinded when determining which of their patients met the ARFID criteria, potentially

introducing bias into the determined ARFID prevalence rate.

Few studies have examined ARFID prevalence outside of specialty eating

disorder population samples. Kurz et al., 2015 found that 3.2% of 1,444 Swiss school

children age 8 to 13 self-reported characteristics consistent with meeting ARFID criteria.

However, these results must be interpreted with caution, as self-reported data were used

without parent-reported data or formal diagnoses by experts. Eddy et al., 2015 found that

3.9% of new referrals to 19 different Boston-area pediatric gastroenterology clinics,

ranging in age from 8 to 18 years, met the criteria for ARFID. The authors contend that

18
these relatively low prevalence rates suggest that the addition of ARFID to the DSM

might not substantially add to diagnoses of feeding disorders in pediatric populations

(Eddy et al., 2015). However, this study did not assess for use of nutritional supplements

as one of the criteria for ARFID diagnosis, meaning that the true ARFID prevalence of

the sample could be higher than they reported. Also, the results are not generalizable for

those under the age of 8.

No studies to date have examined ARFID prevalence or associated clinical

characteristics among infants and young children below the age of 8 years. Also, no

studies have investigated mealtime behaviors common among those who meet the criteria

for ARFID. Further investigation is required to better understand the population of

patients below the age of 8 who meet ARFID criteria, and to assess the clinical utility of

the ARFID diagnosis in this age group.

Dovey et al., 2016 examined whether the BPFAS could discriminate between

pediatric patients who meet ARFID criteria and a group of typically developing children

who do not meet ARFID criteria. Large significant differences were observed between

the clinical and non-clinical samples across all BPFAS subscales, suggesting that children

who meet ARFID criteria and their parents exhibit problematic feeding behaviors

significantly more than a nonclinical population. A BPFAS Child Problem sub-score > 6

retained 96.4% of the ARFID group, and removed 94% of the nonclinical group. A

BPFAS Parent Problem sub-score > 3 removed 97.3% of the nonclinical group, but also

removed much of the ARFID group, leaving only 39.3%. A BPFAS Child Frequency

sub-score >59 removed 88.7% of the nonclinical group, and retained 64.3% of the

19
ARFID group. A BPFAS Parent Frequency sub-score >22 removed 82.4% of the

nonclinical group, and retained 67.9% of the ARFID group. While the paper suggests the

BPFAS may successfully discriminate between an ARFID and nonclinical pediatric

population, the authors only used one of the four criteria, “dependence on enteral feeding

or oral nutritional supplements,” when creating the ARFID group. Inclusion of subjects

using the three other criteria for ARFID may have increased the overlap between subjects

meeting ARFID criteria and the nonclinical group, weakening the ability of the BPFAS to

distinguish these two groups. Thus, the complete ARFID criteria must be used to more

comprehensively test if the BPFAS can be used to separate those who meet ARFID

criteria from those who do not.

20
 SPECIFIC AIMS

In this study, we assessed the utility of ARFID as a clinical diagnosis for infants

and young children with feeding difficulties by 1) determining ARFID prevalence among

a population, age 9 months to 7 years, referred to a multidisciplinary feeding clinic due to

feeding difficulties; 2) investigating if there is a relationship between having a BPFAS

score above threshold and meeting ARFID criteria; 3) comparing clinical characteristics

between those who meet ARFID criteria and those who do not. The specific aims were:

1. To describe the clinical characteristics, including ARFID prevalence, of the Growth

and Nutrition Program’s patients with feeding difficulties.

2. To compare clinical characteristics, including BPFAS score, of patients who meet

ARFID criteria to those who do not.

Hypotheses:

1. 75% of subjects will meet the criteria for ARFID.

2. There will be a statistically significant relationship between meeting the criteria for

ARFID and having at least one BPFAS sub-score above threshold.

21
 METHODS

Subjects

Each of the 69 subjects in this prospective cohort study were new patients referred

to Boston Children Hospital’s Growth and Nutrition Program, a multidisciplinary

outpatient clinic in the Division of Gastroenterology and Nutrition at Boston,

Massachusetts, USA. Patients are eligible for the Growth and Nutrition Program if they

are under the age of 6 years old, and have malnutrition and/or feeding difficulties.

Subjects were included in this study if their parent or guardian completed the Growth and

Nutrition Program’s New Patient Questionnaire prior to or at the time of their first

appointment at the program. Appointment dates ranged from November 2013 to April

2016. Exclusion criteria included those younger than nine months and those older than

seven years because the BPFAS was validated in children 9 months to 7 year in age (Crist

& Napier-Phillips, 2001). For calculations involving the BPFAS or ARFID, four

additional subjects were excluded (N=65) because 2 or more items were not completed

on all of their BPFAS sub-scores. For calculations involving each of the BPFAS sub-

scores, subjects were excluded if more than 1 item was not completed for that sub-score

(Child Frequency N=56, Parent Frequency N=64, Child Problem N=41, and Parent

Problem N=41). Boston Children’s Hospital Institutional Review Board approved this

study.

22
Data collection

Parents or guardians of newly-referred patients routinely completed the Growth

and Nutrition Program’s New Patient Questionnaire in advance of, at the time of their

child’s initial Program appointment, or shortly after the appointment. The 12-page self-

administered questionnaire includes items regarding their child’s prenatal history, birth

history, medical history, feeding history from first year of life, current feeding practices,

family history, and development. The questionnaire also includes the Behavioral

Pediatric Feeding Assessment (BPFAS), a validated 35-item measure for assessing

problematic mealtime behaviors (Crist & Napier-Phillips, 2001). Additionally, we

collected anthropometric data (length/height, weight, BMI, and weight-for-length) for

each subject from the electronic medical record (PowerChart™).

All data from the questionnaire and electronic medical record was deidentified.

Questionnaire data was entered into a Research Electronic Data Capture (REDCap)

database, and data from the electronic medical record was entered into a secure Microsoft

Excel spreadsheet. The finalized REDCap database and Excel spreadsheet was imported

into Statistical Analysis System (SAS) for final data cleaning and statistical analyses.

Determining ARFID prevalence

In order to determine whether or not each subject met ARFID criteria, each

subject was assessed for failure to achieve expected weight gain, nutritional deficiency,

dependence on enteral feeding or oral nutritional supplements, and interference with

psychosocial functioning (American Psychiatric Association, 2013). A subject over the

23
age of 24 months with a BMI z-score of -2 or below, or a subject 24 months or younger

with weight-for-length z-score of -2 or below, was considered to have failed to achieve

expected weight gain/be nutritionally deficient. A subject whose caregiver indicated on a

questionnaire item regarding current use of formula feeding that the child was currently

consuming child formula/supplement or was tube fed was considered to be dependent on

enteral feeding or oral nutritional supplements. Marked interference with psychosocial

function was assessed using relevant questionnaire items—indicating “Often” in response

to any of the following meal strategies to increase the child’s intake of food or liquid:

• Feed in front of the television or electronic devices (e.g. iPad)

• Syringe or spoon feed
• Send to room for time out
• Punish

Or, indicating “4” or “5” (on a Likert scale with 1 meaning “Never,” 3 meaning

“Sometimes,” and 5 meaning “Always”) in response to any of the following items from

the BPFAS relating to psychosocial functioning:

• Tantrums at mealtimes
• I use threats to get my child to eat
• If my child does not like what is being served, I make something else
• When my child has refused to eat, I have put the food in his/her mouth by force if
necessary

Statistical analyses

SAS software was used to generate descriptive statistics used to describe the

demographic and clinical characteristics of the sample collected from the questionnaire

and BPFAS results, including proportions, means, and standard deviations. SAS was also

24
used to run t-tests and Chi-squared tests to identify the strength of any relationships

between meeting ARFID criteria and various clinical characteristics. The Cochran-

Mantel-Haenszel Chi-squared test was used for ordinal variables.

25
 RESULTS

Demographics

Of the 69 subjects included in the study, 41 (59.4%) were male, while 28 (40.6%)

were female, with the majority (65.7%) ranging from 1 to 4 years of age.

Table 5. Sample demographics (N=69).

Characteristic N (%)
Sex:
Male 41 (59.4)
Female 28 (40.6)
Age:
9 months – 12 months 8 (12.3)
1 – 4 years 44 (65.7)
4 – 7 years 13 (20.0)
Race:
American Indian/Alaskan Native 0 (0)
Asian 10 (14.5)
Black, African American 7 (10.1)
Native Hawaiian/Other Pacific Islander 0 (0)
White 51 (73.9)
Other 1 (1.5)
Unknown 2 (2.9)
Insurance:
None 0 (0)
Medicaid/Mass Health 17 (24.6)
Commonwealth Care/Health Safety Net 0 (0)
Private Insurance 39 (56.5)
Other 12 (17.4)
Unknown 1 (1.5)

Medical History

Premature birth was common, with 32.3% of the subjects being born at a

gestational age below 37 weeks. The mean birth weight was 2670 grams, and the mean

birth length was 46.4 centimeters. 31.9% of the sample experienced some problem during

childbirth, with 10.1% undergoing emergency C-section.

26
 Table 6. Birth history. (N=69)
Characteristic N (%)
Gestational age:
≥37 weeks 46 (67.7)
34-36 weeks 6 (8.8)
28-33 weeks 3 (4.4)
<28 weeks 13 (19.1)
Unknown 1 (1.5)
Mean birth weight (grams): 2670.0
Mean birth length (cm): 46.4
Problems during childbirth: 22 (31.9)
Emergency C-section 7 (10.1)

69.2 % of the sample had a medical history of at least one digestive condition, the

most common being gastroesophageal reflux (36.9% of the sample) and constipation

(33.8%). 56.9% of the sample had at least one developmental condition, such as speech

delay or gross motor delay, each of which occurred in 30.8% of the sample.

Food allergies were common with a prevalence of 20.3%. 6 subjects (8.8%) had

multiple food allergies. The most common food allergies were egg (7.3% of the sample),

milk (5.8%), and soy (5.8%). 50.7% had a first-degree relative with a food allergy. Mood

and anxiety disorders were also commonly found in the family history: 24.6% had a first-

degree relative with depression, and 26.1% had a first-degree relative with anxiety.

27
Table 7. Child medical history. (N=65)
Characteristic N (%)
At least one digestive condition: 45 (69.2)
Gastroesophageal reflux 24 (36.9)
Constipation 22 (33.8)
Diarrhea 6 (9.2)
Slow stomach emptying 4 (6.1)
Eosinophilic esophagitis 1 (1.5)
Other 7 (10.8)
Unknown 4 (6.1)
At least one developmental condition: 37 (56.9)
Speech delay 20 (30.8)
Gross motor delay 20 (30.8)
Developmental delay 5 (7.7)
Autism/PDD 4 (6.1)
Other 10 (15.2)
Unknown 4 (6.1)
At least one respiratory condition: 29 (44.6)
Apnea 5 (7.7)
CPAP therapy 5 (7.7)
Mechanical ventilation 4 (6.1)
2+ ear infections 4 (6.1)
Other 19 (29.2)
Unknown 2 (3.1)
At least one dermal condition: 27 (41.5)
Eczema 16 (24.6)
Other 6 (9.2)
Unknown 7 (10.8)
At least one neurological condition: 18 (27.7)
Hypotonia 6 (9.2)
Intraventricular hemorrhage 2 (3.1)
Cerebral palsy 1 (1.5)
Other 10 (15.4)
Unknown 5 (7.7)
At least one cardiovascular condition: 15 (23.1)
Murmur 5 (7.7)
ASD 4 (6.1)
VSD 3 (4.6)
Other 5 (7.7)
Unknown 3 (4.6)
At least one endocrine condition: 7 (10.8)
Low growth hormone 2 (3.1)
Other 5 (7.7)
Unknown 7 (10.8)

28
Current feeding behaviors

34.8% of the sample was fed formula daily, with 33.3% of the sample consuming

formula orally and 5.8% feeding via tube. 56.5% of the sample took 30 minutes or less to

finish a meal, while 36.2% took longer than 30 minutes (7.3% did not respond).

Table 8. Feeding behaviors in the past month.

Response Category
Behavior Strongly Disagree Agree Strongly
Disagree Agree
N (%)
Does not trust new foods 7 (10.6) 17 (25.8) 21 (31.8) 21 (31.8)
Will not try a food s/he 8 (12.1) 18 (27.3) 21 (31.8) 18 (27.3)
does not know
Afraid to eat things s/he 8 (12.1) 19 (28.8) 20 (30.3) 16 (24.2)
has never tried before
Will eat almost anything 38 (57.6) 12 (18.2) 11 (16.7) 3 (4.6)
Is very particular about 4 (6.2) 7 (10.8) 21 (32.3) 31 (47.7)
the foods s/he will eat
Constantly sampling new 30 (45.5) 19 (28.8) 10 (15.2) 4 (6.1)
and different foods
Refused to eat 8 (12.1) 14 (21.2) 28 (42.4) 13 (19.7)
Threw food/utensils 15 (22.7) 28 (42.4) 13 (19.7) 7 (10.6)
Only ate foods that were 19 (28.8) 26 (39.4) 12 (18.2) 6 (9.1)
certain brands or
packages
Refused foods that were 8 (12.1) 16 (24.2) 20 (30.3) 17 (25.8)
certain textures
Only ate foods that were 27 (40.9) 32 (48.5) 4 (6.1) 2 (3.0)
specific colors
Gagged or vomited when 15 (23.1) 28 (43.1) 16 (24.6) 3 (4.6)
given new foods

The sample showed a variety of problematic feeding behaviors. Nearly half

(47.7%) of the caregivers strongly agreed that their child was “very particular about the

foods s/he will eat.” 62.1% agreed or strongly agreed that their child “refused to eat,” and

56.1% agreed or strongly agreed that their child “refused foods that were certain

29
textures.” 29.2% agreed or strongly agreed that their child “gagged or vomited when

given new foods.”

The subjects’ caregivers employed a wide range of feeding strategies in order to

increase consumption of food or liquid. For example, most (60.9%) reported they often

“offered only foods my child likes,” and 30.4% reported they often “feed in front of the

television or electronic devices.”

Table 9. Feeding strategies used by caregivers in the past month.

Response Category
Strategy Never Sometimes Often
N (%)
Praise 2 (2.9) 16 (23.2) 45 (65.2)
Offer reward 22 (31.9) 24 (34.8) 20 (29.0)
Offer only foods my child likes 4 (5.8) 21 (30.4) 42 (60.9)
Offer small amounts often 10 (14.5) 27 (39.1) 30 (43.5)
Let my child snack whenever they 17 (24.6) 27 (39.1) 19 (27.5)
wants
Feed in front of television or 25 (36.2) 21 (30.4) 21 (30.4)
electronic devices
Distract with toys 33 (47.8) 24 (34.8) 10 (14.5)
Feed when falling asleep or asleep 58 (84.1) 6 (8.7) 2 (2.9)
(“dream feed”)
Syringe or spoon feed 33 (47.8) 16 (23.2) 16 (23.2)
Punish 57 (82.6) 8 (11.6) 0 (0)
Ignore 44 (63.8) 19 (27.5) 3 (4.4)

Child and parent problematic feeding behaviors were also assessed using the

BPFAS and the BPFAS threshold sub-scores. The BPFAS identified nearly all the

subjects as having a feeding difficulty, with 90.8% of the sample having at least one sub-

score above threshold. Of the four sub-scores, Parent Frequency most effectively

30
identified feeding difficulties, with 78.1% of the sample above the Parent Frequency

threshold.

Table 10. BPFAS sub-scores.

Sub-score N Threshold N (%) above Mean of Mean of
threshold scores below scores above
threshold threshold
Child Frequency 56 >61 34 (60.7) 56.4 69.2
Parent Frequency 64 >20 50 (78.1) 19.1 26.1
Child Problem 44 >6 44 (54.6) 3.0 12.5
Parent Problem 41 >2 28 (68.3) 1.3 4.8

ARFID and clinical characteristics

83.1% of the sample met criteria for ARFID, with 73.9% of the sample

demonstrating interference with psychosocial functioning, 30.8% demonstrating a

dependence on enteral or oral nutritional supplements, and 15.4% demonstrating failure

to achieve expected weight gain or nutritional deficiency.

Table 11. ARFID prevalence.

Characteristic N N (%)
Meets ARFID criteria 65 54 (83.1)
Does not meet ARFID criteria 11 (16.92)
BMI z-score ≤ -2 (if older than 24 months old) 44 6 (13.6)
WFL z-score ≤ -2 (if 24 months or younger) 21 4 (19.0)
Currently fed child formula/supplement: 65 18 (27.7)
Currently tube-fed 65 2 (3.1)
Interference with psychosocial functioning: 65 48 (73.9)

64.8% of those who met ARFID criteria were male, while 54.5% of those who did

not meet ARFID criteria were male. While the proportion of males in the ARFID group

31
was larger, the relationship between ARFID and sex was not statistically significant

(p=0.52).

No statistically significant relationship was found between meeting ARFID

criteria and having at least one BPFAS score above threshold (p=0.26). No statistically

significant relationship was found between meeting the ARFID criteria and having any of

the individual sub-scores above threshold either, though of all the sub-scores, being

above the Parent Frequency threshold appeared to be the most closely related to ARFID

(p=0.16).

Table 12. No statistically significant association between ARFID and BPFAS score
above threshold.
Meets ARFID Does not meet P-value
criteria ARFID criteria
At least 1 sub-score above threshold 50 (92.6) 9 (81.8) 0.26
No sub-score above threshold 4 (7.4) 2 (18.2)

CF above threshold 27 (57.4) 5 (62.5) 0.79

CF not above threshold 20 (42.6) 3 (37.5)

PF above threshold: 43 (82.7) 7 (63.6) 0.16

PF not above threshold 9 (17.3) 4 (36.4)

There was no statistically significant difference between the mean birth weight of

the ARFID group (2591.0 grams) and the non-ARFID group (3057.9 grams) (p=0.21).

There was also no statistically significant difference between the mean birth length of the

ARFID group (46.4 cm) and the non-ARFID group (46.6 cm) (p=0.95).

Similarly, no statistically significant relationship was found between presence of

food allergies and meeting the criteria for ARFID; of 12 patients with food allergies,

83.3% met the criteria for ARFID and 16.7% did not (p=0.98). Of the 20 subjects with a

32
first-degree relative with a food allergy, 75% met the criteria for ARFID, while 25% did

not (p=0.25), suggesting no statistically significant relationship between meeting ARFID

criteria and having a first-degree relative with a food allergy.

There was no statistically significant relationship found between meeting ARFID

criteria and meal duration (p=0.71), and any of the feeding behaviors described in Table

8, or the meal strategies described in Table 9 (excluding those items used as part of the

ARFID criteria). Results from theses analyses can be found in the Table 13 and 14.

Table 13. ARFID and meal behaviors in the last month.

Meal behavior ARFID Non-ARFID P-value
N=54 N=11
N (%)
Meal duration: 0.71
<15 min 6 (11.1) 2 (18.2)
15-30 min 23 (42.6) 4 (36.4)
>30 min 21 (38.9) 5 (45.4)
Does not trust new foods: 0.81
Strongly disagree 4 (7.8) 1 (9.1)
Disagree 13 (25.5) 2 (18.2)
Agree 17 (33.3) 4 (36.4)
Strongly agree 17 (33.3) 4 (36.4)
Will not try a food s/he does not know: 0.64
Strongly disagree 5 (9.8) 1 (9.1)
Disagree 13 (25.5) 3 (27.3)
Agree 17 (33.3) 4 (36.4)
Strongly agree 15 (29.4) 3 (27.3)
Afraid to eat things s/he has never tried 0.49
before:
Strongly disagree 5 (9.8) 2 (18.2)
Disagree 13 (25.5) 3 (27.3)
Agree 18 (35.3) 3 (27.3)
Strongly agree 13 (25.5) 3 (27.3)
Will eat almost anything: 0.61
Strongly disagree 29 (56.9) 7 (63.6)
Disagree 10 (19.6) 3 (27.3)
Agree 9 (17.6) 1 (9.1)
Strongly agree 2 (3.9) 0 (0.0)

33
Is very particular about the foods s/he will 0.49
eat:
Strongly disagree 1 (2.0) 1 (9.1)
Disagree 7 (14.0) 0 (0.0)
Agree 15 (30.0) 6 (54.5)
Strongly agree 25 (50.0) 4 (36.4)
Constantly sampling new and different 0.35
foods:
Strongly disagree 27 (52.9) 4 (36.4)
Disagree 10 (19.6) 5 (45.4)
Agree 8 (15.7) 2 (18.2)
Strongly agree 2 (3.9) 0 (0.0)
Refused to eat: 0.33
Strongly disagree 6 (11.8) 1 (9.1)
Disagree 8 (15.7) 3 (27.3)
Agree 22 (43.1) 6 (54.6)
Strongly agree 11 (21.6) 1 (9.1)
Threw food/utensils: 0.37
Strongly disagree 10 (19.6) 4 (36.4)
Disagree 18 (35.3) 6 (54.5)
Agree 13 (25.5) 1 (9.1)
Strongly agree 7 (13.7) 0 (0.0)
Only ate foods that were certain brands or 0.43
packages:
Strongly disagree 14 (27.4) 4 (36.4)
Disagree 21 (41.2) 2 (18.2)
Agree 9 (17.6) 3 (27.3)
Strongly agree 4 (7.8) 2 (18.2)
Refused foods that were certain textures: 0.30
Strongly disagree 6 (11.8) 1 (9.1)
Disagree 14 (27.4) 1 (9.1)
Agree 13 (25.5) 6 (54.5)
Strongly agree 13 (25.5) 3 (27.3)
Only ate foods that were specific colors: 0.51
Strongly disagree 21 (41.2) 3 (27.3)
Disagree 21 (41.2) 8 (72.7)
Agree 5 (9.8) 0 (0.0)
Strongly agree 2 (3.9) 0 (0.0)
Gagged or vomited when new foods were 0.68
given:
Strongly disagree 13 (26.0) 1 (9.1)
Disagree 20 (40.0) 5 (45.4)
Agree 12 (24.0) 3 (27.3)
Strongly agree 2 (4.0) 1 (9.1)

34
Table 14. ARFID and meal strategies to increase food or liquid consumption in the
last month.
Meal strategy ARFID Non-ARFID P-value
N=54 N=11
N (%)
Praise: 0.25
Never 1 (1.8) 0 (0.0)
Sometimes 12 (22.2) 2 (18.2)
Often 35 (64.8) 9 (81.8)
Offer reward: 0.36
Never 16 (29.6) 4 (36.4)
Sometimes 19 (35.2) 2 (18.2)
Often 15 (27.8) 5 (45.4)
Offer only foods my child likes: 0.47
Never 1 (1.8) 2 (18.2)
Sometimes 16 (29.6) 5 (45.4)
Often 35 (64.8) 4 (36.4)
Offer small amounts often: 0.37
Never 5 (9.3) 4 (36.4)
Sometimes 20 (37.0) 6 (54.5)
Often 26 (48.1) 1 (9.1)
Let my child snack whenever they 0.22
want:
Never 10 (18.5) 5 (45.4)
Sometimes 20 (37.0) 6 (54.5)
Often 18 (33.3) 0 (0.0)
Distract with toys: 0.37
Never 22 (40.7) 10 (90.9)
Sometimes 19 (35.2) 1 (9.1)
Often 10 (18.5) 0 (0.0)
Feed when falling asleep or asleep 0.42
(“dream feed”):
Never 44 (81.5) 10 (90.9)
Sometimes 6 (11.1) 1 (9.1)
Often 1 (1.8) 0 (0.0)
Ignore: 0.35
Never 32 (59.3) 7 (63.6)
Sometimes 15 (27.8) 4 (36.4)
Often 3 (5.6) 0 (0.0)

35
 DISCUSSION

This study sought to describe the clinical characteristics of the Growth and

Nutrition Program patient population. The finding that premature birth was common in

this sample of subjects with feeding difficulties is unsurprising, as early development of

sucking activity, and later oral motor development, depends in part on physiological

maturation (Sullivan, 2016). Similarly, the high prevalence of gastroesophageal reflux

and constipation are expected, as gastroesophageal reflux has previously been strongly

correlated with feeding difficulties such as food refusal, dysphagia, and oral motor

problems, and constipation, often resulting from poor diet, can reinforce feeding

difficulties (Field et al., 2003). The high prevalence of developmental conditions is

another factor that could contribute to the development of feeding difficulties in this

sample, as speech or motor delay could result in uncoordinated oral-motor activity. The

20.3% prevalence of food allergy corroborates the results of Yeung et al., 2015,

suggesting that food allergies are much more prevalent in populations with feeding

difficulties than in the general US population (which is about 8% as estimated by Gupta

et al., 2011). It is possible that food allergies play a role in the development of feeding

difficulties, because adverse reactions to specific foods may make it more likely that

affected children may refuse to eat or become very selective eaters. Over a third of the

sample population took longer than 30 minutes on average to finish a meal, which is in

line with previous findings that those with feeding difficulties are significantly more

36
likely to take longer than 30 minutes for a meal than those without feeding difficulties

(Benjasuwantep et al., 2013).

This study also sought to better understand the clinical characteristics associated

with ARFID in infants and young children, in order to better understand ARFID’s

possible utility for patients of those ages. This is the first study examining ARFID in

infants and children under the age of 8. The prevalence of ARFID in this sample was

83.1%, which is much higher than what was found in previous studies. This high

prevalence is not surprising, because patients in the Growth and Nutrition program are

specifically referred for evaluation of either malnutrition or feeding difficulties, but

another possible explanation is the difference in ages between our studies and previous

studies. For example, it is possible that feeding difficulties in younger children may be

more likely to result in greater interference in psychosocial functioning, as 73.9% of

these subjects met that criterion. The high ARFID prevalence rate could also be due to

the way in which we operationalized the ARFID criteria. This may particularly be the

case for the “interference in psychosocial functioning” component, given the large

proportion of the sample that met that criterion through their responses to the

questionnaire items we selected, but even if we eliminated the interference in

psychosocial functioning component, the ARFID prevalence would remain higher than

previously reported.

Interestingly, no statistically significant relationship was found between meeting

ARFID criteria and having at least one BPFAS sub-score above threshold. This contrasts

with the results of a study by Dovey et al., 2016, in which the BPFAS was used to

37
separate those who met ARFID criteria from those who did not. However, in that study,

“dependence on enteral feeding” was the only criterion used to diagnose ARFID.

Inclusion of subjects using the three other criteria for ARFID may have increased the

overlap between subjects meeting ARFID criteria and subjects not meeting ARFID

criteria, weakening the ability of the BPFAS to distinguish these two groups. Also, the

subjects with ARFID were separated from typically developing subjects in the study by

Dovey et al., 2016. In contrast, our study separated subjects with ARFID from subjects

with feeding difficulties who did not meet the criteria for ARFID. Also, we used

threshold scores that had been validated in multiple studies in different geographic

locations (Dovey et al., 2013; Marshall et al., 2015), while Dovey et al., 2016 used

slightly different threshold scores (see Background section 2f) that were calculated to

optimize sensitivity and specificity for their study’s sample. The lack of statistically

significant relationships between ARFID and the clinical characteristics shown in this

study could also be due to the small sample size and the small number of patients who

did not meet the criteria for ARFID (11 subjects).

Limitations

There are several limitations to this study. The study relied on parent-reported

data, which could cause over-reporting or underreporting of information related to the

subjects’ feeding difficulties. In additional, some parents did not complete several

questions on the BPFAS, forcing us to exclude those patients and reduce our sample size.

These exclusions raise the possibility of selection bias, because it is possible that parents

38
with children who had problematic feeding behaviors may have been more motivated to

complete the BPFAS. The relatively small number of subjects who did not meet ARFID

criteria could obscure any possible relationships that exist between ARFID and the

clinical characteristics we examined. We did not assess each patient for “faltering

growth,” a potential criterion for ARFID, as the anthropometric data collected from the

New Patient Questionnaire and electronic medical record was from only one time-point,

and the assessment of faltering growth requires data collection at multiple time points.

Conclusion and Future Directions

The current study described the demographics, medical history, and feeding

behaviors of a sample of patients referred to Boston Children Hospital’s Growth and

Nutrition Program due to feeding difficulties. It is the first study to investigate the

prevalence of ARFID in a population younger than 8 years, and the first to investigate the

clinical characteristics associated with this new diagnosis in this age group. No

statistically significant relationship was found between ARFID and an above-threshold

BPFAS score. The next iteration of this project will involve increasing the sample size

and collecting the data necessary to assess faltering growth, in order to assess the

associated clinical characteristics and utility of ARFID with greater certainty.

39
 REFERENCES

American Psychiatric Association. (2013). Diagnostic and statistical manual of mental

disorders (5th ed.). Washington, DC: Author.

Benjasuwantep, B., Chaithirayanon, S., & Eiamudomkan, M. (2013). Feeding problems

in healthy young children: prevalence, related factors and feeding practices.

Pediatric Reports, 5(2), 10. https://doi.org/10.4081/pr.2013.e10

Bryant-Waugh, R. (2013). Avoidant restrictive food intake disorder: An illustrative case

example. International Journal of Eating Disorders, 46(5), 420–423.

https://doi.org/10.1002/eat.22093

Bryant-Waugh, R., Markham, L., Kreipe, R. E., & Walsh, B. T. (2010). Feeding and

eating disorders in childhood. The International Journal of Eating Disorders,

43(2), 98–111. https://doi.org/10.1002/eat.20795

Budd, K. S., McGraw, T. E., Farbisz, R., Murphy, T. B., Hawkins, D., Heilman, N., &

Werle, M. (1992). Psychosocial Concomitants of Children’s Feeding Disorders.

Journal of Pediatric Psychology, 17(1), 81–94.

https://doi.org/10.1093/jpepsy/17.1.81

Burklow, K. A., Phelps, A. N., Schultz, J. R., McConnell, K., & Rudolph, C. (1998).

Classifying Complex Pediatric Feeding Disorders. Journal of Pediatric

Gastroenterology and Nutrition, 27(2), 143–147.

Carruth, B. R., Ziegler, P. J., Gordon, A., & Barr, S. I. (2004). Prevalence of picky eaters

among infants and toddlers and their caregivers’ decisions about offering a new

40
 food. Journal of the American Dietetic Association, 104, Supplement 1, 57–64.

https://doi.org/10.1016/j.jada.2003.10.024

Crist, W., & Napier-Phillips, A. (2001). Mealtime behaviors of young children: a

comparison of normative and clinical data. Journal of Developmental and

Behavioral Pediatrics: JDBP, 22(5), 279–286.

Dahl, M. (1987). Early feeding problems in an affluent society. III. Follow-up at two

years: natural course, health, behaviour and development. Acta Paediatrica

Scandinavica, 76(6), 872–880.

Dahl, M., & Kristiansson, B. (1987). Early feeding problems in an affluent society. IV.

Impact on growth up to two years of age. Acta Paediatrica Scandinavica, 76(6),

881–888.

Dahl, M., & Sundelin, C. (1992). Feeding problems in an affluent society. Follow-up at

four years of age in children with early refusal to eat. Acta Pædiatrica, 81(8),

575–579. https://doi.org/10.1111/j.1651-2227.1992.tb12303.x

Davies, W. H., Satter, E., Berlin, K. S., Sato, A. F., & al, et. (2006). Reconceptualizing

Feeding and Feeding Disorders in Interpersonal Context: The Case for a

Relational Disorder. Journal of Family Psychology, 20(3), 409.

Dovey, T. M., Aldridge, V. K., Martin, C. I., Wilken, M., & Meyer, C. (2016). Screening

Avoidant/Restrictive Food Intake Disorder (ARFID) in children: Outcomes from

utilitarian versus specialist psychometrics. Eating Behaviors, 23, 162–167.

https://doi.org/10.1016/j.eatbeh.2016.10.004

41
Dovey, T. M., Jordan, C., Aldridge, V. K., & Martin, C. I. (2013). Screening for feeding

disorders. Creating critical values using the behavioural pediatrics feeding

assessment scale. Appetite, 69, 108–113.

https://doi.org/10.1016/j.appet.2013.05.019

Eddy, K. T., Thomas, J. J., Hastings, E., Edkins, K., Lamont, E., Nevins, C. M., …

Becker, A. E. (2015). Prevalence of DSM-5 avoidant/restrictive food intake

disorder in a pediatric gastroenterology healthcare network. International Journal

of Eating Disorders, 48(5), 464–470. https://doi.org/10.1002/eat.22350

Field, D., Garland, M., & Williams, K. (2003). Correlates of specific childhood feeding

problems. Journal of Paediatrics and Child Health, 39(4), 299–304.

https://doi.org/10.1046/j.1440-1754.2003.00151.x

Fisher, M. M., Rosen, D. S., Ornstein, R. M., Mammel, K. A., Katzman, D. K., Rome, E.

S., … Walsh, B. T. (2014). Characteristics of Avoidant/Restrictive Food Intake

Disorder in Children and Adolescents: A “New Disorder” in DSM-5. Journal of

Adolescent Health, 55(1), 49–52. https://doi.org/10.1016/j.jadohealth.2013.11.013

Frank, D. A., & Zeisel, S. H. (1988). Failure to thrive. Pediatric Clinics of North

America, 35(6), 1187–1206.

Gupta, R. S., Springston, E. E., Warrier, M. R., Smith, B., Kumar, R., Pongracic, J., &

Holl, J. L. (2011). The Prevalence, Severity, and Distribution of Childhood Food

Allergy in the United States. Pediatrics, 128(1), e9–e17.

https://doi.org/10.1542/peds.2011-0204

42
Kerzner, B., Milano, K., MacLean, W. C., Berall, G., Stuart, S., & Chatoor, I. (2015). A

Practical Approach to Classifying and Managing Feeding Difficulties. Pediatrics,

135(2), 344–353. https://doi.org/10.1542/peds.2014-1630

Kreipe, R. E., & Palomaki, A. (2012). Beyond Picky Eating: Avoidant/Restrictive Food

Intake Disorder. Current Psychiatry Reports, 14(4), 421–431.

https://doi.org/10.1007/s11920-012-0293-8

Kurz, S., Dyck, Z. van, Dremmel, D., Munsch, S., & Hilbert, A. (2015). Early-onset

restrictive eating disturbances in primary school boys and girls. European Child

& Adolescent Psychiatry, 24(7), 779–785. https://doi.org/10.1007/s00787-014-

0622-z

Marchi, M., & Cohen, P. (1990). Early Childhood Eating Behaviors and Adolescent

Eating Disorders. Journal of the American Academy of Child & Adolescent

Psychiatry, 29(1), 112–117. https://doi.org/10.1097/00004583-199001000-00017

Marshall, J., Raatz, M., Ward, E. C., & Dodrill, P. (2015). Use of parent report to screen

for feeding difficulties in young children. Journal of Paediatrics and Child

Health, 51(3), 307–313. https://doi.org/10.1111/jpc.12729

Mitchell, M. J., Powers, S. W., Byars, K. C., Dickstein, S., & Stark, L. J. (2004). Family

functioning in young children with cystic fibrosis: observations of interactions at

mealtime. Journal of Developmental & Behavioral Pediatrics, 25(5), 335–346.

Nicely, T. A., Lane-Loney, S., Masciulli, E., Hollenbeak, C. S., & Ornstein, R. M.

(2014). Prevalence and characteristics of avoidant/restrictive food intake disorder

in a cohort of young patients in day treatment for eating disorders. Journal of

43
 Eating Disorders, Journal of Eating Disorders, 2, 2(1), 21–21.

https://doi.org/10.1186/PREACCEPT-1149759184129961, 10.1186/s40337-014-

0021-3

Norris, M. L., & Katzman, D. K. (2015). Change Is Never Easy, but It Is Possible:

Reflections on Avoidant/Restrictive Food Intake Disorder Two Years After Its

Introduction in the DSM-5. Journal of Adolescent Health, 57(1), 8–9.

https://doi.org/10.1016/j.jadohealth.2015.04.021

Norris, M. L., Robinson, A., Obeid, N., Harrison, M., Spettigue, W., & Henderson, K.

(2014). Exploring avoidant/restrictive food intake disorder in eating disordered

patients: A descriptive study. International Journal of Eating Disorders, 47(5),

495–499. https://doi.org/10.1002/eat.22217

Ornstein, R. M., Rosen, D. S., Mammel, K. A., Callahan, S. T., Forman, S., Jay, M. S., …

Walsh, B. T. (2013). Distribution of Eating Disorders in Children and

Adolescents Using the Proposed DSM-5 Criteria for Feeding and Eating

Disorders. Journal of Adolescent Health, 53(2), 303–305.

https://doi.org/10.1016/j.jadohealth.2013.03.025

PATTON, S. R., DOLAN, L. M., & POWERS, S. W. (2006). Parent report of mealtime

behaviors in young children with type 1 diabetes mellitus: Implications for better

assessment of dietary adherence problems in the clinic. Journal of Developmental

& Behavioral Pediatrics, 27(3), 202–208.

Rommel, N., De Meyer, A.-M., Feenstra, L., & Veereman-Wauters, G. (2003). The

complexity of feeding problems in 700 infants and young children presenting to a

44
 tertiary care institution. Journal of Pediatric Gastroenterology and Nutrition,

37(1), 75–84.

Sha’ari, N., Manaf, Z. A., Ahmad, M., & Rahman, F. N. A. (2016). Nutritional status and

feeding problems in pediatric attention deficit–hyperactivity disorder. Pediatrics

International, n/a-n/a. https://doi.org/10.1111/ped.13196

Silverman, A. H. (2010). Interdisciplinary Care for Feeding Problems in Children.

Nutrition in Clinical Practice, 25(2), 160–165.

https://doi.org/10.1177/0884533610361609

Silverman, A. H. (2015). Behavioral Management of Feeding Disorders of Childhood.

Annals of Nutrition & Metabolism; Basel, 66, 33–42.

https://doi.org/http://dx.doi.org/10.1159/000381375

Varley, C. K. (2013). Overview of DSM-5 changes. In North Star BHS-CME seminar.

Retrieved from https://www.omh.ny.gov/omhweb/resources/providers/dsm-5-

overview.pdf

Yeung, K. A., Taylor, T., Scheimann, A., Carvalho, R., Reinhardt, E., Girolami, P., &

Wood, R. (2015). The Prevalence of Food Allergies in Children Referred to a

Multidisciplinary Feeding Program. Clinical Pediatrics, 54(11), 1081–1086.

https://doi.org/10.1177/0009922815593499

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