Avoidant/Restrictive Food Intake Disorder Prevalence is High in Children with Gastroparesis and Functional Dyspepsia

Abstract

Background:

Avoidant/restrictive food intake disorder (ARFID) prevalence in children with gastroparesis (Gp) and/or functional dyspepsia (FD) is unknown. We aimed to identify ARFID prevalence and trajectory over two months in children with Gp, FD, and healthy children (HC) using two screening questionnaires. We also explored the frequency of a positive ARFID screen between those with/without delayed gastric emptying or abnormal fundic accommodation.

Methods:

In this prospective longitudinal study conducted at an urban tertiary care hospital, patients ages 10-17 years with Gp or FD and age- and gender-matched HC completed two validated ARFID screening tools at baseline and 2-month follow-up: the Nine Item ARFID Screen (NIAS) and the Pica, ARFID, and Rumination Disorder Interview-ARFID Questionnaire (PARDI-AR-Q). Gastric retention and fundic accommodation (for Gp and FD) were determined from gastric emptying scintigraphy.

Key Results:

At baseline, the proportion of children screening positive for ARFID on the NIAS vs PARDI-AR-Q was Gp: 48.5% vs 63.6%, FD: 66.7% vs 65.2%, HC: 15.3% vs 9.7%, respectively; P<0.0001 across groups. Of children who screened positive at baseline and participated in the follow-up, 71.9% and 53.3% were positive 2 months later (NIAS vs PARDI-AR-Q, respectively). A positive ARFID screen in Gp or FD was not related to the presence/absence of delayed gastric retention or abnormal fundic accommodation.

Conclusions & Inferences:

ARFID detected from screening questionnaires is highly prevalent among children with Gp and FD and persists for at least 2 months in a substantial proportion of children. Children with these disorders should be screened for ARFID.

Graphical Abstract

Avoidant/restrictive food intake disorder (ARFID) prevalence in children with gastroparesis (Gp) and/or functional dyspepsia (FD) is unknown. We investigated ARFID prevalence/trajectory in children with Gp, FD, and healthy children. ARFID is highly prevalent in Gp and FD and persists for at least 2 months in a substantial proportion of children.

INTRODUCTION

Gastroparesis (Gp) is defined as delayed gastric emptying of fluids or solids in the absence of a mechanical obstruction¹; symptoms include nausea, vomiting, abdominal pain, bloating, early satiety, among others, and in severe cases, nutritional deficiencies, and weight loss¹. Symptoms can overlap with those of functional dyspepsia (FD), a symptom-based diagnosis¹. Physiological abnormalities (e.g., impaired gastric accommodation) may contribute to FD symptoms².

In children and adults with Gp or FD, eating may exacerbate gastrointestinal (GI) symptoms³. Treatment may include smaller food portions, low-fat foods, and low fiber foods³. Patients may self-impose dietary restrictions, avoid certain foods, or modify foods to control symptoms⁴. If self-imposed diet restrictions/food avoidance behaviors go beyond that typical for a patient with a GI disorder, a concurrent eating disorder may be present⁵.

Avoidant/Restrictive Food Intake Disorder (ARFID) is characterized by reduced food intake (volume and/or variety) not driven by body image disturbance, but by sensory aversion to foods, lack of interest in eating, and/or fear of aversive consequences of eating. Diagnosis requires accompanying weight loss, nutritional deficiency, nutritional supplement dependence, and/or psychosocial impairment⁶.

ARFID is understudied in pediatric patients with GI disorders. Two studies, limited by retrospective chart review, reported clinically significant ARFID symptoms in up to 4% of general pediatric gastroenterology consults and up to 23% of neurogastroenterology consults^7,8. Only one ARFID study (in adults with Gp/FD symptoms) explored gastric physiology (gastric emptying)⁹. A similar study in children is lacking.

Our study’s primary aim was to prospectively determine the prevalence and characteristics of ARFID in children with Gp and FD over time vs healthy children (HC) based on two validated ARFID screening questionnaires. We also explored the potential association between delayed gastric emptying and/or impaired gastric accommodation and ARFID.

MATERIAL AND METHODS

Study Population

Pediatric patients ages 10-17 years with dyspeptic symptoms (nausea, vomiting, abdominal pain, bloating, early satiety, anorexia, and/or post-prandial fullness) without a clear etiology were enrolled when undergoing gastric emptying scintigraphy (GES) for suspected Gp. Age- and gender-matched HC without GI complaints were recruited from a healthy research volunteer database and did not undergo GES.

Participants completed questionnaires via REDCap^{™ 10, 11} at baseline and two months later. Exclusion criteria were: non-verbal, inability to read or understand English, global developmental delay, GI co-morbidities (e.g., celiac disease, etc.).

Per DSM-5, the eating disturbance in ARFID is not better explained by lack of available food or by an associated culturally sanctioned practice. Thus, we screened for food insecurity using the Hunter Vital Sign^™, a two-question validated tool¹². Furthermore, per DSM-5, ARFID cannot be diagnosed concurrently with anorexia nervosa or bulimia nervosa if the restrictive eating is driven by excessive concern about body weight or shape. Thus, we used validated cutoffs on the child version of the Eating Disorder Examination Questionnaire (ChEDE-Q8)¹³ to rule out clinically significant body shape/weight concern including attempts to restrict eating due to shape/weight concern. Children positive for either were excluded from the analysis.

The study was approved by the Baylor College of Medicine Institutional Research Board. The parent/guardian gave consent and the child assent.

Gastric Emptying Scintigraphy (GES)

Patients underwent a 4-hour GES using the recommended ^99mTc-tagged Tougas meal¹; > 60% retention at 2 hours and/or >10% at 4 hours defined delayed emptying¹. Those with delayed emptying were deemed to have Gp and those with normal emptying, FD.

Because impaired fundic accommodation potentially exacerbates symptoms¹⁴, it was calculated using intragastric meal distribution (IMD), an indirect measure of accommodation comparing the proportion of the meal in the proximal vs distal stomach¹⁴. IMD was calculated from GES results using semiautomated software (MIM Version 7.2.4, MIM Software Cleveland, OH, USA)¹⁴. Fundic content >57% immediately after meal ingestion is abnormal¹⁵.

Questionnaires

At baseline, participants (time of GES for patients) completed a demographic questionnaire and two ARFID symptom screening questionnaires (see below); the latter were completed again two months later.

Formal diagnosis of ARFID is based on DSM-5 criteria requiring a prolonged interview with a psychiatrist or psychologist. Self-report screening instruments for ARFID preliminarily validated in children were administered to all participants - the Nine Item Avoidant/Restrictive Food Intake Disorder Screen (NIAS)¹⁶ and the Pica, ARFID, Rumination Disorder Interview-Questionnaire (PARDI-AR-Q)¹⁷.

The NIAS uses three subscales assessing the three motivations behind ARFID restriction: “Picky Eating” (sensory sensitivity presentation), “Appetite” (lack of interest presentation), and “Fear” (fear of aversive consequences) (rated 0-Strongly Disagree to 5-Strongly Agree). Cutoffs for a positive NIAS ARFID screen were validated in a study that included participants ≥10 years of age (in combination with a negative screen on the Eating Disorder Examination-Questionnaire^{16, 18}). We also employed the ARFID-checklist which has been used in combination with the NIAS to align with DSM-5 criteria assessing for the presence of psychosocial impairment and/or medical consequences¹⁹. ARFID-checklist items are rated: 0-None, 1-Some, 2-Significant. Children screened positive for ARFID based on the NIAS if they met the screening cutoff and had at least one positive ARFID-checklist item.

The PARDI-AR-Q is a 32-item measure preliminarily validated in children ≥14 years¹⁷. A positive screen includes endorsement to “Do you think you have a problem with eating, involving avoidance or restriction of foods or your eating overall?” (yes/no) and presence of ≥1 of the following: 1) weight loss/failure to gain weight; 2) significant nutritional deficiency; 3) dependence of enteral feeding/nutritional supplements; 4) psychosocial impairment¹⁷. We also used the PARDI-AR-Q three subscales, “Sensory Aversion,” “Lack of Interest,” and Fear of Aversive Consequences,” each rated 0-6; higher scores indicating greater severity¹⁷.

Statistical Analysis

Numbers and percentages were used to summarize categorical data. Mean, standard deviation and normality plots were used to examine continuous data. Descriptive summaries were used to show differences among and between groups (Gp, FD, HC) on categorical demographics (gender, race and ethnicity) with the Pearson Chi-square test for independence and continuous demographics (age, weight, height, BMI Z-scores, percent gastric retention and IMD) using the Kruskal-Wallis test. For our primary aim, we calculated the number of children within each group (Gp, FD, HC) who screened positive for ARFID (by NIAS and/or PARDI-AR-Q) at baseline and 2-month follow-up. We compared the prevalence among/between groups using Chi-square tests. The relationship between NIAS and PARDI-AR-Q was assessed using the McNemar test. Agreement between NIAS and PARDI-AR-Q was compared using the Kappa coefficient (κ). For those screening positive for ARFID, symptom subscale scores were compared across groups (Gp, FD, HC) using the Kruskal-Wallis test due to the skewness of the data. If we encountered a significant difference, post-hoc comparisons were conducted between Gp and FD groups using Chi-square tests. For our exploratory aim, we compared the number of children with normal vs abnormal gastric retention and IMD in those who screened positive for ARFID using Chi-square tests. P-value < 0.05 was considered statistically significant. Fisher exact test was performed to determine any association between use of certain medications and ARFID screen. Analyses were conducted using SAS version 9.4 (SAS Institute, INC., Cary, NC).

RESULTS

A total of 206 children were recruited; 35 were excluded because of a positive food insecurity screen (Gp, n=2; FD, n=6; HC, n=17) or positive screen for non-ARFID eating disorders (by ChEDE-Q-8) (Gp, n=1; FD, n=3; HC, n=6). Demographics are shown in Table 1. Median age was 15 years (range: 10-17) with no significant difference among the groups. A smaller proportion of children identified as Black in the Gp (15%) and FD (11%) groups than in the HC (33%) (P<0.05). There were no significant differences among groups in gender, ethnicity, weight, BMI or BMI Z-scores.

Table 1:

Demographics and clinical characteristics of participants at baseline.

	Gastroparesis n=33	Functional Dyspepsia n=66	Healthy Children n=72	P-value
Age (years)	14.8 ± 1.9†	14.6 ± 2.0	14.6 ± 2.1	0.96
Gender - Female (%)	75.8	63.6	72.2	0.44
Race				0.02
Asian (%)	6.1	6.1	8.3
Black (%)	15.2	10.6	33.3
Other/mixed	6.1	12.1	12.5
White (%)	72.7	71.2	45.8
Ethnicity				0.10
Hispanic (%)	27.3	34.9	19.7
Non-Hispanic (%)	72.7	62.1	80.3
Weight (kg)	58.1 ± 20.1	56.5 ± 15.8	59.6 ± 15.4	0.39
BMI	22.3 ± 6.9	21.2 ± 5.0	22.60 ± 5.7	0.25
BMI Z-score	−0.01 ± 1.64	−0.01 ± 1.45	0.45 ± 1.09	0.15

^†mean ± SD

Positive ARFID Screen based on NIAS and PARDI-AR-Q

Prevalence at Baseline (Figure 1):

Figure 1.

Children within Gp, FD, and HC groups who screened positive for ARFID based on NIAS and the PARDI-AR-Q at baseline and at 2-month follow-up. Baseline: There was a significant difference in the proportion of children who screened positive for ARFID based on the NIAS as well as the PARDI-AR-Q among the three groups (Chi-square test, P<0.0001). Follow-up at 2 months: No differences in the proportion of children screening positive for ARFID were noted between the Gp and FD groups by either NIAS or PARDIAR-Q (P=0.08 and P=0.18, respectively); there were too few HC for comparison.

In the total cohort, the proportion of children who screened positive for ARFID was similar using the NIAS vs PARDI-AR-Q (P=1.0). There was moderate agreement for a positive ARFID screen between the NIAS and PARDI-AR-Q (κ=0.64).

There was a significant difference in the proportion of children screening positive for ARFID based on the NIAS as well as the PARDI-AR-Q among the three groups (P<0.0001). The proportion of children with a positive screen based on NIAS was greater in both Gp and FD groups vs HC (P=0.0003 and P<0.0001, respectively). Similarly, based on the PARDI-AR-Q, the proportion of children with a positive screen was greater in both the Gp and FD groups vs the HC group (P=0.01 and P<0.0001, respectively). Positive ARFID screen rates were not significantly different between the Gp and FD groups by either NIAS or PARDI-AR-Q (P=0.08 or P=0.90, respectively).

Prevalence at Two-Month Follow-up (Figure 1):

Follow-up completion rates significantly differed among groups (Gp, 18.5%, FD, 35.9%, HC, 45.6%; P=0.003). Children who completed the questionnaires at baseline and follow-up did not significantly differ in age, gender, race, or ethnicity from children who did not follow-up.

Of those who were ARFID-positive at baseline, 71.9% and 53.3% continued to screen positive at 2-month follow-up (NIAS and PARDI-AR-Q, respectively; P=0.12). At follow-up the proportion of children who were ARFID-positive based on either questionnaire did not differ significantly based on age, gender, race, or ethnicity (data not shown). There was moderate agreement for a positive screen between the NIAS and the PARDI-AR-Q (κ=0.52).

No differences in the proportion children who screened positive for ARFID were noted between the Gp and FD groups by either NIAS or PARDI-AR-Q (P=0.08 and P=0.18, respectively). The small number of children in the HC group at follow-up precluded statistical comparison of HC with Gp and FD.

A few children who did not screen positive for ARFID at baseline screened positive two months later (NIAS: Gp=4, FD=3, HC=1; PARDI-AR-Q: Gp=1, FD=2, HC=1).

NIAS and PARDI-AR-Q ARFID Symptom Subscale Scores for those who Screened Positive (Table 2)

Table 2:

NIAS and PARDI-AR-Q scores at baseline and 2 months.

NIAS
ARFID Subscales	ARFID Positive at Baseline			P-value#
	Gp (n=16)	FD (n=44)	HC (n=11)
Picky Eating	6.3 ± 3.5 †	7.9 ± 4.3	8.6 ± 3.6	0.2
Interest	12.1 ± 2.8	10.1 ± 4.3	8.7 ± 4.3	0.1
Fear	8.9 ± 4.2	9.8 ± 3.9	2.1 ± 3.4	<0.0001
ARFID Positive at 2 Months
	(n=7)	(n=15)	(n=1)
Picky Eating	6.3 ± 4.7	9.2 ± 3.8	11	0.3
Interest	12.3 ± 3.9	9.9 ± 4.3	4	0.1
Fear	8.4 ± 4.9	8.4 ± 4.4	0	0.3
PARDI-AR-Q
		ARFID Positive at Baseline		P-value*
	Gp (n=21)	FD (n=43)	HC (n=7)
Sensory Aversion	1.4 ± 1.3	1.8 ± 1.7	1.1 ± 0.9	0.6
Lack of interest	3.8 ± 1.2	3.5 ± 1.3	2.9 ± 0.9	0.2
Fear of aversive
consequences	2.6 ± 1.8	3.2 ± 1.8	0.5 ± 0.7	0.002
ARFID Positive at 2 Months
	(n=6)	(n=9)	(n=1)
Sensory Aversion	0.6 ± 1.0	2.4 ± 2.0	0	0.1
Lack of interest	3.6 ± 1.9	3.4 ± 1.0	1	0.3
Fear of aversive
consequences	2.2 ± 1.7	2.8 ± 1.8	1	0.5

Gp = gastroparesis, FD = functional dyspepsia, HC = healthy controls

^#Difference among groups; P-values were generated using Kruskal-Wallis test

^†Mean ± SD

Baseline: Scores aligning with fear of aversive consequences (NIAS-Fear and PARDI-AR-Q Fear of Aversive Consequences) were higher in Gp and FD groups vs HC (NIAS: Gp vs HC, P = 0.002; FD vs HC, P < 0.0001; PARDI-AR-Q: Gp vs HC, P = 0.008; FD vs HC, P = 0.002). Scores aligning with sensory sensitivity (NIAS-Picky Eating and PARDI-AR-Q Sensory Aversion) and with lack of interest (NIAS-Interest and PARDI-AR-Q Lack of Interest) did not differ among groups. Gp and FD groups did not differ on any ARFID subscale scores (NIAS, P = 0.53; PARDI-AR-Q, P = 0.19).

2 months: Scores for those continuing to screen positive for ARFID did not differ across the Gp, FD, and HC groups by either NIAS or PARDI-AR-Q nor between the Gp and FD groups.

Baseline

Subscale scores for fear of aversive consequences (NIAS-Fear and PARDI-AR-Q Fear of Aversive Consequences) were significantly higher in both Gp and FD groups vs HC (NIAS: Gp vs HC, P=0.002; FD vs HC, P<0.0001; PARDI-AR-Q: Gp vs HC, P=0.008; FD vs HC, P=0.002). Subscale scores for sensory sensitivity (NIAS-Picky Eating and PARDI-AR-Q Sensory Aversion) and lack of interest (NIAS-Interest and PARDI-AR-Q Lack of Interest) did not differ among groups. Gp and FD groups did not differ on any ARFID subscale scores.

Two-Month Follow-up

Subscale scores for those who continued to screen positive for ARFID did not differ across the Gp, FD, and HC groups by either NIAS or PARDI-AR-Q.

NIAS and PARDI-AR-Q ARFID Symptom Subscale Scores for those who Screened Negative (Supplementary Table 1)

Baseline

Findings generally paralleled those who screened positive for ARFID symptoms: Subscale scores for fear of aversive consequences (NIAS-Fear and PARDI-AR-Q Fear of Aversive Consequences) were significantly higher in both Gp and FD groups vs HC.

Two-Month Follow-up

In contrast to those who screened positive for ARFID symptoms at follow-up, those who screened negative had greater scores for fear of aversive consequences in the Gp and FD groups versus the HC.

Gastric Emptying and Accommodation (Table 3)

Table 3:

Number (Percent) of participants with positive ARFID screen in patients with Gp or FD based on gastric emptying and gastric accommodation results.

	Gastric Emptying			Gastric Accommodation
	Delayed (n=33)	Normal (n=66)	P-value	Abnormal (n=23)	Normal (n=76)	P-value
NIAS	16 (48.5%)	44 (66.7%)	0.1	15 (65.2%)	45 (59.2%)	0.6
PARDI-AR-Q	21 (63.6%)	43 (65.2%)	0.9	12 (52.2%)	52 (68.4%)	0.2

Mean ± SD percent gastric retention in the Gp vs FD group was: 63.2 ± 10.6 vs 41.1 ± 11.0 at 2 hours; 14.3 ± 7.4 vs 4.6 ± 2.7 at 4 hours. ARFID positive screen rates did not differ between those with delayed vs normal gastric emptying.

Mean ± SD gastric accommodation was 71.5% ± 13.6% vs 67.9% ± 13.3% in the Gp vs FD groups, respectively (P=0.21). There was a trend (P=0.05) for twice as many children in the FD group (27.1%) vs the Gp group (13.5%) to have abnormal accommodation (fundic content <57%). ARFID positive screen rates did not significantly differ between those with abnormal vs normal gastric accommodation.

Medication Use

There was no significant difference in the use of GI, psychiatric, or pain medications among the Gp, FD, and HC groups at baseline or follow-up. Furthermore, there was no difference in reported medication use (GI or psychiatric) among those who screened positive for ARFID (NIAS or PARDI-ARQ) at baseline but negative at 2-month follow-up.

DISCUSSION

In this prospective longitudinal study using two ARFID screening questionnaires, baseline ARFID prevalence in children with Gp and FD ranged from 49-65% (vs 10-15% in HC) and 50-70% continued to screen positive two months later. We found no significant difference between ARFID prevalence based on the NIAS vs the PARDI-AR-Q at baseline or follow-up. Interestingly, the proportion of those who screened positive for ARFID did not differ between those with delayed vs normal gastric emptying or abnormal vs normal gastric accommodation.

We used two validated ARFID screening questionnaires, as there is not yet a gold-standard instrument to use. There was moderate agreement in screening positive for ARFID between the NIAS and the PARDI-AR-Q, suggesting both questionnaires yielded similar results in our sample. The use of both surveys and their moderate agreement is a major strength of our study, as no prior study has validated a survey for ARFID with a pediatric GI sample. While the PARDI-AR-Q aligns with DSM-5 criteria for ARFID, the NIAS has fewer items and can be easily used in a clinical setting.

As recently reviewed, current pediatric studies on the epidemiology of ARFID are heterogenous regarding settings, sample size, and methodologies (i.e., self-report vs chart review) employed leading to a wide range of estimated prevalence²⁰. For example, our positive screening rates (49-65%) were notably higher than a previous chart review study that found up to 23% of pediatric neurogastroenterology clinic patients met full ARFID criteria or had clinically significant avoidant/restrictive eating⁸. Among adults with Gp/FD, 23% screened positive for ARFID symptoms with accompanying psychosocial/medical impairment, while the overall positive NIAS screening rate was up to 40%⁹; closer to what we now report in children.

A small proportion of HC in our study screened positive for ARFID. Screening tools tend to have high sensitivity to cast a wider initial net that can be followed up by a more specific test - in this case an in-depth evaluation by a psychologist or psychiatrist. It also may suggest overrepresentation of ARFID symptoms in Gp and FD groups by the two questionnaires. Further research is needed to rigorously identify true ARFID rates in individuals with Gp and dyspepsia using semi-structured interviews (e.g., the PARDI interview version), albeit with a greater research and patient burden.

At baseline, fear subscale scores on both the NIAS and PARDI-AR-Q were significantly higher in both our Gp and FD groups compared to HC. No significant differences were found among groups on the lack of interest or sensory sensitivity subscales. Our results align with a retrospective chart review reporting that 67% of children in a neurogastroenterology clinic with symptoms concerning for ARFID reported ‘fear of aversive consequences’ as the main motivation for restrictive eating⁸. One may speculate certain patients “overlearn” food avoidance behaviors on an elimination/exclusion diet, with fear and associated food avoidance becoming excessive and entrenched over time leading to ARFID. High scores on ARFID survey subscales assessing fear around eating may be helpful clinically to detect (and prevent) ARFID in the GI setting.

It may be difficult to distinguish between self-induced food restriction due to a GI disorder vs ARFID²¹. Because ARFID diagnosis requires the feeding disturbance not be fully explained by another physical or mental condition, the feeding disturbance must be beyond that expected to occur in the GI disorder. Hypotheses about the relationship between the development of ARFID and GI disorders range from the two developing concurrently to one increasing the risk of the other²², with some suggesting that ARFID and GI disorders may bidirectionally maintain each other²². Recognizing ARFID is particularly important in patients with Gp and FD, given that Gp/FD management often involves dietary restrictions/avoidance²³. In fact, preliminary research shows that a history of using an exclusion diet is associated with a three times greater likelihood of having ARFID symptoms²⁴. GI providers should be cautious regarding the degree and duration of food avoidance recommended.

A limitation to our study was that we could not determine the degree to which eating behaviors were solely related to GI symptom avoidance vs ARFID, since screening questionnaires were used as opposed to a formal clinical ARFID diagnosis made by a psychiatrist/psychologist. Although the response rate to the questionnaires at 2-months was approximately 43%, it is possible that those who responded were more likely to have had symptoms. Finally, delayed gastric emptying and impaired gastric accommodation available cutoffs used were from adult samples because of the lack of normative pediatric data.

Major strengths of our study include the prospective design to screen for ARFID and to our knowledge, the first use of follow-up and having HC as a comparator. Similarly, previous studies have not used the Hunger Vital Sign^™ to exclude children who might have been thought to have ARFID symptoms but whose eating behavior may have been related to food insecurity. Additionally, we employed the ChEDE-Q8 to exclude children who might appear to have ARFID but in fact, had clinically significant body image-based disordered eating behaviors. Finally, we found no evidence that medication use explained differences in ARFID prevalence at baseline vs follow-up.

CONCLUSIONS

In summary, ARFID was highly prevalent among children with Gp or FD but also was identified in a small percentage of HC. A positive ARFID screen persisted frequently after two months. Delayed gastric emptying or impaired gastric accommodation were not associated with a positive ARFID screen.