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![352 Clin Child Fam Psychol Rev (2010) 13:348–365
require variability in the baseline and/or treatment phase(s) food allergies), and feeding concerns (e.g., food selectivity;
in order to complete the calculation and/or lack conven- tube dependence). The study’s primary intervention target
tions for addressing floor or ceiling levels. Many studies in (e.g., acceptance, swallowing) was recorded, and variations
this review involved no variability during baseline and/or in operational definitions were noted.
treatment phases (e.g., no acceptance of food before Study descriptors included journal, year of publication,
intervention). Finally, standards are available for evaluat- experimental design, number of participants, and reliability
ing and easily communicating treatment effectiveness of observation. Experimental designs coded included non-
(Scruggs and Mastropieri 1998); PND scores below 50% experimental, reversal, multiple treatment reversal, multi-
represent ‘‘ineffective’’ treatments, scores between 50 and ple baselines, alternating treatments, changing criteria or
70% reflect ‘‘questionable’’ treatments, scores from 70 to some combination of these methods. Intervention data
90% are associated with ‘‘effective’’ treatments, and scores coded included type of intervention, treatment setting, and
above 90% reflect ‘‘highly effective’’ treatments. follow-up data. Treatment techniques were coded as
Non-overlap of all pairs (NAP), a recently developed involving (a) extinction-based procedures (e.g., NRS,
index of data overlap in single-case research, was selected physical guidance [PG], non-removal of food/ignoring
as a confirmatory measure of treatment outcomes (Parker disruptions), (b) reinforcement procedures (e.g., differen-
and Vannest 2009). Although less established in the liter- tial reinforcement), (c) enriched feeding environments
ature, NAP holds some advantages over PND. NAP involving non-contingent access (NCA) to preferred items/
represents a variation of an established effect size known in attention, (d) antecedent manipulations (e.g., texture, bite
various forms as area under the curve (AUC), the common size), and (e) combinations of these techniques. If rein-
language effect size (CL), and Mann–Whitney’s U, forcement was implemented, the density of the reinforce-
producing a non-parametric distribution that permits ment schedule (e.g., continuous, fixed ratio) was also
questions regarding the probability a score drawn at ran- included if available. The treatment setting (e.g., school,
dom from treatment to exceed or overlap that of a score outpatient, day treatment, inpatient) and primary therapist
drawn from baseline. Each baseline data point is compared (e.g., parent, teacher, trained therapist) were also identified
with each treatment phase data point, with the total number for each study. The unit of measurement, in terms of days,
of possible overlapping pairs (Total N) representing the weeks, and/or treatment sessions, was documented and, if
number of data points in baseline multiplied by the number conducted, the type of contact (e.g., phone, clinic visit),
of points in treatment (N baseline 9 N treatment). NAP is time frame, and stability at follow-up were recorded.
calculated by dividing the number of pairs that do not The second phase of data extraction involved converting
overlap by the total number of possible pairs. Possible raw data displayed in the primary articles to a standardized
scores range from 0 to 1 (higher scores reflecting more metric by measuring with a ruler the distance between the
effective treatments). The result yields a nomothetic effect horizontal (X) axis and the bottom of each data point in
size that can be interpreted in relation to effect sizes that millimeters. Similar data conversion procedures have been
have gained wide acceptance in large-scale group studies, shown to have a high degree of inter-rater reliability in
with formulas available for estimating Cohen’s d and R previous meta-analyses (Allison et al. 1995; Campbell
squared from NAP (Parker and Vannest 2009). 2003). Decision rules were established for selecting which
data to include in the calculation for PND and NAP.
Variables Coded, Data Extraction, and Reliability Reviewed articles varied in the number of participants,
outcomes measured and/or experimental design (e.g.,
Data were extracted from articles using a two-phase ABAB, ABAC, multi-element). When more than one par-
system. An initial screening of all articles identified through ticipant and/or feeding related behavior was included in a
the literature search was conducted to determine eligibility study and separate data points were graphically illustrated,
and extract descriptive information. Six researchers were outcomes were documented for each behavior of each
trained to collect information regarding participant demo- participant. Implemented in previous research (e.g., Herz-
graphic variables, intervention targets, study descriptors, inger and Campbell 2007), this allows all available data
and treatment techniques/protocols. Characteristics in each across participants and outcomes to be included in the
of these categories were coded using a system modeled after analysis. Because treatment effects were evaluated sepa-
previous single-case reviews (Herzinger and Campbell rately for each dependent variable across studies, this
2007) and involved a checklist system for recording vari- procedure does not inflate the impact of data from a par-
ables (available upon request from the first author). ticular study; in addition, it eliminates potential bias in
Demographic information included age, gender, develop- selecting which variable should be included in the review.
mental concerns (e.g., autism spectrum disorders, mental When a design involved multiple phases, only the first
retardation), medical issues (e.g., gastroesophageal reflux, baseline phase and the last treatment phase were included,
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![Clin Child Fam Psychol Rev (2010) 13:348–365 355
procedures aimed at enriching the feeding environment by Dependent Variables
providing access to social attention and preferred tangible
objects throughout the meal session regardless of a child’s Acceptance of food into the mouth was the most frequent
feeding behavior. Although often referred to as ‘‘non- measure of food intake (72.9% of studies), although studies
contingent reinforcement,’’ the present review uses the varied with regard to how acceptance was operationally
more precise term ‘‘non-contingent access to preferred defined (see Table 4). The definition often included a time
items’’ (NCA). Items selected for use during DRA or NCA limit for the bite to pass the lips after the initial presenta-
procedures included preferred toys and activities, as well as tion for acceptance to be scored (e.g., 5 s acceptance).
highly preferred foods. Empirical procedures for identify- Acceptance was typically presented as a percentage of total
ing highly preferred leisure items, such as paired choice bites entering the mouth during a session or meal (60.4% of
preference assessments (e.g., Fisher et al. 1992), were cited studies); less common (12.5%) were studies presenting
in 17 studies (45.9%) implementing DRA and NCA pro- frequency data (e.g., number of bites accepted; number
cedures. Less common (10.4% of studies) were punish- bites accepted per minute). To increase the consistency
ment-based procedures (e.g., response cost; time-out). In among outcome measures, studies that presented both the
addition to consequence-based procedures, antecedent number of bites accepted and bites refused per session/
manipulations, including modifying food texture, spoon meal were converted to a percentage of bites accepted if
volume, and/or number of bites per meal, were cited in these values equaled the total number of bite presented.
47.8% of studies. Forty-three studies (89.6%) incorporated Swallowing of bites was a less frequent measure of food
more than one element in a ‘‘treatment package’’. The most intake (27.1% of studies). Swallowing was typically
common packages involved EE and DRA (17 studies) or assessed by having the feeder examine the inside of the
EE, DRA and antecedent manipulations (13 studies). child’s mouth. Similar to acceptance, many definitions
Treatment settings included hospital inpatient units included an element of time, such as rapid swallowing
(43.8% of studies), followed by home/school (29.2%), day defined as swallowing before 30 s (i.e., mouth cleans).
treatment programs (16.7%), outpatient clinics (10.4%), Outcomes were most often presented as percentage of bites
and residential facilities (6.3%). While most participants swallowed per session or meal, representing 22.9% of
(60.4%) received treatment in an inpatient or day treatment studies; only two studies presenting frequency data for this
setting, there was a notable trend in terms of the setting variable. As with frequency of acceptance, data were
in which certain feeding issues were addressed. A sig- converted to percentages where possible. Finally, six
nificant proportion of children with tube (69.7%; studies presented data on the total volume of food con-
X2[3, N = 43] = 47.14, p .0001) and bottle dependence sumed measured in grams or cubic centimeters.
(87%; X2[2, N = 15] = 19.2, p .0001) were treated at The decision rules adopted for this review allowed
inpatient or day treatment facilities. In contrast, no sig- results for a single participant to contribute to more than one
nificant difference in treatment setting was detected for dependent measure. Only six studies, however, presented
children treated for food selectivity (inpatient/day treat- data on two measures of food intake (i.e., four with per-
ment: n = 8; home/school: n = 15; outpatient: n = 5; centage acceptance and swallowing; one with percentage
residential facility: n = 2). acceptance and grams and one with number of bites
Trained therapists were identified as treatment providers accepted and grams). This resulted in 14 participants
in 81.3% of studies, with fewer outcomes documented with (14.6% of the sample) contributing to more than one effect
parents or teachers serving as primary interventionist from size calculation, resulting in a total of 109 effect size esti-
the onset of the study. Length of intervention, derived from mates across the three categories of dependent measures.
the horizontal axis of treatment graphs, was most often
presented as 5 or 10 bites sessions (58.3% of studies), Treatment Outcomes
followed by meals (22.9%), and days (14.5%). Two studies
presented data in terms of weeks. The average number of The overall mean PND for all outcome measures was
sessions was 76 (SD = 45), number of meals was 76 87.95% (SD = 29.54%), with a range of 0–100% (See
(SD = 37), days in treatment was 47 (SD = 11), and Table 4). This falls in the effective range of treatment
weeks in treatment was 26 (SD = 20). Although the outcomes based on Scruggs and Mastropieri (1998) crite-
process was implied in most articles, only 58.3% of studies ria. PND scores were high across dependent variables
documented systematic training to generalize treatment (range 81.75–98.85%), with all values falling in the
gains to caregivers. Follow-up was reported in 52.1% of effective to very effective ranges. PND scores were con-
the studies, all of which reported sustained or improved sistent across measures of acceptance (Percentage Data:
feeding outcomes. Table 3 presents a detailed breakdown M = 87.87%; Frequency Data: M = 88.8%), falling in the
of the intervention characteristics by study. effective treatment range, based on established standards.
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Pediatric feeding
The document summarizes a quantitative review of treatment outcomes for pediatric feeding disorders. 48 single-case research studies involving behavioral interventions for 96 participants were included. Most children had complex medical and developmental issues and were treated at multidisciplinary feeding programs. Results indicated behavioral interventions were associated with significant improvements in feeding behavior. Common effective treatments included differential reinforcement and escape extinction procedures. However, more research is needed to document outcomes beyond behavioral changes and to identify key behavioral factors influencing mealtime performance.
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Pediatric feeding
- 1. Clin Child FamPsychol Rev (2010) 13:348–365 DOI 10.1007/s10567-010-0079-7 Pediatric Feeding Disorders: A Quantitative Synthesis of Treatment Outcomes William G. Sharp • David L. Jaquess • Jane F. Morton • Caitlin V. Herzinger Published online: 16 September 2010 Ó Springer Science+Business Media, LLC 2010 Abstract A systematic review of the literature regarding Keywords Autism spectrum disorders Á Behavioral treatment of pediatric feeding disorders was conducted. intervention Á Evidence-based treatment Á Failure to thrive Á Articles in peer-reviewed scientific journals (1970–2010) Feeding Á Feeding disorders Á Mealtime problems evaluating treatment of severe food refusal or selectivity were identified. Studies demonstrating strict experimental control were selected and analyzed. Forty-eight single-case Introduction research studies reporting outcomes for 96 participants were included in the review. Most children presented with Eating is an essential human activity, necessary to sustain complex medical and developmental concerns and were life and ensure growth, but it also is a common challenge treated at multidisciplinary feeding disorders programs. for children and a source of stress for caregivers. Up to All studies involved behavioral intervention; no well- 40% of toddlers and early school-age children experience controlled studies evaluating feeding interventions by other some mealtime difficulties (Manikam and Perman 2000; theoretical perspectives or clinical disciplines met inclu- Mayes and Volkmar 1993). Issues include ‘‘picky’’ eating sion criteria. Results indicated that behavioral intervention patterns, strong food preferences, behaviors aimed at end- was associated with significant improvements in feeding ing meals prematurely (e.g., whining, crying, pushing food behavior. Clinical and research implications are discussed, away), and/or fluctuating hunger (Reau et al. 1996). Mild including movement toward the identification of key difficulties typically resolve spontaneously or with low behavioral antecedents and consequences that promote intensity interventions, such as caregiver education in food appropriate mealtime performance, as well as the need to preparation/presentation, and/or nutritional guidance better document outcomes beyond behavioral improve- (Greer et al. 2009; Kerwin 1999). Between 3 and 10% of ments, such as changes in anthropometric parameters, children, however, develop chronic feeding issues generalization of treatment gains to caregivers, and exceeding ordinary developmental variation and possibly improvements in nutritional status. associated with a number of negative medical and devel- opmental outcomes (Kerwin 1999). These include growth retardation, malnutrition, developmental and psychological deficits, poor academic achievement, social difficulties, invasive medical procedures (e.g., placement of a feeding W. G. Sharp (&) Á D. L. Jaquess Á J. F. Morton Á tube), or death (Benoit 1993; Chatoor 2002; Finney 1986). C. V. Herzinger Feeding problems of this magnitude are characterized as Marcus Autism Center, Atlanta, GA, USA ‘‘feeding disorders’’ due to their chronic and more severe e-mail: william.sharp@choa.org course, often involving the complex interplay among D. L. Jaquess biological, psychological, and social factors and requiring e-mail: David.Jaquess@choa.org intensive intervention to avoid long-term medical and W. G. Sharp Á D. L. Jaquess Á J. F. Morton developmental sequelae (Babbitt et al. 1994; Lindberg Emory University School of Medicine, Atlanta, GA, USA et al. 1991; Sanders et al. 1993). 123
- 2. Clin Child FamPsychol Rev (2010) 13:348–365 349 The process of assessing and treating severe feeding Significant feeding disturbances have also been reported disorders is complicated by a number of interrelated factors. among children with no clear physiological precursor or The psychiatric diagnosis of ‘‘Feeding Disorder of Infancy developmental issues and may continue in children whose or Early Childhood’’ is non-specific, encompassing children organic issues are resolved. Causal factors in these cases are who fail to eat a sufficient quantity and/or variety of food believed to include disrupted family functioning and mal- resulting in chronic malnutrition, poor weight gain and/or adaptive patterns of reinforcement (Babbitt et al. 1994). weight loss before age 6 years in the absence of an active Potentially problematic feeding practices include lack of organic complaint (American Psychiatric Association structure conducive to eating (e.g., unrestrained access to 2000). The medical diagnosis for these disorders ‘‘Feeding food; irregular mealtimes), exposure to developmentally Difficulties and Mismanagement’’ is similarly broad, inappropriate textures, and/or parental modeling of inap- (World Health Organization 2009). Children meeting these propriate eating habits (Sanders et al. 1993). Many long- criteria represent a heterogeneous group with numerous standing feeding problems involve learned behaviors whose etiological pathways. A variety of organic factors that lead function is to escape unpleasant feeding experiences and/or to difficult or painful eating may precipitate or play a role in gain attention from caregivers (Piazza, Fisher et al. 2003). the development of feeding concerns. These include (1) Behavioral mismanagement in the form of positive rein- metabolic abnormalities or defects in absorption that forcement (e.g., caregiver attention for inappropriate accompany conditions such as cystic fibrosis, mitochondrial behaviors) and negative reinforcement (e.g., removing food disease, short bowel syndrome, or lactose intolerance; (2) and/or ending meals due to problem behaviors) may inad- gastrointestinal issues involving persistent emesis and/or vertently shape and strengthen problem behaviors. When a diarrhea (e.g., gastroesophageal reflux, gastroenteritis, caregiver inadvertently reinforces problem behaviors, those dysmotility), (3) structural or anatomical defects (e..g, behaviors tend to become more frequent or intense, which, in bronchopulmonary dysplasia, malrotated intestine, micro- turn, may lead to greater efforts to manage problem behav- gnathia), (4) oral motor deficits (dysphagia), and 5) hyper- iors. The resulting coercive cycle often terminates only after sensitivity to food tastes, smells, and textures (Arvedson the child or caregiver withdraws from the feeding situation or 2008; Babbitt et al. 1994; Sanders et al. 1993). Children stops responding altogether. At the familial and inter-sys- with no known organic factors, however, also develop temic levels of analysis, critical developmental experiences severe feeding problems, suggesting that additional causal are circumvented or severely disrupted (Davies et al. 2006). factors result in maladaptive feeding patterns. Without direct intervention, this pattern is likely to increase Children with developmental disabilities are also at high in frequency and severity over time (Lindberg et al. 1991). risk for developing feeding disorders (Babbitt et al. 1994). Given this breadth of diagnostic inclusion and possible Approximately one-third of all children with develop- etiological pathways, feeding disorders often include more mental disabilities experience a clinically significant than one causal factor and involve a wide range of topog- feeding concern (Dahl and Sunderlin 1986; Palmer and raphies. Typical consumption involves a number of suc- Horn 1978; Palmer et al. 1975). Common issues include cessive steps: bringing a bite to the lips, accepting food into lack of independent self-feeding skills, disruptive mealtime the mouth, chewing and forming a bolus, and swallowing behaviors, and/or limited intake related to food selectivity (Gulotta et al. 2005). When this process is interrupted, (Munk and Repp 1994; Sisson and Van Hasselt 1989). problems may arise at different points along this chain of Prevalence estimates have been reported to be much higher consumption, which further complicates the diagnostic and among certain subgroups. For example, up to 89% of intervention picture (Riordan et al. 1980; Sevin et al. 2002). children with autism spectrum disorders (ASD) display For example, some children display disruptive behaviors strong preferences for certain foods (by type, texture, color, (e.g., head turning, batting at the spoon) that interfere with or packaging), consume a narrower range and quantity of accepting a bite into the mouth, while other children fail to food when compared with peers, and/or display elevated consume an adequate volume of food due to packing or rates of disruptive behavior when presented with non-pre- expelling bites. In addition, treatment resolving refusal at ferred food (Ahearn et al. 2001; Bowers 2002; Collins, one point along the chain of consumption (e.g., acceptance) et al. 2003; Cornish 1998, 2002; Field et al. 2003; Schreck can lead to a collateral increase in topographies of food et al. 2004). In past reports, the emergence and mainte- refusal further down the chain (e.g., expulsions, packing) nance of severe feeding problems in ASD often has no after a child is accepting bites without difficulty (Gulotta identifiable organic factors or gastrointestinal etiology, et al. 2005). This presents a unique challenge for caregivers leading to the hypothesis that aberrant feeding habits and professionals addressing feeding concerns, with the among those with ASD may be a manifestation of goal of an intervention often shifting over time. restricted interests, behavioral rigidity, and/or persevera- The multifaceted and mercurial nature of severe feeding tion (Ledford and Gast 2006). problems combined with their complex biopsychosocial 123
- 3. 350 Clin Child Fam Psychol Rev (2010) 13:348–365 etiology intensifies the need to identify evidence-based elements included DRA, EE, and stimulus fading. While treatments. Although numerous researchers have docu- providing an updated survey the literature, Williams and mented treatment outcomes for feeding disorders, few colleagues did not screen the studies in terms of method- attempts have been made to summarize or evaluate this body ological rigor or experimental control. of evidence (see Kerwin 1999; Ledford and Gast 2006; Kerwin’s (1999) work and subsequent reviews provided Williams et al. 2010). Kerwin conducted the first compre- an important springboard for research focusing on the spe- hensive literature review in this area, summarizing studies cific etiological factors associated with feeding problems published between 1970 and 1997. Twenty-nine studies were and treatments effecting their remediation, while also laying identified as meeting the methodological criteria of the Task the groundwork for an updated quantitative review of the Force on Promotion and Dissemination of Psychological literature. The past decade has seen a significant increase in Procedures (1995) of the American Psychological Associa- the number of studies focusing on the analysis and treatment tion, all of which involved behavioral intervention. Differ- of severe feeding disorders, yielding important data ential reinforcement (DRA) contingent upon appropriate regarding key treatment elements and outcomes. In addition, eating behaviors, ignoring inappropriate response, and statistical procedures for estimating and combining the size physically guiding appropriate feeding responses were of treatment outcomes for both group and single-case studies identified as effective interventions. DRA of acceptance in have also been developed and refined (Busk and Serlin combination with escape extinction (EE) procedures target- 1992). With these advances in place, the current review ing avoidance of food in the form of non-removal of the spoon seeks to: (1) survey the medical, habilitative, and psycho- (NRS, e.g., Ahearn et al. 1996a, b) or swallow elicitation logical literature, focusing on identifying studies using strict (e.g., Hagopian et al. 1996) was identified as a promising methodological rigor and experimental control to investigate intervention. Kerwin acknowledged that non-behavioral interventions aimed at improving intake among children interventions may be effective in treating feeding problems with severe feeding disorders; (2) determine the overall but noted the need for well-controlled studies by other theo- effect size of identified treatments using statistical proce- retical perspectives or disciplines. In addition, the review dures for synthesizing outcome data; and (3) describe the highlighted the need to investigate the setting in which treatment elements, population, and settings associated with evidence-based treatments are developed and evaluated. significant improvements in feeding patterns. Subsequent less comprehensive literature reviews by Ledford and Gast (2006) and Williams et al. (2010) added support for the effectiveness of behavioral intervention to Method address chronic feeding concerns. Ledford and Gast focused specifically on the treatment of children with ASD Study Identification and Eligibility Criteria and feeding difficulties, reviewing studies with experi- mental control published between 1994 and 2000. Nine Studies investigating the treatment of pediatric feeding single-case design studies were identified, all involving one disorders were identified through searches of the MedLine or more behavioral elements to address severe food and PsychINFO databases. The search parameters included selectivity. In each case, the use of behavioral intervention combinations of the following key words: feeding, food was associated with significant improvements in the variety refusal, feeding disorder, pediatric feeding disorders, and/or quantity of food consumed. Strategies included treatment, intervention, behavioral, psychosocial, family DRA, simultaneous and/or sequential presentation of pre- therapy, psychodynamic, pediatric dysphagia, oral motor, ferred and non-preferred food, EE, and stimulus fading and nutrition. These search terms were expressly selected in (Ahearn 2003; Najdowski et al. 2003; Piazza et al. 2002). order to capture all experiments demonstrating efficacy of More recently, Williams et al. (2010) identified 38 inter- treatments across possible disciplines or theoretical for- vention studies (published between 1979 and 2008) tar- mulations providing feeding therapy. In addition, references geting children with food refusal, defined as refusing to eat in identified articles were evaluated for possible inclusion. all or most foods resulting in a failure to meet caloric needs The central inclusion criterion for the review was the or reliance on supplemental tube feedings. In all 38 studies, use of an experimental design to investigate treatment improvements in oral intake were reported, with more than outcomes, including the use of a control group with group half of the children who received some form of supple- designs or experimental single-case research methodology mental tube feeds being described as weaned from these (e.g., changing criterion, reversal, alternating treatments, feedings. Treatments were primarily multidisciplinary and multiple baseline). This excluded group designs without involved one or more behavioral interventions incorporated randomization to a control condition and single-case into larger treatment packages at inpatient (57%) or day studies using simple AB designs if no additional experi- treatment (24%) feeding programs. Common treatment mental elements were incorporated into the study to control 123
- 4. Clin Child FamPsychol Rev (2010) 13:348–365 351 for alternative hypotheses. In addition, studies needed to was empirically measured and graphically illustrated with meet the following criteria: clearly identifiable baseline and treatment phases for each participant. Repeated data points, not mean scores or 1. The article was published in an English language peer- trends/lines, had to be reported; (b) Reliability data (e.g., reviewed journal between January 1970 and June interobserver agreement) was provided in the article and 2010. reached at least 80% for each dependent measure; and (c) If 2. The study evaluated the effects of an intervention an article included multiple participants or studies, only aimed at treating children (birth to 18 years of age) partially meeting inclusion criteria, only those participants presenting with severe feeding disorders characterized or components that met criteria were included in the by chronic food refusal, tube/bottle dependence, food review. selectivity, and/or poor oral intake. 3. The intervention aimed at improving solid food intake, not liquids. Studies focusing only on reducing problem Quantifying Treatment Outcomes of Feeding behaviors (e.g., expulsions; packing) or analyzing the Interventions function of refusal behaviors, including descriptive and functional analysis, were excluded if this was the sole A quantitative synthesis of findings from single-case purpose of the study, and no data on intake during research relies on the availability of graphs published in treatment were presented. Studies evaluating the articles and involves quantifying data points for the anal- impact of antecedent manipulations (e.g., food texture; ysis, rather than relying solely on visual inspection to presentation methods) on refusal behaviors were also determine treatment effectiveness (Busk and Serlin 1992). excluded if the analysis did not focus on changes in Several commonly used metrics for quantifying treatment intake from baseline levels. outcomes include mean baseline reduction (MBLR), stan- 4. The dependent variable(s) was a measure of food dard mean difference (SMD), percentage of non-overlap- intake (e.g., acceptance; swallowing; grams). ping data (PND), and/or percentage of zero data (PZD; see 5. Children meeting the Diagnostic and Statistical Man- Campbell 2003 for review). These metrics provide overall ual-IV (DSM-IV-TR; American Psychiatric Associa- estimates of treatment effectiveness but are not considered tion 2000) criteria for anorexia nervosa, bulimia traditional effect size measures because the relative nervosa, binge-eating disorder, or eating disorder not standing of the average treatment point within a population otherwise specified were excluded from the review. distribution is not reported (Herzinger and Campbell 2007). Articles describing children with rumination, pica, Regression-based approaches, such as d (Cohen 1988), vomiting, rapid and/or messy eating, poor table have also been developed. Recent studies indicate that manners, lack of utensil use, and/or lack of self- MBLR, SMD, PND, PZD, and regression-based measures feeding skills were excluded from the review unless are comparable in detecting treatment effects in single-case these behaviors interfered with appropriate nutritional meta-analysis (Campbell 2004). intake and/or promoted tube dependence. PND was selected as the non-regression metric in this Studies involving both group and single-case designs analysis. This statistic involves determining the percentage were initially considered for inclusion in the meta-analysis; of treatment data not overlapping with baseline data. Cal- however, since only three studies with group design culations involved dividing the number of treatment data involved experimental control (i.e., Benoit et al. 2000; points exceeding the highest baseline data point by the total Stark et al. 1996, Turner et al. 1994), those studies were number of data points in the treatment phase and multi- reviewed separately. The present meta-analysis focused on plying this value by 100 (Scruggs et al. 1987). Possible combining findings from studies involving single-case scores range from 0 to 100%, with higher scores reflecting designs. Other group studies consisted of program evalu- more effective treatments. To address the influences of ations that lacked experimental control or involved a single outliers, the stringent conventions set forth by Scruggs demonstration of positive outcomes (e.g., Berger-Gross et al. were adopted, with a PND score recorded as zero et al. 2004; Greer et al. 2009; Kindermann et al. 2008; when a single baseline data point reached ceiling level on Williams et al. 2007). Outcomes from group studies will be the dependent variable of interest. discussed below in relation to the results of the meta- A number of considerations guided the selection of analysis. PND. The primary focus was to quantify results of treat- In order to provide data appropriate for single-case ments aimed at increasing appropriate intake of food, meta-analytic procedures (described below), three addi- which eliminated measures designed to quantify results of tional criteria were used to select single-case articles for studies involving behavioral reduction (e.g., PZD, MBLR). inclusion in the analysis: (a) The effect of the intervention In addition, some effect size measures (e.g., MBLR, SMD) 123
- 5. 352 Clin Child Fam Psychol Rev (2010) 13:348–365 require variability in the baseline and/or treatment phase(s) food allergies), and feeding concerns (e.g., food selectivity; in order to complete the calculation and/or lack conven- tube dependence). The study’s primary intervention target tions for addressing floor or ceiling levels. Many studies in (e.g., acceptance, swallowing) was recorded, and variations this review involved no variability during baseline and/or in operational definitions were noted. treatment phases (e.g., no acceptance of food before Study descriptors included journal, year of publication, intervention). Finally, standards are available for evaluat- experimental design, number of participants, and reliability ing and easily communicating treatment effectiveness of observation. Experimental designs coded included non- (Scruggs and Mastropieri 1998); PND scores below 50% experimental, reversal, multiple treatment reversal, multi- represent ‘‘ineffective’’ treatments, scores between 50 and ple baselines, alternating treatments, changing criteria or 70% reflect ‘‘questionable’’ treatments, scores from 70 to some combination of these methods. Intervention data 90% are associated with ‘‘effective’’ treatments, and scores coded included type of intervention, treatment setting, and above 90% reflect ‘‘highly effective’’ treatments. follow-up data. Treatment techniques were coded as Non-overlap of all pairs (NAP), a recently developed involving (a) extinction-based procedures (e.g., NRS, index of data overlap in single-case research, was selected physical guidance [PG], non-removal of food/ignoring as a confirmatory measure of treatment outcomes (Parker disruptions), (b) reinforcement procedures (e.g., differen- and Vannest 2009). Although less established in the liter- tial reinforcement), (c) enriched feeding environments ature, NAP holds some advantages over PND. NAP involving non-contingent access (NCA) to preferred items/ represents a variation of an established effect size known in attention, (d) antecedent manipulations (e.g., texture, bite various forms as area under the curve (AUC), the common size), and (e) combinations of these techniques. If rein- language effect size (CL), and Mann–Whitney’s U, forcement was implemented, the density of the reinforce- producing a non-parametric distribution that permits ment schedule (e.g., continuous, fixed ratio) was also questions regarding the probability a score drawn at ran- included if available. The treatment setting (e.g., school, dom from treatment to exceed or overlap that of a score outpatient, day treatment, inpatient) and primary therapist drawn from baseline. Each baseline data point is compared (e.g., parent, teacher, trained therapist) were also identified with each treatment phase data point, with the total number for each study. The unit of measurement, in terms of days, of possible overlapping pairs (Total N) representing the weeks, and/or treatment sessions, was documented and, if number of data points in baseline multiplied by the number conducted, the type of contact (e.g., phone, clinic visit), of points in treatment (N baseline 9 N treatment). NAP is time frame, and stability at follow-up were recorded. calculated by dividing the number of pairs that do not The second phase of data extraction involved converting overlap by the total number of possible pairs. Possible raw data displayed in the primary articles to a standardized scores range from 0 to 1 (higher scores reflecting more metric by measuring with a ruler the distance between the effective treatments). The result yields a nomothetic effect horizontal (X) axis and the bottom of each data point in size that can be interpreted in relation to effect sizes that millimeters. Similar data conversion procedures have been have gained wide acceptance in large-scale group studies, shown to have a high degree of inter-rater reliability in with formulas available for estimating Cohen’s d and R previous meta-analyses (Allison et al. 1995; Campbell squared from NAP (Parker and Vannest 2009). 2003). Decision rules were established for selecting which data to include in the calculation for PND and NAP. Variables Coded, Data Extraction, and Reliability Reviewed articles varied in the number of participants, outcomes measured and/or experimental design (e.g., Data were extracted from articles using a two-phase ABAB, ABAC, multi-element). When more than one par- system. An initial screening of all articles identified through ticipant and/or feeding related behavior was included in a the literature search was conducted to determine eligibility study and separate data points were graphically illustrated, and extract descriptive information. Six researchers were outcomes were documented for each behavior of each trained to collect information regarding participant demo- participant. Implemented in previous research (e.g., Herz- graphic variables, intervention targets, study descriptors, inger and Campbell 2007), this allows all available data and treatment techniques/protocols. Characteristics in each across participants and outcomes to be included in the of these categories were coded using a system modeled after analysis. Because treatment effects were evaluated sepa- previous single-case reviews (Herzinger and Campbell rately for each dependent variable across studies, this 2007) and involved a checklist system for recording vari- procedure does not inflate the impact of data from a par- ables (available upon request from the first author). ticular study; in addition, it eliminates potential bias in Demographic information included age, gender, develop- selecting which variable should be included in the review. mental concerns (e.g., autism spectrum disorders, mental When a design involved multiple phases, only the first retardation), medical issues (e.g., gastroesophageal reflux, baseline phase and the last treatment phase were included, 123
- 6. Clin Child FamPsychol Rev (2010) 13:348–365 353 as recommended by Faith et al. (1996) and applied in Table 1 Description of studies and experimental characteristics similar review studies (e.g., Campbell 2003). In studies Characteristic n % involving multi-element designs conducted across baseline and treatment phases, a single effect size was calculated Journal title only if both treatment paths were presented in the baseline Journal of Applied Behavior Analysis 25 52.1 and final treatment phases. This allowed analysis of overall Behavior Modification 7 14.6 treatment effect rather than breaking down individual Behavioral Interventions 3 6.3 treatment elements. PND and NAP were calculated using Journal of Behavioral Therapy and Experimental Psychiatry 2 4.2 all data points in the first baseline and last treatment pha- Education and Treatment of Children 2 4.2 ses, allowing for common outcome metrics to be generated American Journal of Mental Retardation 1 2.1 for all studies. Applied Research in Mental Retardation 1 2.1 Twenty-seven percent of the articles (n = 13) in this Childcare, Health, and Development 1 2.1 analysis were randomly selected for independent coding by Focus on Autism and Other Developmental Disabilities 1 2.1 two trained staff to calculate inter-rater reliability. These Journal of Clinical Child Psychology 1 2.1 articles involved 23 different participants (24% of all par- Journal of Behavioral Education 1 2.1 ticipants) contributing data for 32 separate dependent Journal of Developmental and Physical Disabilities 1 2.1 variables (29% of all outcomes). For descriptive informa- Journal of Intellectual Disability Research 1 2.1 tion extracted during the review process, inter-rater Journal of Positive Behavior Interventions 1 2.1 agreement was calculated through the percent agreement Total studies 48 Number of participants contributed per study method: # agreements/(# agreements ? # disagree- 1 25 52.1 ments) 9 100, as well as the Kappa statistic. The mean 2 7 14.5 inter-rater agreement across all variables was 94.1% (range 3 8 16.7 87.5–100%) with a corresponding Kappa of .8 (range .7 to 4 7 14.6 .99). For quantitative information extracted via ruler, reli- 5 1 2.1 ability was calculated on all individual data points using Total participants 96 Spearman’s q. The overall inter-rater reliability for quan- Study breakdown (n = 48) titative data was q = .942. Inter-rater agreement for both Year published qualitative and quantitative exceeded the 80% acceptable 2000–2010 29 60.4 standard of agreement widely adopted and recommended 1990–1999 13 27.1 during quantitative synthesis of single-case research (e.g., 1980–1989 6 12.5 Campbell 2003). To further ensure the accuracy, the first Primary experimental design author conducted a second review of all articles included in Reversal 20 41.7 the study, focusing on potential areas of discrepancy Multiple baseline 11 22.9 highlighted by the inter-rater analysis and consensus with Changing criterion 7 14.6 the second author was reached in cases of ambiguity. Multielement and reversal 5 10.4 Multielement 2 4.2 Multielement and multiple baseline 2 4.2 Results Multiple baseline and reversal 1 2.0 Unit of measurement reported for data collection Characteristics of Studies and Participants Sessions 28 58.3 Meals 11 22.9 The search yielded 48 studies meeting inclusion criteria out Days 7 14.6 of a pool of 124 possible articles, resulting in 96 partici- Weeks 2 4.2 pants included in the summary. Table 1 presents descrip- Inter-rater reliability of observations by study: M = 96.7; tive and experimental characteristics of the identified SD = 3.5; range = 85–100 studies. More than half of the studies were published after % Calculated based on total sample n = 82 2000, indicating a recent increase in studies employing a high degree of experimental control to investigate the treatment of severe feeding problems. Interestingly, only 9 of the 29 studies identified by Kerwin (1999) were included liquid intake (n = 1), did not present individual data points in the present review. The other 20 studies did not meet the and/or reliability data (n = 6), or analyzed treatment out- present inclusion criteria because they focused on inde- comes using a group design (n = 3). Studies meeting pendent feeding skills or healthy eating habits (n = 11) or inclusion criteria were published in 14 journals, with the 123
- 7. 354 Clin Child Fam Psychol Rev (2010) 13:348–365 Journal of Applied Behavior Analysis contributing the Feeding tube dependence was the most prevalent feeding largest number of articles (52.1%). All studies recorded concern (44.8% of participants), followed by food selec- discreet behaviors through direct observation. Inter-rater tivity (31.3%), bottle/liquid dependence (15.6%), and poor reliability of overall observations exceeded the inclusion oral intake (8.3%). Only a small subgroup of participants criterion of 80% agreement, with an average of 96.7% (10.5%) were described as ‘‘typically developing’’, with across all studies. most cases (65.6%) identified as having a developmental Participant characteristics are presented in Table 2. The issue in addition to a feeding disorder. Developmental mean age of participants was 4 years; however, the sample concerns were most often described as global develop- captured a wide age range (10 months to 14 years). mental delays (31.2%), followed by, ASD (23.7%), intel- lectual disability (21.5%), and language/speech issues Table 2 Description of participants (9.7%). Consistent with the literature to date, 90.9% of children with ASD (20 of 22 cases) presented with food Characteristic n % selectivity rather than food refusal, X2(1, N = 22) = 14.7, Age (in months) M = 48.06; SD = 30.47; p .0001, representing the majority (67%) of participants range 10–168 identified as food selective. Medical concerns were com- Gender mon, with 67.7% of the sample having at least one reported Male 62 64.6 medical concern. Forty of the sixty-five children (61.5%) Female 34 35.4 presented with multiple medical issues, suggesting severe Total 96 feeding problems often co-occur with complex medical Feeding concerns histories. A significant number of children with feeding Feeding tube 43 44.8 tube dependence (42 out of 43) had one or more medical Food selectivity 30 31.3 issues, X2(1, N = 43) = 36.1, p .0001. In contrast, only Bottle/liquid dependence 15 15.6 5 of the 22 children with ASD also had medical issues Poor oral intake 8 8.3 reported. Developmental issues Reported 63 65.6 Intervention Characteristics Not reported 23 23.9 ‘‘Typically developing’’ 10 10.5 All of the studies meeting inclusion criteria emphasized Breakdown of developmental issues a behavioral interventions. While a few articles described Developmental delay 29 31.2 conceptualization or treatment approaches through family Autism spectrum disorder 22 23.7 therapy, psychodynamic, sensory therapy or oral motor Mental retardation 20 21.5 therapy, no actual outcomes of treatment effectiveness Speech/language delay 9 9.7 were included in these articles. A few articles described Other 4 4.3 medication interventions in pre- post-treatment studies for small groups of patients; however, none of them included a Medical issues control group. Among the behavioral interventions Reported 65 67.7 reviewed, EE was the most widely applied intervention, Not reported 31 32.3 with 83.3% of the treatments involving some form of this Breakdown of medical issuesa procedure. NRS, which involves keeping a bite at the lips Failure to thrive 25 26.0 and ignoring problem behaviors until acceptance occurs, Gastroesophageal reflux 21 22.8 was used is 47.9% of the studies, whereas PG, or the use of Gastrointestinal problems 14 15.2 a prompt to open the mouth if a bite was not initially Anatomical abnormalities 10 10.9 accepted, was used in 20.8% of studies. Although often not Genetic disorder 10 10.9 explicitly described by their authors as EE, a quarter of the Pulmonary disorder/dysfunction 7 7.6 studies (25%) involved treatments in which children were CNS disorder/malformation 6 6.5 asked to feed themselves and refusal behaviors were placed Prematurity 4 4.3 on extinction with a less intrusive level of prompting (i.e., Food allergies 3 3.3 ‘‘non-removal of the food’’ by ignoring disruptive behav- Cardiac impairment 2 2.2 iors plus redirecting a child back to the table in response to Other 11 12.0 leaving plus not removing the food for a set amount of a Subheadings may not add up to 100% due to multiple medical or time). DRA was also a common treatment element, with developmental issues per participant reinforcement of acceptance or swallowing cited in 77.1% % Calculated based on total sample, n = 92 of studies. A smaller number of studies (10.4%) involved 123
- 8. Clin Child FamPsychol Rev (2010) 13:348–365 355 procedures aimed at enriching the feeding environment by Dependent Variables providing access to social attention and preferred tangible objects throughout the meal session regardless of a child’s Acceptance of food into the mouth was the most frequent feeding behavior. Although often referred to as ‘‘non- measure of food intake (72.9% of studies), although studies contingent reinforcement,’’ the present review uses the varied with regard to how acceptance was operationally more precise term ‘‘non-contingent access to preferred defined (see Table 4). The definition often included a time items’’ (NCA). Items selected for use during DRA or NCA limit for the bite to pass the lips after the initial presenta- procedures included preferred toys and activities, as well as tion for acceptance to be scored (e.g., 5 s acceptance). highly preferred foods. Empirical procedures for identify- Acceptance was typically presented as a percentage of total ing highly preferred leisure items, such as paired choice bites entering the mouth during a session or meal (60.4% of preference assessments (e.g., Fisher et al. 1992), were cited studies); less common (12.5%) were studies presenting in 17 studies (45.9%) implementing DRA and NCA pro- frequency data (e.g., number of bites accepted; number cedures. Less common (10.4% of studies) were punish- bites accepted per minute). To increase the consistency ment-based procedures (e.g., response cost; time-out). In among outcome measures, studies that presented both the addition to consequence-based procedures, antecedent number of bites accepted and bites refused per session/ manipulations, including modifying food texture, spoon meal were converted to a percentage of bites accepted if volume, and/or number of bites per meal, were cited in these values equaled the total number of bite presented. 47.8% of studies. Forty-three studies (89.6%) incorporated Swallowing of bites was a less frequent measure of food more than one element in a ‘‘treatment package’’. The most intake (27.1% of studies). Swallowing was typically common packages involved EE and DRA (17 studies) or assessed by having the feeder examine the inside of the EE, DRA and antecedent manipulations (13 studies). child’s mouth. Similar to acceptance, many definitions Treatment settings included hospital inpatient units included an element of time, such as rapid swallowing (43.8% of studies), followed by home/school (29.2%), day defined as swallowing before 30 s (i.e., mouth cleans). treatment programs (16.7%), outpatient clinics (10.4%), Outcomes were most often presented as percentage of bites and residential facilities (6.3%). While most participants swallowed per session or meal, representing 22.9% of (60.4%) received treatment in an inpatient or day treatment studies; only two studies presenting frequency data for this setting, there was a notable trend in terms of the setting variable. As with frequency of acceptance, data were in which certain feeding issues were addressed. A sig- converted to percentages where possible. Finally, six nificant proportion of children with tube (69.7%; studies presented data on the total volume of food con- X2[3, N = 43] = 47.14, p .0001) and bottle dependence sumed measured in grams or cubic centimeters. (87%; X2[2, N = 15] = 19.2, p .0001) were treated at The decision rules adopted for this review allowed inpatient or day treatment facilities. In contrast, no sig- results for a single participant to contribute to more than one nificant difference in treatment setting was detected for dependent measure. Only six studies, however, presented children treated for food selectivity (inpatient/day treat- data on two measures of food intake (i.e., four with per- ment: n = 8; home/school: n = 15; outpatient: n = 5; centage acceptance and swallowing; one with percentage residential facility: n = 2). acceptance and grams and one with number of bites Trained therapists were identified as treatment providers accepted and grams). This resulted in 14 participants in 81.3% of studies, with fewer outcomes documented with (14.6% of the sample) contributing to more than one effect parents or teachers serving as primary interventionist from size calculation, resulting in a total of 109 effect size esti- the onset of the study. Length of intervention, derived from mates across the three categories of dependent measures. the horizontal axis of treatment graphs, was most often presented as 5 or 10 bites sessions (58.3% of studies), Treatment Outcomes followed by meals (22.9%), and days (14.5%). Two studies presented data in terms of weeks. The average number of The overall mean PND for all outcome measures was sessions was 76 (SD = 45), number of meals was 76 87.95% (SD = 29.54%), with a range of 0–100% (See (SD = 37), days in treatment was 47 (SD = 11), and Table 4). This falls in the effective range of treatment weeks in treatment was 26 (SD = 20). Although the outcomes based on Scruggs and Mastropieri (1998) crite- process was implied in most articles, only 58.3% of studies ria. PND scores were high across dependent variables documented systematic training to generalize treatment (range 81.75–98.85%), with all values falling in the gains to caregivers. Follow-up was reported in 52.1% of effective to very effective ranges. PND scores were con- the studies, all of which reported sustained or improved sistent across measures of acceptance (Percentage Data: feeding outcomes. Table 3 presents a detailed breakdown M = 87.87%; Frequency Data: M = 88.8%), falling in the of the intervention characteristics by study. effective treatment range, based on established standards. 123
- 9. Table 3 Interventioncharacteristics by study 356 Study Treatment elements Setting Extinction Non- Physical Ignoring/non- Differential Non- Antecedent Punishment Other Inpatient Day Outpatient Home/ Group home/ 123 removal of guidance removal of plate/ reinforcement contingent manipulation/ treatment school residential the spoon food access fading facility Ahearn (2003) X X Ahearn et al. (1996a) X x x X X Ahearn et al. (1996b) X x x X X Anderson and McMillan (2001) X x X X Casey et al. (2006) X x X X Casey et al. (2009) X x X X Coe et al. (1997) X x X X Cooper et al. (1999) X x X X X Cooper et al. (1995) X x X X X Dawson et al. (2003) X x X DeMoor et al. (2007) X x X X X X Didden et al. (1999) X x X X X Duker (1981) X x X X Freeman and Piazza (1998) X x X X Gentry and Luiselli (2008) X x X X Greer et al. (1991) X X X Hoch et al. (1994) X x X X Hoch et al. (2001) X x X X Johnson and Babbitt (1993) X x X X X Kahng et al. (2003) X x x X X X Kahng et al. (2001) X X X Kern and Marder (1996) X x X X Kerwin et al. (1995) X x x X X Lamm and Greer (1988)a X X X X X X Levin and Carr (2001) X X X Luiselli (1994) X X Luiselli (2000) X x X X X Luiselli et al. (1985) X x X X Luiselli and Gleason (1987) X x X X X McCartney et al. (2005) X x X X X Mueller et al. (2004) X x X X X X Najdowski et al. (2003) X x X X X Najdowski et al. (2010) X x X X X X O’Reilly and Lancioni (2001) X x X X Patel et al. (2002) X x X X Patel et al. (2002) X x X X X Clin Child Fam Psychol Rev (2010) 13:348–365
- 10. Table 3 continued Study Treatment elements Setting Extinction Non- Physical Ignoring/non- Differential Non- Antecedent Punishment Other Inpatient Day Outpatient Home/ Group home/ removal of guidance removal of plate/ reinforcement contingent manipulation/ treatment school residential the spoon food access fading facility Patel et al. (2007) X X X Piazza et al. (2003)a X x x X X X Piazza et al. (2002)a X x X X X Reed et al. (2005) X x X X Reed et al. (2004) X x X X Riordan et al. (1984) X x X X X Riordan et al. (1980) X x X X X Sevin et al. (2002) X x X Clin Child Fam Psychol Rev (2010) 13:348–365 Tarbox et al. (2010) X x X X Werle et al. (1993) X x X X X Wilder et al. (2005) X X Wood et al. (2009) X x X X X n 40 23 10 12 37 5 22 5 2 21 8 5 14 3 % Of total studies (n = 48) 83.3 47.9 20.8 25.0 77.1 10.4 45.8 10.4 4.2 43.8 16.7 10.4 29.2 6.3 Study Primary therapist Generalization Trained therapist Parent Teacher Follow-up reported (Y/N) Parent training reported (Y/N) Ahearn (2003) X X Ahearn et al. (1996a) X X Ahearn et al. (1996b) X X X Anderson and McMillan (2001) X X Casey et al. (2006) X X Casey et al. (2009) X X X Coe et al. (1997) X Cooper et al. (1999) X X X Cooper et al. (1995) X X X X Dawson et al. (2003) X DeMoor et al. (2007) X X X Didden et al. (1999) X X X Duker (1981) X X Freeman and Piazza (1998) X Gentry and Luiselli (2008) X X Greer et al. (1991) X Hoch et al. (1994) X X X Hoch et al. (2001) X X 357 123
- 11. Table 3 continued 358 Study Primary therapist Generalization Trained therapist Parent Teacher Follow-up reported (Y/N) Parent training reported (Y/N) 123 Johnson and Babbitt (1993) X Kahng et al. (2003) X X Kahng et al. (2001) X X Kern and Marder (1996) X X Kerwin et al. (1995) X X Lamm and Greer (1988)a X X X Levin and Carr (2001) X Luiselli (1994) X X X Luiselli (2000) X X X Luiselli et al. (1985) X X X Luiselli and Gleason (1987) X X McCartney et al. (2005) X X X Mueller et al. (2004) X X Najdowski et al. (2003) X X X Najdowski et al. (2010) X X X O’Reilly and Lancioni (2001) X X X Patel et al. (2002) X Patel et al. (2002) X Patel et al. (2007) X X X Piazza et al. (2003)a X X X Piazza et al. (2002)a X Reed et al. (2005) X Reed et al. (2004) X Riordan et al. (1984) X X X Riordan et al. (1980) X X Sevin et al. (2002) X Tarbox et al. (2010) X X X Werle et al. (1993) X X Wilder et al. (2005) X Wood et al. (2009) X n 39 9 1 28 25 % Of total studies (n = 48) 81.3 18.8 2.1 58.3 52.1 a Studies involving multiple participants receiving treatment in different settings Clin Child Fam Psychol Rev (2010) 13:348–365
- 12. Clin Child FamPsychol Rev (2010) 13:348–365 359 Table 4 PND, NAP, and effect size values by dependent variable Dependent variable # Contributing # Contributing Mean PND (Standard Mean NAP (Standard Effect size studies (%) participants (%) deviation) n = 109a deviation) n = 109a (d) n = 106a Acceptance (Percent) n = 29 (60.4%) n = 54 (56.3%) 87.87 (31.63) .97 (.09) 2.598 Acceptance (Frequency) n = 6 (12.5%) n = 17 (17.7%) 88.8 (24.8) .98 (.04) 2.698 Swallowing (Percent) n = 11 (22.9%) n = 22 (22.9%) 81.75 (36.04) .91 (.20) 1.81 Swallowing (Frequency) n = 2 (4.2%) n = 7 (7.3%) 98.85 (3.27) .98 (.03) 2.88 Volume n = 6 (12.5%) n = 9 (9.4%) 95.40 (5.5) .97 (.03) 2.89 Total n = 54 n = 109a 87.95 (29.54) .96 (.12) 2.46 PND percent of non-overlapping data, NAP non-overlap of all pairs a Data for some participants contributed to more than one dependent variable PND scores regarding swallowing varied slightly according involving bottle/liquid dependence provided data regarding to whether percentage (M = 81.75) or frequency data improved intake, all noting discontinuation of bottle feed- (M = 98.85) were reported, falling in the effective and ing. Only one case (12.5%) involving poor oral intake highly effect ranges, respectively. Studies reporting vol- provided data regarding consumption following treatment, ume of food consumed during meals had a mean PND of with a 50% improvement reported. Improvements in die- 95.80%, which falls in the highly effective range. tary variety were reported in 75 out of the 96 cases NAP values reflected similar levels of improved per- (78.1%), with children most often described as consuming formance, with an overall mean NAP score of .96 of foods from ‘‘all food groups’’ following treatment (30 of (SD = .12; range of .29 to 1). The size of the treatment 75 cases; 40%). A specific number of foods targeted were effect was large for overall outcomes (d = 2.46), with all reported in 26 of 75 cases (34.7%), with 16 foods repre- values across measures of acceptance reflecting large senting the modal number introduced during treatment. The treatment effects by conventional standards (range remaining 19 participants were described as improving d = 1.81–2.89). No subgroup differences in effectiveness their nutritional status, but no dietary details were pro- were detected in terms of feeding concern and treatment vided. Finally, data regarding anthropometric parameters setting. However, the high degree of heterogeneity among were reported in 23 of the 96 cases (23.9%). Change in the sample in terms of outcome measures, presenting weight from pre-treatment levels was reported for 19 problem, and the aforementioned trend for different types children, with an average increase of 1.67 kg (range of feeding issues addressed in settings with varying 0–5.4 kg); average weight gain per day was presented for intensity levels may represent an uncontrolled bias in this four children (M = 39.25 g/day; range 11–58 g). analysis. Treatment elements were not evaluated separately due to lack of sufficient studies with single treatment packages appropriate for this level of component analysis. Discussion Medical and Nutritional Outcomes Findings from this review provide further support for the use of behavioral intervention in the treatment of severe Outcomes beyond behavioral change were not consistently feeding disorders. The identified studies represent an documented. Tube reductions were reported in 25 of 43 experimentally sound body of literature demonstrating children (58.1%) reliant on this method for their nutritional significant improvements in mealtime behaviors among a needs. In the remaining 14 cases, although improved intake sample of 96 children. The majority of studies included was implied, specific volume reductions associated with were published since Kerwin (1999) first reviewed the treatment were not specified. Of the 25 cases reporting on literature, highlighting the growth of research in this area. tube feedings, they were eliminated in 16 cases (64%) and Findings also reflect a noticeable increase in the use of reduced by an average of 57.1% (range 42–60%) in 7 extinction-based procedures, such as NRS and PG, when cases. Two cases involved a specific volume of tube compared with Kerwin’s findings. This likely reflects the reduction per day (e.g., 6 oz), but improvement was not current review’s focus on more severe feeding issues, as translated into a percentage of daily needs. Less outcome well as refinements in the behavioral technology used to data were available for bottle/liquid dependence, poor oral address chronic feeding concerns. Refinements in treatment intake, or food selectivity. Five of the fifteen cases (33%) appear, in part, guided by descriptive assessments and 123
- 13. 360 Clin Child Fam Psychol Rev (2010) 13:348–365 functional analyses (e.g., Piazza et al. 2003) indicating that professionals. In addition to behavioral psychology, pro- negative reinforcement (i.e., escape from feeding demands) fessions cited as collaborating in treatment development and often maintains inappropriate mealtime behaviors. In evaluation included medicine, dietetics, speech/language addition, several studies (e.g., Hoch et al. 2001; Piazza pathology, and/or occupational therapy. Given the general et al. 2003; Reed et al. 2004) comparing the relative con- acceptance that these disorders involve problems that cross tribution of different treatment elements (e.g., EE, DRA, areas of expertise, a multidisciplinary approach, at a mini- NCA) have demonstrated the importance of EE in elimi- mum, provides safeguards against possible complications nating disruptive behaviors that preclude food acceptance. with treatment (e.g., aspiration; metabolic concerns; severe As long as escape contingencies persist, these children do weight loss), while allowing design of treatment packages not eat and thus lack exposure to the sensory experience of unique to each child. Components besides behavioral treat- food and the opportunity to contact the primary and sec- ment, however, have not been evaluated in published reports, ondary reinforcement contingent upon eating (Hoch et al. suggesting the need to examine the relative contributions of 2001). Despite support for using EE, it should be noted that disciplines besides behavioral techniques in the context of milder levels of feeding difficulty (not the focus of the multidisciplinary treatment outcomes. present review) may respond to less intrusive interventions, While the current review provides support for behavioral and extinction-based procedures may be contraindicated in treatment to address severe feeding disorders, there are these cases (Farrow and Blissett 2008). limitations to the evidence for these conclusions. The goal While EE represented a common intervention, most of a feeding intervention is to achieve the closest approxi- studies incorporated additional behavioral elements into mation of age-appropriate mealtime behavior, including larger packages, which may afford additional treatment both proximate behavior change and more distal nutritional benefits. Packages involving DRA (e.g., Piazza et al. 2003) and medical goals. This entails replacing supplemental or NCA (e.g., Reed et al. 2004) have been associated with feedings, in cases involving tube or bottle dependence, and/ reduced rates of negative behaviors (e.g., crying, disrup- or increasing dietary diversity among children with severe tions) during extinction bursts. For example, Reed et al. food selectivity. Improvements in intake should be reported that, although extinction was necessary to increase accompanied by increased levels of appropriate mealtime and maintain food acceptance, the addition of NCA was behaviors (e.g., acceptance, swallowing) and, to assure associated with decreased rates of inappropriate behavior external validity, treatment gains need to be generalized to and crying in some cases. Evidence also supports the caregivers and transitioned into the home environment. potential role of antecedent manipulations (e.g., texture, While this review suggests behavioral treatment is associ- bite size, utensil) as an avenue for modifying the feeding ated with significant improvements in mealtime behaviors, demands during treatment and/or accommodating possible it also reveals the need to better document outcomes in oral motor skill deficits. For example, Kerwin et al. (1995) other areas, including changes in tube dependence, food reported an inverse relationship between appropriate variety, weight status, oral motor status, generalization of mealtime behaviors and increasing bite sizes, suggesting treatment effects, and long-term follow-up. When docu- that beginning treatment with smaller bite sizes may mented, outcomes suggested improvements in these areas, decrease possible negative side effects associated with but more systematic evaluation is recommended. Suggested introduction of food. The use of antecedent manipulations refinements in future studies include documenting behav- and/or rich reinforcement schedules along with EE suggest ioral (e.g., acceptance, swallowing), medical (e.g., weight, movement in the field toward highly specific treatment tube reductions), physical (tongue control), and social (e.g., packages that balance addressing the operant function of parent satisfaction; caregiver stress) data. Increased breadth food refusal with maintaining the least restrictive envi- and standardization in outcome measures will expand the ronment and ameliorating possible side effects associated knowledge base and strengthen conclusions from feeding with extinction procedures. intervention studies. The treatment context in which multi-component treat- The ‘‘file drawer problem’’ or the editorial practice of ment packages have been developed and evaluated is also publishing only those studies demonstrating positive out- noteworthy. Most of this research was conducted at comes also represents a potential source of bias intrinsic to intensive feeding programs involving inpatient or day comprehensive literature reviews. On the other hand, a treatment admissions. This likely reflects the need for high small number of randomized controlled studies supporting degrees of structure and supervision during intensive behavioral intervention (Benoit et al. 2000; Stark et al. treatment of severe feeding problems (Kerwin 1999), 1996, Turner et al. 1994) and recent program evaluations of especially with concurrent significant medical concerns. By multidisciplinary feeding treatment programs (Greer et al. and large, treatment packages were implemented by trained 2009; Williams et al. 2007) provide some evidence against therapists under the guidance of a multidisciplinary team of the general suppression of negative findings among single- 123
- 14. Clin Child FamPsychol Rev (2010) 13:348–365 361 case reports analyzed in the present review. For example, from supplemental tube feedings after discharge from a Benoit et al. randomly assigned a sample of 64 child/feeder day treatment program. Treatment was described as dyads involving children with tube dependence and food involving intensive behavioral therapy with input from a refusal to either a treatment group involving behavioral multidisciplinary team. At the year 2 follow-up, 74% of intervention (n = 32) or a treatment group involving the sample (n = 34) no longer received tube feedings; an nutritional education (n = 32). Nutritional education additional 17% (n = 8) received 50% or less of their involved advice regarding volume and concentration of caloric needs by feeding tube. The authors also high- oral feedings, feeding schedules and routine, and guidance lighted the cost-effectiveness of intensive feeding treat- on reducing tube feeding; behavioral intervention included ment when compared with supplemental tube feedings; the identical nutritional guidance, as well as training on annual cost of tube feeding exceeded the cost of day behavioral techniques (e.g., EE in the form of NRS; treatment in all but one case. The uniformly positive stimulus fading) to address refusal behaviors. At 8-week outcomes across those studies, combined with the size of follow-up, 15 (47%) of the 32 patients in the behavioral the treatment effects reported in the present review intervention group were no longer dependant on tube (medium to large by conventional standards), provide feedings, while no change in tube feeding status occurred convergent support for the efficacy of behavioral inter- in the nutrition group (p = .0001). Stark et al. investigated ventions in highly controlled settings. Prospective ran- the use of behavioral intervention to increase caloric intake domized controlled trials would clearly strengthen this and weight gain among a sample of five children with conclusion, providing additional protection against possi- cystic fibrosis (CF) compared with a waitlist control ble publication bias while addressing possible threats to involving four children with CF. Treatment included child internal validity (e.g., history, maturation). behavior management training focusing on differential It is noteworthy that no eligible studies from other attention, contingency management, and implementation of theoretical perspectives in psychology (e.g., family ther- mealtime rules and consequences. Following treatment, apy, psychodynamic therapy) or from other habilitative children in the behavioral intervention group experienced disciplines were identified in the current literature search. significantly greater improvement in calories per meal and This void is particularly notable, given prior research weight gain compared with the waitlist control (p = .03). showing an association between parent–child interactions Finally, Turner et al. compared the use of behavioral parent and disrupted feeding (e.g., Amaniti et al. 2004) and training versus dietary education to address feeding prob- Kerwin’s (1999) call for such research a decade ago. While lems in a sample of 20 children with feeding problems it can be argued that behavioral intervention, with its focus lasting longer than 3 months. Both groups demonstrated on repeated assessment of operationally defined behaviors, improvement in mealtime behaviors, while behavioral lends itself more readily to the stringent methodological parent training was associated with improved caregiver criteria established for this review, this does not nullify the attention during meals. importance of establishing an evidence base for other dis- Comprehensive chart reviews have also documented ciplines providing feeding therapy (e.g., medical, occupa- levels of treatment efficacy similar to those in this review, tional therapy, speech therapy, dietetics). Even with the while providing transparency regarding outcomes for all methodological criteria of the review removed, there were children treated at multidisciplinary treatment programs. few descriptions of feeding interventions, let alone well- For example, Greer et al. (2009) presented data on 121 controlled outcomes studies by other disciplines. The few children discharged from a pediatric feeding program after studies identified as evaluating non-behavioral treatment receiving treatment for tube dependence (n = 72), liquid approaches involved single demonstrations of effectiveness dependence (n = 17) or food selectivity (n = 32). During without replication, often without experimental control. meals, behavioral protocols were systematically imple- For example, Linscheid (2006) described effective treat- mented and involved antecedent and consequence manip- ment of feeding disorders as combining behavioral ele- ulations similar to the interventions described in this ments with hunger manipulations; however, only one review. Across all groups, significant improvements outcome study was located focusing on hunger provocation (p .001) were reported across several measures of feed- as a mechanism of change. Specifically, Kindermann et al. ing behavior, including acceptance, mouth cleans, oral (2008) presented outcomes for 10 children treated for tube intake, and grams consumed. Treatment was also associ- dependence in a ‘‘multidisciplinary hunger provocation ated with significant declines (p .001) in caregivers program’’. Following inpatient admission, tube feedings stress compared with pre-treatment values. Similarly, in a were systematically reduced. Concurrently, regularly study focusing on the treatment outcomes of 46 children structured meals were conducted involving reinforcement with complete tube dependence, Williams et al. (2007) for acceptance without pressure or ‘‘forced feeding’’. assessed the percent of the sample successfully removed Kindermann reported that 8 of the 10 children were 123
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