Clin Child Fam Psychol Rev (2010) 13:348–365DOI 10.1007/s10567-010-0079-7Pediatric Feeding Disorders: A Quantitative Synth...
Clin Child Fam Psychol Rev (2010) 13:348–365                                                                              ...
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Clin Child Fam Psychol Rev (2010) 13:348–365                                                                             3...
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Clin Child Fam Psychol Rev (2010) 13:348–365                                                                              ...
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Clin Child Fam Psychol Rev (2010) 13:348–365                                                                              ...
Table 3 Intervention characteristics by study                                                                             ...
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Clin Child Fam Psychol Rev (2010) 13:348–365                                                                              ...
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Clin Child Fam Psychol Rev (2010) 13:348–365                                                                              ...
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Pediatric feeding
Pediatric feeding
Pediatric feeding
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  1. 1. Clin Child Fam Psychol Rev (2010) 13:348–365DOI 10.1007/s10567-010-0079-7Pediatric Feeding Disorders: A Quantitative Synthesisof Treatment OutcomesWilliam G. Sharp • David L. Jaquess •Jane F. Morton • Caitlin V. HerzingerPublished online: 16 September 2010Ó Springer Science+Business Media, LLC 2010Abstract A systematic review of the literature regarding Keywords Autism spectrum disorders Á Behavioraltreatment of pediatric feeding disorders was conducted. intervention Á Evidence-based treatment Á Failure to thrive ÁArticles in peer-reviewed scientific journals (1970–2010) Feeding Á Feeding disorders Á Mealtime problemsevaluating treatment of severe food refusal or selectivitywere identified. Studies demonstrating strict experimentalcontrol were selected and analyzed. Forty-eight single-case Introductionresearch studies reporting outcomes for 96 participantswere included in the review. Most children presented with Eating is an essential human activity, necessary to sustaincomplex medical and developmental concerns and were life and ensure growth, but it also is a common challengetreated at multidisciplinary feeding disorders programs. for children and a source of stress for caregivers. Up toAll studies involved behavioral intervention; no well- 40% of toddlers and early school-age children experiencecontrolled 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’’ eatingsion 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 foodbehavior. Clinical and research implications are discussed, away), and/or fluctuating hunger (Reau et al. 1996). Mildincluding movement toward the identification of key difficulties typically resolve spontaneously or with lowbehavioral antecedents and consequences that promote intensity interventions, such as caregiver education in foodappropriate mealtime performance, as well as the need to preparation/presentation, and/or nutritional guidancebetter document outcomes beyond behavioral improve- (Greer et al. 2009; Kerwin 1999). Between 3 and 10% ofments, such as changes in anthropometric parameters, children, however, develop chronic feeding issuesgeneralization of treatment gains to caregivers, and exceeding ordinary developmental variation and possiblyimprovements 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 feedingW. 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 asMarcus Autism Center, Atlanta, GA, USA ‘‘feeding disorders’’ due to their chronic and more severee-mail: course, often involving the complex interplay amongD. L. Jaquess biological, psychological, and social factors and requiringe-mail: intensive intervention to avoid long-term medical andW. G. Sharp Á D. L. Jaquess Á J. F. Morton developmental sequelae (Babbitt et al. 1994; LindbergEmory University School of Medicine, Atlanta, GA, USA et al. 1991; Sanders et al. 1993).123
  2. 2. Clin Child Fam Psychol Rev (2010) 13:348–365 349 The process of assessing and treating severe feeding Significant feeding disturbances have also been reporteddisorders is complicated by a number of interrelated factors. among children with no clear physiological precursor orThe psychiatric diagnosis of ‘‘Feeding Disorder of Infancy developmental issues and may continue in children whoseor Early Childhood’’ is non-specific, encompassing children organic issues are resolved. Causal factors in these cases arewho 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 oforganic complaint (American Psychiatric Association structure conducive to eating (e.g., unrestrained access to2000). The medical diagnosis for these disorders ‘‘Feeding food; irregular mealtimes), exposure to developmentallyDifficulties 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 whoseetiological pathways. A variety of organic factors that lead function is to escape unpleasant feeding experiences and/orto 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 inappropriateaccompany conditions such as cystic fibrosis, mitochondrial behaviors) and negative reinforcement (e.g., removing fooddisease, 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 adiarrhea (e.g., gastroesophageal reflux, gastroenteritis, caregiver inadvertently reinforces problem behaviors, thosedysmotility), (3) structural or anatomical defects (e..g, behaviors tend to become more frequent or intense, which, inbronchopulmonary 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 aftersensitivity to food tastes, smells, and textures (Arvedson the child or caregiver withdraws from the feeding situation or2008; 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 experiencessevere 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 possibleApproximately one-third of all children with develop- etiological pathways, feeding disorders often include moremental 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 intolack of independent self-feeding skills, disruptive mealtime the mouth, chewing and forming a bolus, and swallowingbehaviors, 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 ofPrevalence estimates have been reported to be much higher consumption, which further complicates the diagnostic andamong 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 behaviorsstrong preferences for certain foods (by type, texture, color, (e.g., head turning, batting at the spoon) that interfere withor packaging), consume a narrower range and quantity of accepting a bite into the mouth, while other children fail tofood when compared with peers, and/or display elevated consume an adequate volume of food due to packing orrates of disruptive behavior when presented with non-pre- expelling bites. In addition, treatment resolving refusal atferred 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 foodet 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 (Gulottaidentifiable organic factors or gastrointestinal etiology, et al. 2005). This presents a unique challenge for caregiversleading to the hypothesis that aberrant feeding habits and professionals addressing feeding concerns, with theamong 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 feedingtion (Ledford and Gast 2006). problems combined with their complex biopsychosocial 123
  3. 3. 350 Clin Child Fam Psychol Rev (2010) 13:348–365etiology intensifies the need to identify evidence-based elements included DRA, EE, and stimulus fading. Whiletreatments. Although numerous researchers have docu- providing an updated survey the literature, Williams andmented 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 providedWilliams 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 problemspublished between 1970 and 1997. Twenty-nine studies were and treatments effecting their remediation, while also layingidentified as meeting the methodological criteria of the Task the groundwork for an updated quantitative review of theForce on Promotion and Dissemination of Psychological literature. The past decade has seen a significant increase inProcedures (1995) of the American Psychological Associa- the number of studies focusing on the analysis and treatmenttion, all of which involved behavioral intervention. Differ- of severe feeding disorders, yielding important dataential 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 sizephysically guiding appropriate feeding responses were of treatment outcomes for both group and single-case studiesidentified as effective interventions. DRA of acceptance in have also been developed and refined (Busk and Serlincombination with escape extinction (EE) procedures target- 1992). With these advances in place, the current reviewing 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 investigateintervention. Kerwin acknowledged that non-behavioral interventions aimed at improving intake among childreninterventions may be effective in treating feeding problems with severe feeding disorders; (2) determine the overallbut 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 thehighlighted the need to investigate the setting in which treatment elements, population, and settings associated withevidence-based treatments are developed and evaluated. significant improvements in feeding patterns. Subsequent less comprehensive literature reviews byLedford and Gast (2006) and Williams et al. (2010) addedsupport for the effectiveness of behavioral intervention to Methodaddress chronic feeding concerns. Ledford and Gastfocused specifically on the treatment of children with ASD Study Identification and Eligibility Criteriaand feeding difficulties, reviewing studies with experi-mental control published between 1994 and 2000. Nine Studies investigating the treatment of pediatric feedingsingle-case design studies were identified, all involving one disorders were identified through searches of the MedLineor more behavioral elements to address severe food and PsychINFO databases. The search parameters includedselectivity. In each case, the use of behavioral intervention combinations of the following key words: feeding, foodwas 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, familyDRA, 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 ofMore 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, referencesgeting 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 theor reliance on supplemental tube feedings. In all 38 studies, use of an experimental design to investigate treatmentimprovements in oral intake were reported, with more than outcomes, including the use of a control group with grouphalf of the children who received some form of supple- designs or experimental single-case research methodologymental 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 withoutinvolved one or more behavioral interventions incorporated randomization to a control condition and single-caseinto 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 control123
  4. 4. Clin Child Fam Psychol Rev (2010) 13:348–365 351for alternative hypotheses. In addition, studies needed to was empirically measured and graphically illustrated withmeet the following criteria: clearly identifiable baseline and treatment phases for each participant. Repeated data points, not mean scores or1. 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) If2. 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; see5. 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 percentagewere 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 datainvolved experimental control (i.e., Benoit et al. 2000; points exceeding the highest baseline data point by the totalStark 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). Possiblecombining findings from studies involving single-case scores range from 0 to 100%, with higher scores reflectingdesigns. Other group studies consisted of program evalu- more effective treatments. To address the influences ofations that lacked experimental control or involved a single outliers, the stringent conventions set forth by Scruggsdemonstration of positive outcomes (e.g., Berger-Gross et al. were adopted, with a PND score recorded as zeroet al. 2004; Greer et al. 2009; Kindermann et al. 2008; when a single baseline data point reached ceiling level onWilliams 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 ofanalysis. 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 oftional 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. 5. 352 Clin Child Fam Psychol Rev (2010) 13:348–365require 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 targettions for addressing floor or ceiling levels. Many studies in (e.g., acceptance, swallowing) was recorded, and variationsthis 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 reliabilitying 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 or70% reflect ‘‘questionable’’ treatments, scores from 70 to some combination of these methods. Intervention data90% are associated with ‘‘effective’’ treatments, and scores coded included type of intervention, treatment setting, andabove 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/ignoringas 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 environmentsature, 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, bitevarious 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 alsoquestions 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 therapistdrawn from baseline. Each baseline data point is compared (e.g., parent, teacher, trained therapist) were also identifiedwith 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, ifnumber 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 convertingoverlap by the total number of possible pairs. Possible raw data displayed in the primary articles to a standardizedscores range from 0 to 1 (higher scores reflecting more metric by measuring with a ruler the distance between theeffective treatments). The result yields a nomothetic effect horizontal (X) axis and the bottom of each data point insize that can be interpreted in relation to effect sizes that millimeters. Similar data conversion procedures have beenhave gained wide acceptance in large-scale group studies, shown to have a high degree of inter-rater reliability inwith formulas available for estimating Cohen’s d and R previous meta-analyses (Allison et al. 1995; Campbellsquared 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 athe 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 eachtrained 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 dataand treatment techniques/protocols. Characteristics in each across participants and outcomes to be included in theof 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, this2007) 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 inDemographic 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 firstretardation), medical issues (e.g., gastroesophageal reflux, baseline phase and the last treatment phase were included,123
  6. 6. Clin Child Fam Psychol Rev (2010) 13:348–365 353as recommended by Faith et al. (1996) and applied in Table 1 Description of studies and experimental characteristicssimilar review studies (e.g., Campbell 2003). In studies Characteristic n %involving multi-element designs conducted across baselineand treatment phases, a single effect size was calculated Journal titleonly if both treatment paths were presented in the baseline Journal of Applied Behavior Analysis 25 52.1and final treatment phases. This allowed analysis of overall Behavior Modification 7 14.6treatment effect rather than breaking down individual Behavioral Interventions 3 6.3treatment elements. PND and NAP were calculated using Journal of Behavioral Therapy and Experimental Psychiatry 2 4.2all data points in the first baseline and last treatment pha- Education and Treatment of Children 2 4.2ses, allowing for common outcome metrics to be generated American Journal of Mental Retardation 1 2.1for 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 2.1analysis were randomly selected for independent coding by Focus on Autism and Other Developmental Disabilities 1 2.1two trained staff to calculate inter-rater reliability. These Journal of Clinical Child Psychology 1 2.1articles involved 23 different participants (24% of all par- Journal of Behavioral Education 1 2.1ticipants) contributing data for 32 separate dependent Journal of Developmental and Physical Disabilities 1 2.1variables (29% of all outcomes). For descriptive informa- Journal of Intellectual Disability Research 1 2.1tion extracted during the review process, inter-rater Journal of Positive Behavior Interventions 1 2.1agreement was calculated through the percent agreement Total studies 48 Number of participants contributed per studymethod: # agreements/(# agreements ? # disagree- 1 25 52.1ments) 9 100, as well as the Kappa statistic. The mean 2 7 14.5inter-rater agreement across all variables was 94.1% (range 3 8 16.787.5–100%) with a corresponding Kappa of .8 (range .7 to 4 7 14.6.99). For quantitative information extracted via ruler, reli- 5 1 2.1ability was calculated on all individual data points using Total participants 96Spearman’s q. The overall inter-rater reliability for quan- Study breakdown (n = 48)titative data was q = .942. Inter-rater agreement for both Year publishedqualitative and quantitative exceeded the 80% acceptable 2000–2010 29 60.4standard of agreement widely adopted and recommended 1990–1999 13 27.1during quantitative synthesis of single-case research (e.g., 1980–1989 6 12.5Campbell 2003). To further ensure the accuracy, the first Primary experimental designauthor conducted a second review of all articles included in Reversal 20 41.7the study, focusing on potential areas of discrepancy Multiple baseline 11 22.9highlighted by the inter-rater analysis and consensus with Changing criterion 7 14.6the second author was reached in cases of ambiguity. Multielement and reversal 5 10.4 Multielement 2 4.2 Multielement and multiple baseline 2 4.2Results Multiple baseline and reversal 1 2.0 Unit of measurement reported for data collectionCharacteristics of Studies and Participants Sessions 28 58.3 Meals 11 22.9The search yielded 48 studies meeting inclusion criteria out Days 7 14.6of a pool of 124 possible articles, resulting in 96 partici- Weeks 2 4.2pants 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 = 85–100studies. More than half of the studies were published after % Calculated based on total sample n = 822000, indicating a recent increase in studies employing ahigh degree of experimental control to investigate thetreatment of severe feeding problems. Interestingly, only 9of the 29 studies identified by Kerwin (1999) were included liquid intake (n = 1), did not present individual data pointsin 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 meetingpendent feeding skills or healthy eating habits (n = 11) or inclusion criteria were published in 14 journals, with the 123
  7. 7. 354 Clin Child Fam Psychol Rev (2010) 13:348–365Journal of Applied Behavior Analysis contributing the Feeding tube dependence was the most prevalent feedinglargest 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 poorreliability of overall observations exceeded the inclusion oral intake (8.3%). Only a small subgroup of participantscriterion of 80% agreement, with an average of 96.7% (10.5%) were described as ‘‘typically developing’’, withacross 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. Developmentalmean 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 issuesTable 2 Description of participants (9.7%). Consistent with the literature to date, 90.9% of children with ASD (20 of 22 cases) presented with foodCharacteristic 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 severeTotal 96 feeding problems often co-occur with complex medicalFeeding 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 emphasizedBreakdown 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 aMedical 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 thisBreakdown 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 ofa Subheadings may not add up to 100% due to multiple medical or time). DRA was also a common treatment element, withdevelopmental 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%) involved123
  8. 8. Clin Child Fam Psychol Rev (2010) 13:348–365 355procedures aimed at enriching the feeding environment by Dependent Variablesproviding access to social attention and preferred tangibleobjects throughout the meal session regardless of a child’s Acceptance of food into the mouth was the most frequentfeeding behavior. Although often referred to as ‘‘non- measure of food intake (72.9% of studies), although studiescontingent reinforcement,’’ the present review uses the varied with regard to how acceptance was operationallymore precise term ‘‘non-contingent access to preferred defined (see Table 4). The definition often included a timeitems’’ (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 totaling highly preferred leisure items, such as paired choice bites entering the mouth during a session or meal (60.4% ofpreference assessments (e.g., Fisher et al. 1992), were cited studies); less common (12.5%) were studies presentingin 17 studies (45.9%) implementing DRA and NCA pro- frequency data (e.g., number of bites accepted; numbercedures. Less common (10.4% of studies) were punish- bites accepted per minute). To increase the consistencyment-based procedures (e.g., response cost; time-out). In among outcome measures, studies that presented both theaddition 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 ifvolume, 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 foodmore than one element in a ‘‘treatment package’’. The most intake (27.1% of studies). Swallowing was typicallycommon packages involved EE and DRA (17 studies) or assessed by having the feeder examine the inside of theEE, 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 bitesand 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 thissetting, there was a notable trend in terms of the setting variable. As with frequency of acceptance, data werein which certain feeding issues were addressed. A sig- converted to percentages where possible. Finally, sixnificant 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 allowedinpatient or day treatment facilities. In contrast, no sig- results for a single participant to contribute to more than onenificant difference in treatment setting was detected for dependent measure. Only six studies, however, presentedchildren 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 percentageresidential 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 participantsin 81.3% of studies, with fewer outcomes documented with (14.6% of the sample) contributing to more than one effectparents 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 oftenpresented as 5 or 10 bites sessions (58.3% of studies), Treatment Outcomesfollowed by meals (22.9%), and days (14.5%). Two studiespresented data in terms of weeks. The average number of The overall mean PND for all outcome measures wassessions 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 treatmentweeks 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 variablesdocumented systematic training to generalize treatment (range 81.75–98.85%), with all values falling in thegains 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 theof the intervention characteristics by study. effective treatment range, based on established standards. 123
  9. 9. Table 3 Intervention characteristics 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. 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 357123
  11. 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. 12. Clin Child Fam Psychol Rev (2010) 13:348–365 359Table 4 PND, NAP, and effect size values by dependent variableDependent variable # Contributing # Contributing Mean PND (Standard Mean NAP (Standard Effect size studies (%) participants (%) deviation) n = 109a deviation) n = 109a (d) n = 106aAcceptance (Percent) n = 29 (60.4%) n = 54 (56.3%) 87.87 (31.63) .97 (.09) 2.598Acceptance (Frequency) n = 6 (12.5%) n = 17 (17.7%) 88.8 (24.8) .98 (.04) 2.698Swallowing (Percent) n = 11 (22.9%) n = 22 (22.9%) 81.75 (36.04) .91 (.20) 1.81Swallowing (Frequency) n = 2 (4.2%) n = 7 (7.3%) 98.85 (3.27) .98 (.03) 2.88Volume n = 6 (12.5%) n = 9 (9.4%) 95.40 (5.5) .97 (.03) 2.89Total n = 54 n = 109a 87.95 (29.54) .96 (.12) 2.46PND percent of non-overlapping data, NAP non-overlap of all pairsa Data for some participants contributed to more than one dependent variablePND scores regarding swallowing varied slightly according involving bottle/liquid dependence provided data regardingto 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 intakehighly 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 consumingformance, 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 wereeffect 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. Thetreatment effects by conventional standards (range remaining 19 participants were described as improvingd = 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 parameterssetting. However, the high degree of heterogeneity among were reported in 23 of the 96 cases (23.9%). Change inthe sample in terms of outcome measures, presenting weight from pre-treatment levels was reported for 19problem, and the aforementioned trend for different types children, with an average increase of 1.67 kg (rangeof feeding issues addressed in settings with varying 0–5.4 kg); average weight gain per day was presented forintensity 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 separatelydue to lack of sufficient studies with single treatmentpackages appropriate for this level of component analysis. DiscussionMedical and Nutritional Outcomes Findings from this review provide further support for the use of behavioral intervention in the treatment of severeOutcomes beyond behavioral change were not consistently feeding disorders. The identified studies represent andocumented. Tube reductions were reported in 25 of 43 experimentally sound body of literature demonstratingchildren (58.1%) reliant on this method for their nutritional significant improvements in mealtime behaviors among aneeds. In the remaining 14 cases, although improved intake sample of 96 children. The majority of studies includedwas implied, specific volume reductions associated with were published since Kerwin (1999) first reviewed thetreatment were not specified. Of the 25 cases reporting on literature, highlighting the growth of research in this feedings, they were eliminated in 16 cases (64%) and Findings also reflect a noticeable increase in the use ofreduced by an average of 57.1% (range 42–60%) in 7 extinction-based procedures, such as NRS and PG, whencases. Two cases involved a specific volume of tube compared with Kerwin’s findings. This likely reflects thereduction per day (e.g., 6 oz), but improvement was not current review’s focus on more severe feeding issues, astranslated into a percentage of daily needs. Less outcome well as refinements in the behavioral technology used todata were available for bottle/liquid dependence, poor oral address chronic feeding concerns. Refinements in treatmentintake, or food selectivity. Five of the fifteen cases (33%) appear, in part, guided by descriptive assessments and 123
  13. 13. 360 Clin Child Fam Psychol Rev (2010) 13:348–365functional 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 andoften maintains inappropriate mealtime behaviors. In evaluation included medicine, dietetics, speech/languageaddition, several studies (e.g., Hoch et al. 2001; Piazza pathology, and/or occupational therapy. Given the generalet al. 2003; Reed et al. 2004) comparing the relative con- acceptance that these disorders involve problems that crosstribution 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 complicationsnating disruptive behaviors that preclude food acceptance. with treatment (e.g., aspiration; metabolic concerns; severeAs long as escape contingencies persist, these children do weight loss), while allowing design of treatment packagesnot 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 of2001). Despite support for using EE, it should be noted that disciplines besides behavioral techniques in the context ofmilder 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 behavioraland extinction-based procedures may be contraindicated in treatment to address severe feeding disorders, there arethese 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, includinglarger packages, which may afford additional treatment both proximate behavior change and more distal nutritionalbenefits. Packages involving DRA (e.g., Piazza et al. 2003) and medical goals. This entails replacing supplementalor 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 severetions) during extinction bursts. For example, Reed et al. food selectivity. Improvements in intake should bereported that, although extinction was necessary to increase accompanied by increased levels of appropriate mealtimeand maintain food acceptance, the addition of NCA was behaviors (e.g., acceptance, swallowing) and, to assureassociated with decreased rates of inappropriate behavior external validity, treatment gains need to be generalized toand 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 inoral motor skill deficits. For example, Kerwin et al. (1995) other areas, including changes in tube dependence, foodreported an inverse relationship between appropriate variety, weight status, oral motor status, generalization ofmealtime 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. Suggestedintroduction 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 breadthfood refusal with maintaining the least restrictive envi- and standardization in outcome measures will expand theronment and ameliorating possible side effects associated knowledge base and strengthen conclusions from feedingwith extinction procedures. intervention studies. The treatment context in which multi-component treat- The ‘‘file drawer problem’’ or the editorial practice ofment 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 tointensive feeding programs involving inpatient or day comprehensive literature reviews. On the other hand, atreatment admissions. This likely reflects the need for high small number of randomized controlled studies supportingdegrees 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 ofespecially 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 againsttherapists under the guidance of a multidisciplinary team of the general suppression of negative findings among single-123
  14. 14. Clin Child Fam Psychol Rev (2010) 13:348–365 361case reports analyzed in the present review. For example, from supplemental tube feedings after discharge from aBenoit et al. randomly assigned a sample of 64 child/feeder day treatment program. Treatment was described asdyads involving children with tube dependence and food involving intensive behavioral therapy with input from arefusal to either a treatment group involving behavioral multidisciplinary team. At the year 2 follow-up, 74% ofintervention (n = 32) or a treatment group involving the sample (n = 34) no longer received tube feedings; annutritional education (n = 32). Nutritional education additional 17% (n = 8) received 50% or less of theirinvolved 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; theidentical nutritional guidance, as well as training on annual cost of tube feeding exceeded the cost of daybehavioral techniques (e.g., EE in the form of NRS; treatment in all but one case. The uniformly positivestimulus fading) to address refusal behaviors. At 8-week outcomes across those studies, combined with the size offollow-up, 15 (47%) of the 32 patients in the behavioral the treatment effects reported in the present reviewintervention group were no longer dependant on tube (medium to large by conventional standards), providefeedings, 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 thisand 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 toinvolving 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 otherattention, 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 habilitativechildren 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 researchweight gain compared with the waitlist control (p = .03). showing an association between parent–child interactionsFinally, Turner et al. compared the use of behavioral parent and disrupted feeding (e.g., Amaniti et al. 2004) andtraining versus dietary education to address feeding prob- Kerwin’s (1999) call for such research a decade ago. Whilelems in a sample of 20 children with feeding problems it can be argued that behavioral intervention, with its focuslasting 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 methodologicalparent training was associated with improved caregiver criteria established for this review, this does not nullify theattention 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 thewhile providing transparency regarding outcomes for all methodological criteria of the review removed, there werechildren 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 fewchildren discharged from a pediatric feeding program after studies identified as evaluating non-behavioral treatmentreceiving treatment for tube dependence (n = 72), liquid approaches involved single demonstrations of effectivenessdependence (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 onereview. 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 behavior, including acceptance, mouth cleans, oral (2008) presented outcomes for 10 children treated for tubeintake, and grams consumed. Treatment was also associ- dependence in a ‘‘multidisciplinary hunger provocationated with significant declines (p .001) in caregivers program’’. Following inpatient admission, tube feedingsstress compared with pre-treatment values. Similarly, in a were systematically reduced. Concurrently, regularlystudy focusing on the treatment outcomes of 46 children structured meals were conducted involving reinforcementwith 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
  15. 15. 362 Clin Child Fam Psychol Rev (2010) 13:348–365successfully weaned from tube feedings, although the study Referenceslacked a control group/condition, and a closer inspection ofage-adjusted weight for height (i.e., z-scores) at the Ahearn, W. H. (2003). Using simultaneous presentation to increase vegetable consumption in a mildly selective child with autism.6-month follow-up suggests actual decline on that outcome Journal of Applied Behavior Analysis, 36, 361–365.measure as a group. Ahearn, W. H., Castine, T., Nault, K., & Green, G. (2001). An Small groups of children have also been shown to assessment of food acceptance in children with autism orrespond to medication intervention. Three children showed pervasive developmental disorder-not otherwise specified. Jour- nal of Autism and Developmental Disorders, 31(5), 505–511.enhanced response to a multidisciplinary feeding disorders Ahearn, W. H., Kerwin, M. E., Eicher, P., & Lukens, C. T. (1996a).program when combined with the medication risperidone, An ABAC comparison of two of two intensive intervention foralthough the authors did not analyze contribution of sepa- food refusal. Behavior Modification, 35, 385–405.rate components of the program (Berger-Gross et al. 2004). Ahearn, W. H., Kerwin, M. E., Eicher, P., Shantz, J., & Swearingin, W. (1996b). An alternating treatments comparison of twoTreatments addressing pain-related organic conditions also intensive interventions for food refusal. Journal of Appliedhold promise in avoiding longstanding feeding concerns Behavior Analysis, 29, 321–332.and/or precluding the need for further treatment. For Allison, D. B., Faith, M. S., & Franklin, R. D. (1995). Antecedentexample, thirteen participants with eosinophilic esophagitis exercise in the treatment of disruptive behavior: A meta-analytic review. 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