Research Protocol Study: Behavioural assessment and treatment to transition children from enteral to oral nutrition Co-ordinating Investigator (CI) details Name Title Address Phone Email Sarah Leadley PhD Candidate School of Psychology Faculty of Science The University of Auckland Bldg 721 Tamaki Campus Private Bag 92019 Auckland 027 931 3551 s.leadley@auckland.ac.nz Principal Investigators (PI) Name Dr. Javier Virues-Ortega Institution University of Auckland Dr. Suzanne Purdy Bianca Gordon University of Auckland University of Auckland Title Senior Lecturer, Applied Behaviour Analysis Programme Head, Speech Science Speech Language Therapist, Professional Teaching Fellow Peer reviewer Name Dr. Timothy Jelleyman Affiliation Waitemata District Health Board, Pediatrics and Newborn Services Title Paediatrician SUMMARY This project aims to use Applied Behaviour Analysis to transition children from enteral (tube feeding) to oral nutrition. We will conduct the study primarily in the home setting with caregivers and their child, in collaboration with the child’s health team. The study will involve the following phases: 1) a structured caregiver interview that focuses on the identification of mealtime conditions under which the child may be more likely to consume food or drink (oral stimuli), 2) an observational assessment involving the manipulation of these conditions, 3) implementation of treatment based on assessment results to increase acceptance of oral nutrition. Once treatment is demonstrated as effective, caregivers will be trained to implement the intervention. Throughout the duration of treatment we will work with the child’s health team with regards to feeding safety, assessment and treatment methods, and enteral nutrition reductions. BACKGROUND A severe feeding disorder is diagnosed when a child is unable, or refuses to eat or drink sufficient quantities to maintain their nutritional needs (Budd et al., 1992). In the most extreme cases, the child must then rely on enteral nutrition (tube feeding). Enteral nutrition is provided by a 1 tube inserted through the nose (Nasogastric, NG tube), or abdomen (Gastrostomy tube, G tube) which runs to the stomach or small intestine (Pearce & Duncan, 2002). A recent survey conducted in New Zealand (Jelleyman, 2013), estimated that there were 630 children aged under 15 (65.9 per 100,000) that relied on home enteral nutrition (i.e., those discharged from hospital) for greater than three months. The initiation of enteral nutrition commonly leads to weight gain and improved nutritional status (Ferluga, Sathe, Krishnaswami, & Mcpheeters, 2014). However, associated complications include aspiration and aspiration pneumonia, infection, and tube dislodgement (Chait et al., 1996; Gomes, Pisani, Macedo, & Campos, 2003). Children who remain dependent on enteral nutrition longer than they medically require continue to be at risk of these complications, and normal oral motor development is disrupted (Babbit et al., 1994). Enteral nutrition also requires ongoing medical involvement, significant costs, exclusion from community activities, and has been associated with significant levels of stress in caregivers (Greer, Gulotta, Masler, & Laud, 2008; Williams, Riegel, Gibbons, & Field, 2007). The cost savings from intervention when compared with long term enteral nutrition have been estimated at $US 32,959.10 for one year, and $US 135,209.80 for five years (Piazza & Carroll-Hernandez, 2004). The reasons for ongoing dependence on enteral nutrition are complex but most often occur because of a medical condition with interacting oral-motor, and behavioural factors (Piazza & Roane, 2009; Rommel, De Meyer, Feenstra, & Veereman-Wauters, 2003). Rommel et al., (2003) examined clinical data from 700 children referred to a hospital-based feeding program (who had all initially received NG nutrition). Of the children, 86% had an identifiable medical problem, in combination with 1) an oral-motor condition (48.5%), 2) a behavioural problem (5.9%), or 3) a combination of all three (5.2%). Medical complications, or lack of oral-motor skills required for eating (e.g., swallowing, sucking) may cause pain or discomfort while eating, resulting in an association between eating and aversive stimulation (Babbit et al., 1994; Hyman, 1994). Because of this aversive stimulation, the child may then develop behaviours such as crying, pushing utensils away, and expulsion. If the caregiver removes the food or ends the meal following these behaviours, they are more likely to occur, and continue when the medical complications are resolved. The child therefore misses opportunities to learn that eating is no longer painful and fails to practice the oral-motor skills required (Piazza & Carroll-Hernandez, 2004). It has been recommended that treatment for feeding disorders should be interdisciplinary, given the interacting medical, oral-motor, and behavioural causes (Brown et al., 2014; Piazza & Addison, 2007). A number of literature reviews have been conducted with regards to the treatment of pediatric feeding disorders. In these reviews, the only studies to meet inclusion criteria were those involving behavioural intervention (Kerwin, 1999; Sharp, Jaquess, Morton, &, Herzinger, 2010; Williams, Field, & Seiverling, 2010). Many of these studies involved participants dependent on enteral nutrition, who were partially or totally transitioned to oral nutrition at the end of the study. Applied behavior Analysis (ABA) is a scientific approach which focuses on improving objectively defined behaviours of social significance. Data is used to demonstrate that the changes in behavior were because of the procedures implemented (Cooper, Heron, & Heward, 2007). With regards to the treatment of feeding disorders, ABA considers the acceptance of nutrition to be a function of antecedents (environmental events that occur before the behaviour), or consequences (environmental events that occur after the behaviour). Interventions based on the manipulation of antecedents (hereinafter, antecedent manipulations) have included modifications to food or liquid 2 properties (e.g., texture, volume), the feeding method (e.g., type or position of utensil), the mealtime environment (e.g., increasing attention or toys available) or the sequence of instructions. Interventions based on the manipulation of consequences have generally included providing reinforcement for the target behaviour, or preventing escape from the oral stimuli (escape extinction) following inappropriate behaviours. The intervention known as “escape extinction” has been the most widely used treatment component and generally involves non removal of the spoon, whereby the spoon is held at the mouth and problem behaviours are ignored until acceptance occurs (Groff, Piazza, Zeleny, & Dempsey, 2011; Patel, Piazza, Martinez, Volkert, & Santana, 2002) or some form of gentle physical guidance in order to open the mouth (Didden, Seys, & Schouwink, 1999; Patel, Piazza, Santana, & Volkert, 2002). The manipulation of antecedents (e.g., modification of food or liquid properties) and the delivery of rewards have also been commonly used, but a limited number of studies have demonstrated their effectiveness in the absence of escape extinction for children dependent on enteral nutrition (Casey, Cooper-Brown, Wacker, & Rankin, 2006; Kadey, Piazza, Rivas, & Zeleny, 2013). Despite this, reinforcement or antecedent manipulation components may serve as an optimal starting point for treatment (Rivas, Piazza, Kadey, Volkert, & Stewart, 2011), reduce subsequent side effects of escape extinction, such as crying and inappropriate behaviours (Patel et al., 2006; Rivas, Piazza, Patel, & Bachmeyer, 2010), and may be more acceptable to caregivers (Bachmeyer, Gulotta, & Piazza, 2013; Rivas et al., 2010). Despite the effectiveness of behaviour analytic intervention in the treatment of feeding disorders, a gap exists in the literature with regards to systematic pre-treatment assessment conducted with caregivers. Whilst the systematic assessment of consequences (Functional Analysis) has been cited in a range of studies, there is yet to be a similar assessment method that evaluates antecedent variables with the same scientific rigor. It is possible that antecedent-based treatment may be effective alone, when matched to the results of a pre-treatment assessment. Kadey et al. (2013) demonstrated that acceptance varied by food texture and type during assessment, and successfully implemented treatment based on these results. An initial treatment package that involves antecedent manipulations can also be viewed as a least restrictive starting point for treatment, and components of greater restrictiveness can be added if required (Borrero, Woods, Borrero, Masler, & Lesser, 2013; Kerwin, Ahearn, Eicher, & Burd, 1995). Kerwin et al., (1995) first manipulated the volume of food presented on a spoon in combination with reinforcement. Whilst escape extinction was required to demonstrate clinically significant results with increased volumes, antecedent assessment served to determine the initial volume that was most likely accepted. We hypothesise that utilising a systematic pretreatment assessment with caregivers and their children may lead to 1) effective least restrictive treatment, or 2) selection of an appropriate starting point that helps to reduce side effects of required restrictive treatment components. ADVANCEMENT OF KNOWLEDGE AND UNDERSTANDING This study aims to advance knowledge of home-based behavioural intervention to transition children from enteral nutrition. We aim to increase the understanding of the role of systematic behavioural assessment in selection of effective treatment methods. We will present our assessment package in a replicable manner so that it may be used by other researchers and clinicians. 3 PROPOSED RESEARCH Specific objectives Study 1. Caregiver interviews (See p.5) Conduct a structured caregiver interview. We will select a series of antecedent or consequence dimensions that are reported to influence acceptance and/or inappropriate behaviour to test in assessment. If the caregiver reports the child to refuse all oral nutrition, we will collaborate with the child’s health team to determine introductory test conditions. We will also conduct assessment of the child’s preferences with regards to toys or activities. Identified preferred items are necessary to maintain the participants’ motivation throughout the duration of the study. Study 2. Assessment (See p. 6) Conduct an experimental assessment of antecedent manipulations. Following undifferentiated results, we will add consequence manipulations, by asking caregivers to change their responses to the child’s behaviour. Following differentiated assessment results, we will develop an intervention that is matched to those results. Study 3. Experimental Treatment Evaluation (See p. 8) We will conduct and evaluate the matched intervention. Following clinically significant results, we will train caregivers to implement the treatment package with regular monitoring by the CI. If we do not observe clinically significant results we will return to assessment and determine additional treatment components. Following this we will evaluate the combined treatment package. PROPOSED METHOD Research Design The study will involve single-subject design, where each participant will serve as their own control. Repeated measurement of target behaviours will occur throughout the study. Each participant’s data will be graphed and analysed separately. Single-subject experimental designs are the highest scientific standard for the experimental evaluation of assessment and treatment procedures in applied behaviour analysis. This methodological approach is also highly suitable to this study because (a) it facilitates clinical decisionmaking, and (b) allows for the experimental demonstration of treatment effects in participants with rare clinical conditions (e.g., tube dependence). Participants We will initially recruit 3 child-caregiver dyads (a total of 6 participants, 3 children, and 3 adults). We may recruit further participants, if time is made available by participant/s in the first group meeting termination criteria. The maximum number of participants expected in this study would be 6 childcaregiver dyads (a total of 12 participants). Participants will be recruited via advertisements placed at District Health Board locations, or professional networks. Children will be included in the study if they meet the following criteria. 4 These criteria will be implemented based on information gathered from the caregiver and health professionals. - - - Aged between 1 year and 16 years old and reside within the Auckland area Living at home or in a long-term care placement with a primary caregiver that is available to be present for mealtime sessions Dependent on enteral nutrition for at least six months. There may be a range in the degree of dependence and the scheduling of enteral nutrition (e.g., 50% of daily needs, feeding at night only) The support of professionals that are involved in the child’s health Confirmation from a paediatrician with regards to : o Stability or resolution of the original medical problem contributing to the initiation of enteral nutrition o No pending medical interventions (e.g., surgery) that could affect feeding ability o Absence of anatomic or functional impairment precluding safe oral feeding o Maintenance of clinically safe weight Confirmation from a Speech Language Therapist with regards to: o Safe liquid swallow Procedure Study 1. Caregiver interviews 1) Pediatric Feeding Assessment Interview. The structured interview (see document 4a) will be completed with caregivers. The survey is designed to elicit information about potential antecedents and consequences that influence the child’s feeding behaviour. Consent will be gained to access relevant paediatric reports to identify previous and current medical diagnoses and oral motor skill. Specific details will be collected with regards to liquids/foods (oral stimuli) previously or currently accepted (antecedent dimensions), as well as caregiver responses to the child’s inappropriate mealtime behaviour (consequence dimensions). 2) Pediatric Inventory for Parents. This self-report questionnaire has been considered well established at assessing family functioning in the context of chronic childhood medical problems. The questionnaire will also be administered following treatment. 3) Preference assessment. An interview, The Reinforcement Assessment for Individuals with Severe Disabilities (RAISD, see Document 4b) will be used to determine the preferences of a participant. This interview tool will be completed by a caregiver, or child where able. This interview provides information on useful reinforcers for the individual. We will then conduct a paired-stimulus preference assessment where we will record the participant’s frequency of approach to items when presented in varied pairs. The most preferred items and activities will be used during the assessment and treatment, and to develop rapport with the participant Study 2. Assessment 5 Assessment will take place within the family’s regularly scheduled mealtimes (i.e., breakfast, lunch, dinner, and snacks). We will follow recommendations from the child’s health team with regards to appetite regulation, such as scheduling meals 2-3 hours following the child’s last EN feed. Assessment will consist of blocks of 10-trials (conditions), whereby each trial involves the presentation of an oral stimulus. Assessment will continue for as long as the mealtime allows, or for a maximum of 30 min. The child’s health team will be consulted with regards to safety criteria prior to assessment that would warrant immediate termination. These criteria may include the physical condition of the participant (e.g., fatigue), and likely feeding related complications (e.g., vomiting, choking). The caregiver may also choose to terminate sessions at any time. In addition, the assessment session will be terminated in the following situations: - Inappropriate behavior occurs at an intensity severe enough to present immediate physical harm to the participant or others present (e.g., hitting, kicking, biting, property destruction, throwing objects). - Low severity inappropriate behaviour occurs for a duration longer than 10 minutes (e.g., verbal refusal, turning head away or leaving mealtime area and refusing to return). More severe behavior (e.g., property destruction) may result in session termination within less than 10 minutes. The assessment will involve both test, and control conditions. Test conditions will be those identified during the caregiver interview. Example conditions are presented in Table 1. If the child was reported to refuse all oral stimuli, we will consult with the child’s health team and developmental guidelines to determine conditions. Table 1: Example assessment conditions Interview summary Dimension Antecedent A Control A Antecedent B Control B Participant 1 Accepts teaspoon of banana, apple, potato puree Condition Volume (Chopped fine texture) 1 ¾ teaspoon 2 ½ teaspoon 3 ¼ teaspoon 4 Empty spoon Teaspoon Texture (Teaspoon volume) 1 Chopped fine 2 Wet ground 3 Puree Table Participant 2 Accepts formula/water in baby bottle Volume (Formula/Water in bottle) 40 ml 30 ml 20 ml 10 ml 60 ml formula/water Food type (In bottle, 10 ml) 75% rice cereal, 25% formula 50% rice cereal, 50% formula 25% rice cereal, 75% formula Puree rice cereal General texture definitions. Puree - Blended smooth, no lumps Wet Ground – Blended to liquid medium with small chunks Chopped fine –Regular texture, chopped to pieces < 2mm Table – Regular texture, chopped to pieces < 1cm Antecedent test conditions. Antecedent dimensions (and levels of manipulation) will be evaluated against a matched control condition. If the child is reported to accept a range of liquids or foods, we will randomize those items across trials (e.g., apple, banana, pear puree). 6 During a test condition, a trial will start when the caregiver provides a verbal instruction and presents the oral stimulus. The oral stimulus will be held or positioned stationary in front of the child for 5-s. If the oral stimulus is accepted, the caregiver will provide praise, and after 30-s has elapsed, the child’s mouth will be checked. If the child’s mouth is clean, praise will be provided. If the oral stimulus is not accepted, the caregiver will withdraw the stimulus and make no comment. A brief break will occur between all trials. If inappropriate behaviours occur during the break, the following trial will be delayed until behavior has ceased for at least 3-s. There will be no other consequences provided for acceptance, refusal, expulsion, or other problem behaviours. Caregivers will be instructed to ‘act as you normally would’ and will be able to present instructions, praise, and attention in a manner of their choosing. During the assessment the child will also have access to preferred items that were identified in the preference assessment during Study 1 (e.g., Ipad movie). All assessment conditions will be videotaped to check for any variability in caregiver behavior across sessions. Antecedent control conditions. The control condition will correspond with the preceding test conditions, whereby oral stimuli will be presented in an age-typical manner with respect to the antecedent dimension. For example, the control condition for volume would involve oral stimuli presented in the same texture, and feeding apparatus, but at age-typical volume. This may require modifications to the feeding apparatus (e.g., enlarging the nipple of baby bottle to allow for increased texture). We will collaborate with the child’s health team with respect to exact stimuli for control conditions and feeding safety. All other procedures for the control condition are identical to test conditions. Consequence test conditions. If we do not observe differentiated results when manipulating antecedents, we will add consequence-based test conditions, based on information reported in the caregiver interview. During these conditions we will manipulate caregiver responses to inappropriate behavior, whilst keeping antecedent dimensions the same across all conditions. We may include test or control conditions that involve manipulating the following responses to inappropriate mealtime behavior: 1) caregiver provides attention, 2) caregiver removes oral stimulus, and 3) caregiver provides preferred food or toy. Consequence control conditions. Consequence control conditions will correspond with the corresponding test condition. For example, the control for the attention condition would involve providing high rates of attention, regardless of inappropriate behavior. Data analysis. Conditions will be compared to assess whether a particular antecedent or consequence results in more differentiation of acceptance or problem behaviour when compared with control conditions. Those that show the most differentiation will be matched to an intervention. Study 3. Experimental treatment evaluation We will recommend an intervention based on assessment results. We will ensure that the treatment recommendation is acceptable to the family and consistent with other ongoing interventions by the child’s health team. A goal for intervention will also be determined. This goal will include the target 7 oral stimuli (e.g., to consume an age-appropriate portion of pureed textures), as well as associated nutritional outcomes (e.g., for oral consumption to increase to 50% of daily requirements). Caregivers will be asked to provide written consent for the proposed treatment plan. Intervention sessions will initially be conducted by the CI, with the caregiver observing. The procedure of treatment sessions will be identical to that described for the experimental assessment (Study 2). However, oral stimuli will be presented in 10-trial blocks and sessions will continue to run for up to 30 min, or until an age-appropriate portion of oral stimuli is consumed (recommended by the child’s health team). Termination criteria will be the same as during assessment, with the exception of escape extinction (see b). Assessment results will serve as the initial baseline of acceptance and/or inappropriate behaviour. We will also return to baseline conditions via baseline probes (block of 10-trials), regularly throughout treatment. a) Antecedent-based intervention. Intervention will involve progressively altering antecedents to increase acceptance of oral stimuli. We will continue to progress until the predetermined target stimuli. Our starting point will be the condition associated with the highest level of acceptance in assessment. Treatment examples could include one or a combination of the following progressions: - Volume (e.g., empty teaspoon to full teaspoon) - Texture (e.g., puree to chopped fine food) - Feeding method (e.g., baby bottle to a spoon) - Food preference (e.g., blending non-preferred food with preferred food) - Feeding-related instructions (e.g., instructions with high compliance to instructions with low compliance) Criteria for advancing treatment levels. The general criteria for advancing levels of antecedent manipulation will be three consecutive sessions in which acceptance and mouth clean occur in more than 80% of trials, and inappropriate behaviours occur in less than 20% of trials. The participant may also advance levels faster if they meet the same criteria during a baseline probe. There may be flexibility within these criteria, based on the individual participant. Implementing caregiver training. The general criteria for implementing caregiver training will be stable treatment progress (i.e., the child has progressed through at least two levels as above), and caregiver readiness. Mastery criteria. The general criteria for mastery of the child’s target oral stimuli will be three consecutive sessions in which acceptance and mouth clean occur in more than 80% of trials, and inappropriate behaviours occur in less than 20% of trials. Further assessment and additional treatment components. If data do not demonstrate a continued increase in acceptance or reduction in inappropriate behaviour relative to baseline in at least three consecutive sessions we will return to assessment conditions to determine a 8 consequence-based treatment component. treatment. This decision may be made at any point during b) Consequence-based intervention Consequence-based intervention will be conducted in the same manner as a). We will first add a reinforcement component, followed by escape extinction dependent on the treatment criteria as presented in a). Reinforcement. We will use differential reinforcement procedures whereby we will provide reinforcement for consumption of the oral stimuli, whilst providing no reinforcement for any inappropriate behaviours. The items used as reinforcers for each child will be based on the initial preference assessment conducted in Study 1, as well as any oral stimuli that the child is regularly consuming (e.g., if previous intervention has served to establish consumption of pureed foods). Following the consumption of the oral stimulus, we will provide access to the preferred food or item (for a period up to 15 s). Escape Extinction. The escape extinction method will be determined with caregiver and health team input, but will take either of the following forms if the oral stimulus is not accepted within 5-s. - Nonremoval of the spoon – the oral stimulus will be held near the child’s mouth - Physical guidance – gentle pressure will be applied the child’s jaw to allow the oral stimulus to be placed in the mouth With either method, the procedure will continue until the child accepts the oral stimulus, or until 30 min has elapsed. Safety criteria will be the same as that applied during the assessment, with one exception. Inappropriate behavior that occurs beyond 10-min duration will not result in session termination. c) Caregiver training The completion of training will allow caregivers to continue intervention procedures without direct supervision by the CI. Training will involve the following components: 1) Caregiver reviews written procedures and data collection 2) Caregiver observes CI-led meals and records data 3) Caregiver role plays procedures with CI 4) Caregiver implements intervention with child under CI supervision 5) Caregiver implements intervention without supervision During all phases caregivers will have opportunity to ask questions and receive feedback from the CI. Once a caregiver meets an accuracy criterion, they will progress to the next training phase. During the final phase, caregivers will be required to send video of one session per week to the CI, and the CI will continue to visit the family on a minimum fortnightly basis. The CI will provide feedback to the caregiver, based on the mealtime video, and that observed in vivo. Study Termination Criteria 9 The study will terminate for a participant when data reach mastery criteria at the target oral stimuli and when associated nutritional outcomes are achieved, or at 12 months. The entire study will terminate when this occurs for six parent-child dyads, or by January 31st, 2017. The study will be terminated early for an individual participant given the following: - The child no longer meets inclusion criteria during the course of the study (for example, a medical complication arises which prevents safe eating). - The physical condition of the child or level of behaviour requires termination of three consecutive assessment or treatment sessions (see safety criteria, p. 6) - The caregiver requests withdrawal The entire study will be terminated, and redesigned if the intervention matched to assessment does not result in clinically significant results in three participants. Outcome measures Outcome measures will include observed behavioural data collected during mealtime sessions, and volume of oral nutrition consumed. Data during mealtimes will be collected by in a format specific to the CI or caregiver. Data recorded by the CI or other trained observers will be trial-by-trial and include acceptance (taking food into the mouth) and mouth clean (no food present in mouth 30 s after acceptance), as well as the presence of food refusal behavior specific to the child (e.g., expulsion). Measures will be reported as percentage of trials. Volume of liquid/food consumed (grams or ml) will also be calculated for each meal, and members of the child’s health team may then calculate further measures relevant to enteral nutrition reductions (e.g., converted to calories). The child’s weight will also be recorded at a frequency as recommended by the health team, or at least weekly. We will also administer the Pediatric Inventory for Parents following treatment, and compare results with those obtained before treatment. Data recorded by caregivers will consist solely of volume per type of food/liquid consumed, as well as a rating of food refusal behavior within each mealtime. When present, the CI will use the same behavior rating scale in order to monitor data reliability. The CI will use ABC Data PRO to collect data. It is a software application for handheld computers (e.g., IPhone touch) that is well suited for behavioural observation procedures. The application allows the observer to record up to nine events concurrently operating nine independent buttons on a touchscreen. Each button is assigned either a participant behaviour (e.g., acceptance, mouth clean) or the caregivers’ behaviour (e.g., starting the trial by presenting the food). For instance, if button 1 is called “trial start”, the researcher will be required to press this button when the caregiver presents oral stimuli to the child. Once caregiver training commences, data will also be recorded with respect to caregiver implementation of the intervention. For each intervention component (e.g., ‘present food at child’s lips for 5 seconds’), caregiver behaviour will be scored as ‘correct’ or ‘incorrect’ and data will be summarised as the percentage of components implemented correctly. To evaluate the reliability of the data we will use video recordings of at least 20% of assessment and treatment sessions to measure interobserver agreement. Interobserver agreement involves 10 calculating the degree to which two observers record the same events, and reporting this as a percentage of agreement. References Babbit, R. L., Hoch, T. A., Coe, D. A., Cataldo, M. F., Kelly, K. J., Stackhouse, C., & Perman, J. A. (1994). 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