Evidence to support treatment options for children with swallowing and feeding disorders: A systematic review

3.1 Clinical Question 1 (Oral Motor)

One of the 61 included studies examined clinical question one [21]. The 20 participants in this experimental, randomized control trial study had profound intellectual disability or neuromuscular disorders, lived in a residential care facility, and were identified by the residential staff as being problem feeders. The authors implemented an oral motor program that included manual intervention, direct guidance, stretches, pressure, and oral desensitization administered 30–40 minutes daily, five times a week for nine weeks for the experimental group ( n = 10). The control group continued with their previously prescribed therapeutic plan of care. Only nine subjects from the experimental group and two from the control group had both pre and post treatment evaluations of oral motor functioning. Analysis of the nine experimental subjects that had pre and post treatment oral motor function testing revealed no significant change following treatment. Statistical analysis of the full sample ( n = 20) revealed no significant difference in body weight gains between the experimental and the control group at discharge [21]. See Table 3 for details.

3.2 Clinical Question 2 (Sensory)

None of the 61 articles included examined only sensory-based interventions.

3.3 Clinical Question 3 (Behavioral)

Thirty-seven of the 61 included studies examined clinical question three [22, 23, 24, 25, 26, 27, 28, 29, 30, 31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41, 42, 43, 44, 45, 46, 47, 48, 49, 50, 51, 52, 53, 54, 55, 56, 57, 58]. Of 919 participants across 37 studies, a majority of participants (86%) were between two and seven years old [22, 23, 24, 25, 26, 27, 28, 29, 30, 31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41, 42, 43, 44, 45, 46, 47, 48, 49, 50, 51, 52, 53, 54, 55, 56, 57, 58]. Overall sample sizes were small (1–13 participants) [23, 26, 27, 28, 30, 31, 32, 33, 35, 36, 37, 40, 42, 43, 44, 45, 46, 47, 49, 51, 52, 53, 54, 55, 56, 57, 58], however some studies had larger sample sizes ranging from 24 to 490 participants [22, 24, 25, 29, 34, 38, 39, 41, 48, 50]. The majority of the reports examined the prescribed intervention’s impact on volume and variety of oral intake as well as weight gain. The studies varied greatly in reported frequency and duration of treatment. One study, the largest study identified by this systematic review, did not specify the types of behavioral interventions used in their protocol and instead assessed the impact of unspecified behavioral therapy techniques on outcome success and cost in a retrospective chart review of 490 participants [24]. The results are presented with subheadings in order of behavioral techniques utilized in the articles: positive reinforcement and extinction, stimulus shaping and fading, caregiver involvement and education, and operant conditioning and systematic desensitization.

3.4 Positive reinforcement and extinction

Eleven of the 37 studies reported on the use of escape extinction (behavioral technique used to treat behaviors that are maintained by escaping or avoiding; removing the possibility of escaping from the challenging stimulus) and reinforcement (the use of any stimulus which should increase the probability of a desired response, can be positive or negative) in single-subject and small cohort designs and their impact on acceptance, intake, and presence of inappropriate behaviors at mealtime [23, 26, 27, 28, 30, 31, 32, 33, 36, 40]. Patel and colleagues reported that differential reinforcement (advantages positive reinforcement to distinguish appropriate behavior from inappropriate behavior; used to increase appropriate behavior and decrease inappropriate behavior) alone was not effective for increased oral intake, but escape extinction in combination with differential reinforcement did increase oral intake and mouth clean (upon inspection, no visible food or liquid in the subject’s mouth following intake without expulsion) in a small sample ( n = 3) [42]. Larue et al. similarly found that using escape reinforcement (negative reinforcement technique, utilizing escape) alone did not increase mouth clean or decrease inappropriate behaviors; change in these behaviors was dependent on the application of escape extinction regardless of the presence or absence of reinforcement ( n = 5) [35]. Penrod and VanDalen corroborated the need for escape prevention in the reduction of problematic hyper food selectivity and reported that increased acceptance of non-preferred foods depended on escape prevention in the form of non-removal of the spoon plus re-presentation of expelled bites ( n = 3) [43]. Bachmeyer et al. found participants decreased inappropriate mealtime behaviors and increased acceptance of food utilizing an escape and attention extinction (removal of ability to escape and removal of attention) combination ( n = 4) [23]. Wilkins and colleagues also found that a chin prompt (gentle pressure applied in upward motion to child’s chin and lower lip by the feeder/therapist) in combination with representation of expulsion per bite decreased rates of expulsion and increased volume consumed ( n = 4) [57]. Pizzo et al. examined the effects of an intensive 4–5 day behavioral intervention that included meal and taste probing sessions with escape extinction for a small cohort ( n = 3) and found there was an overall increase in the volume and variety of foods eaten and a decrease in inappropriate mealtime behaviors that was maintained at 1-month post treatment [44]. Gale et al. reported that non-contingent negative reinforcement (positive reinforcement not related to the child producing the target behavior; reinforcement delivered on a predetermined time schedule), positive reinforcement and extinction (disappearance of a learned behavior when that behavior is not reinforced) of disruptive behavior lead to a decrease in undesirable behavior during mealtime and an increase in volume of food eaten in a small cohort of toddlers with autism spectrum disorder ( n = 3) [30].

Blisset and Harris described the effects a highly structured, behaviorally based intervention program that integrated positive and negative reinforcement, self-feeding, parent education, structured mealtimes, and repeated presentation of novel foods in their case study report. Improvements in variety and volume of oral intake, reduction in undesirable behaviors associated with feeding, improved parent-child interactions during mealtimes and increases in both height and weight were reported after completing the intervention and at specified follow-up points, up to three years post treatment [27]. Volkert and colleagues utilized reinforcement, escape extinction, and caregiver training in an outpatient treatment program to enhance mastication and self-feeding skills for three study participants and found mastication, mouth clean, and acceptance increased with early swallow decrease for all subjects [56]. Rivas et al. looked at self-feeding skills utilizing a similar therapeutic approach (reinforcement, escape extinction, caregiver training) to enhance self-feeding skills. They reported that although the probability of self-feeding increased for two subjects as the number of caregiver-fed bites increased, the remaining subject did not self-feed consistently, even when the ratio of self-fed to caregiver-fed bites was 1:5 [47]. Greer et al. conducted consecutive studies in which weight gain and intake were monitored in both a child who had never swallowed food or liquid secondary to dysphagia and a child who chronically refused food following application of treatment with positive reinforcement and peer modeling. These authors reported an increase in the volume and variety of accepted foods, as well as significant weight gain with the participant in study one having weaned from tube feeding by two months’ post treatment [31].

3.5 Stimulus shaping and fading

Eleven of the 37 articles reviewed examined shaping (behavior training that uses differential reinforcement of successive approximations of the desired behavior) and stimulus fading (initial prompting to perform behavior is gradually withdrawn, ensuring the child does not become dependent on the stimulus) of placement, texture, taste and quantity combined with ranging levels of reinforcement to eliminate food refusal or discontinue tube feeding [28, 29, 32, 33, 36, 37, 46, 48, 49, 51, 58]. De Moor et al. reported generalization of oral acceptance in their case series for a variety of food items during follow up at three to twelve months with tube feeding discontinued in each subject ( n = 5) [28]. Luiselli and Luiselli reported clear increases in oral intake with the reduction of gastrostomy-tube feedings and addition of oral feeding meals in their case study [37]. Luiselli and Luiselli used visual cueing, demand fading and positive reinforcement with a subject who was weaned from tube feeding to full oral feeding and gained more than two pounds from pre-treatment [36]. In a retrospective analysis, Schadler et al. examined the effect of positive reinforcement, escape extinction, and flooding (rapid and excessive exposure to a feared stimulus) on oral intake with 86 ex-premature participants with feeding disorders and found that ∼ 60% of them presented with some improvement in oral intake [48]. Seiverling et al. found an increased acceptance of bites (50% at baseline, at least 86% at follow up in single subject design) and a decreased number of disruptive responses from children in their small study ( n = 3) [49].

Groff et al. and Rivas et al. had similar findings assessing acceptance, mouth clean, and inappropriate behaviors during mealtime in single subject and small cohort designs [33, 46]. For Groff et al. acceptance was 0% during the reversal to baseline with both a spoon and cup. During fading steps: acceptance and mouth clean remained high and inappropriate behavior remained low ( n = 1) [33]. For Rivas et al., fading plus escape extinction resulted in low rates of inappropriate behaviors and high rates of acceptance (Study 1: n = 2/ Study 2: n = 1) [46]. Groff et al., looked to increase acceptance and mouth clean and decrease inappropriate meal time behaviors with extinction, syringe fading and planned ignoring in their later case study; baseline mean for inappropriate behaviors decreased from 31 to 3.8 per minute following extinction intervention. Acceptance and mouth clean increased to 98% given trials across five meal blocks daily [32].

Wilkins et al., combined escape extinction techniques (including non-removal of a spoon), bite representation, and a Nuk brush to increase mouth clean and acceptance, and decrease packing (holding food in the mouth and cheeks) and expulsions per bite in 12 subjects with generalized feeding complaints, developmental delays and G-tube dependency. All participants transitioned from a Nuk brush to a spoon during either an intensive outpatient or day-treatment admission or during outpatient follow up. Their findings suggest that utensil manipulation during initial treatment of children with severe feeding problems may be warranted [58].

Sharp et al. found retrospectively that using a combination including stimulus fading and non-contingent access provided significant improvements regarding food variety, consumption, and appropriate mealtime behaviors in a case series report ( n = 13). They found that all participants increased their overall intake by at least 12 foods (relatively distributed across all food categories) and treatment was effective in reducing mealtime stress during food presentations [51]. Dovey and Martin found that all subjects ( n = 24) increased in height, weight, and BMI when they utilized a program that incorporated behavioral principles in teaching, parent education, and concurrent presentations of preferred foods with target foods [29].

3.6 Caregiver involvement and education

Thirteen of the 37 studies focused on parent education and involvement in combination with behavioral strategies such as reinforcement, escape extinction and fading [22, 25, 26, 34, 39, 40, 41, 45, 50, 52, 53, 54, 55]. Half of these reported on findings from case studies. Vaz et al. reported an increase in the intake of pureed foods and an increase in self-feeding in a case study with eventual parent training and carryover [55], while Najdowski et al. reported an increase in the variety of novel foods accepted when using these techniques mediated by parent involvement and in-home therapy [40]. Similarly, Binnendyk and Lucyshyn reported increased variety and calorie intake of previously accepted and recently rejected foods as well as a decreased reliance on Pediasure TM , a liquid nutritional supplement, with implementation of a family-centered positive behavior support program in the home [26]. Tarbox et al. provided parent training regarding non-removal of the meal and preparation of meals without consideration of child selectivity. This single subject design found a significant increase in volume and variety of food consumption following the second implementation of the intervention with a secondary decrease in the length of meal times down to below 30 minutes at follow up [54].

Stark and colleagues prospectively implemented parent education provided by clinical psychologist, pediatric dietician, and physician with personalized nutritional and behavioral management strategies, including relaxation strategies and token reward system to improve feeding in three children with cystic fibrosis. Reported outcomes included meeting calorie goals at meals and average weight gain of 0.66 kg. Utilizing positive and negative reinforcement, relaxation techniques, and parent education that included nutritional education and behavior management training [52], Stark et al. reported increased participant calorie intake ( n = 9) and significant weight gain as compared to a control group over six weeks of treatment with generalization reported at six month follow-up studies [53]. Powers et al. utilized a randomized controlled design to target increased caloric intake and nutritional content with nutritional counseling and child behavior management skills with the goal of meeting 120% recommended daily allowance (RDA) per day or greater. No one met the goal at baseline but following treatment 100% of the participants in the experimental, behavioral group met the goal and only 16% of the control group (randomized to a waiting list) met the goal [45].

Sharp, Burrell, and Jacquess introduced the Autism MEAL Plan to address caregiver training for families of children with ASD ( n = 19) and included didactic workshops for skill acquisition which targeted eight key subjects of parent education: introduction, structuring meals and monitoring behaviors, ways to increase appropriate behavior, effective communication, ways to decrease inappropriate behavior during meals, methods of introducing foods, teaching self-feeding skills, and monitoring and maintaining progress. Following treatment, caregivers reported a significant reduction in overall stress and large effect sizes ( d = 1.1) were seen on the Parenting Stress Index short form scores [50]. Johnson and colleagues utilized a larger sample of families ( n = 14) to implement a highly structured intervention package which included individual (not group) parent training (modeling, role play, and giving clear instructions, mealtime plans, reinforcement, shaping and fading) with video modeling and feedback used for caregiver skills acquisition. Reported results included statistically significant decrease in disruptive mealtime behaviors across assessment time points and significantly reduced amounts of reported parent stress [34].

Four of the remaining studies that examined the effects of parent training utilized larger cohorts (combined n = 271) [22, 25, 39, 41]. Adamson et al. found in their randomized control trial that parents who received intervention with their children reported significant improvements in mealtime behavior and in their parenting styles when compared to controls ( n = 96). The intensity of disruptive child behaviors, general parenting styles, and parental efficacy were also significantly improved suggesting generalization of program skills outside of meal time with these changes observed to be present and reliable at a 6-month follow up [22]. Opipari-Arrigan et al. reviewed the effects of parent training on behavioral interventions versus the effects of nutritional education alone ( n = 67) and found that behavioral intervention education plus nutritional counseling (Be-In-CHARGE!) resulted in significantly greater improvements in volume of oral intake and calories with more weight gain over 9 weeks versus nutritional education alone [41]. Similarly, Benoit et al. (2000) reviewed the benefits of nutritional counseling versus the benefits of behavioral technique training in the reduction of tube-feedings utilizing a randomized controlled trial ( n = 64). At the third follow-up visit, 15 (47%) of 32 patients in behavioral therapy and none of the 32 subjects (0%) in nutritional therapy were no longer dependent on tube feeding, indicating behavior therapy is more efficacious in eliminating the need for tube feeding than nutritional counseling alone [25]. Morawska et al. used a controlled group discussion and in-home workbooks to educate parents on hassle free mealtimes ( n = 44) and found that parents reported a significant decrease in challenging mealtime behaviors and length of meals with an increase in volume and variety of foods eaten at six months post education [39].

3.7 Comparison of operant conditioning approach with parent education to systematic desensitization approach with parent education

Marshall et al. conducted a randomized controlled trial to compare the effectiveness of operant conditioning (learning approach that utilizes consequences to control behavior; key components include positive and negative reinforcement and positive and negative punishment) and systematic desensitization (learning approach that engages the patient in some type of relaxation exercise and gradual exposure to increasing levels of an anxiety producing stimulus like food) approaches, both provided with a parent education component, to determine their impact on specific primary outcomes of dietary intake, dietary variety, and problem feeding behaviors. The authors found significant improvement in all primary outcomes ( 0.05) with small to medium effect sizes when groups were combined and compared with pre and post treatment measures; however, they did not find significant differences in outcome measures between the two behavioral treatment conditions.

3.8 Behavioral and oral-motor interventions

Twelve of the identified 23 studies featured a combination of behavioral and oral-motor interventions (combined N = 30) [62, 64, 65, 66, 67, 69, 72, 73, 74, 75, 77, 80]. All of the studies identified utilized case study or single subject designs to describe the effects of previously discussed behavioral techniques (such as escape extinction, reinforcement, stimulus fading, and shaping) in combination with oral motor principles, techniques, or tools and described their combined impact on oral feeding outcomes in heterogeneous pediatric populations [62, 64, 65, 66, 67, 69, 72, 73, 74, 75, 77, 80].

Harding, Faiman, and Wright targeted oral versus gastrostomy tube (g-tube) intake ( n = 3) with intensive parent coaching regarding textures, desensitization, and problem mealtime behaviors and reported g-tube feedings were significantly reduced or eliminated with an overall increase in positive parent behavior and meal time situations in 67% of patients (two of the three subjects in the study) [67]. Sharp et al. assessed the reduction of problem behaviors during meals and improved ability to maintain bolus within the oral cavity in one subject with problem feeding behavior and oral motor dysfunction due to diagnosis of cerebral palsy [74]. Data from their case study showed reduced rates of expulsions and increased mouth cleans with adaptation of presentation to accommodate for existing oral motor dysfunction in addition to escape extinction and negative reinforcement [74]. Utilizing similar approaches, Eckman et al. improved variety and texture of food eaten in 2 male participants by implementing a structured approach to teach chewing in combination with behavioral strategies [64]. Both participants in their report were chewing with at least 80% accuracy by discharge from therapy [64]. In a report from Vaz and colleagues escape extinction in the form of non-removal of the spoon and use of a preferred food or liquid chaser following food acceptance ( n = 3) decreased packing and increased mouth cleans [80]. Qvarfordt et al. utilized guided eating (including physical and verbal prompts) to successfully increase oral coordination and participation in mealtimes in three subjects with Rett syndrome [73].

Clawson and colleagues reported on the effectiveness of the Beckman Oral Motor Program with escape extinction, reinforcement, and parent training on food acceptance, mouth clean, negative vocalizations, inappropriate behaviors and caregiver interactions for eight subjects [62]. At the end of treatment, improvements were reported for mouth opening at the presentation of food, timeliness of managing food in the mouth, toleration of longer meals, and acceptance of larger volumes of food per meal. Inappropriate behaviors were reduced and caregivers demonstrated increased ability to successfully feed their children with improvement in ideal weight seen in follow up sessions [62]. A case study from Sheppard utilized incidental teaching and positive reinforcement to teach oral motor concepts and improve sensory tolerances for eating with daily practice at meal and snack times over a two year period [77]. She reported success in the development of functional feeding skills with age appropriate textures [77].

Girolami et al. [66] and Gibbons et al. [65] documented use of a Nuk brush in an oral motor condition to increase tactile stimulation and escape extinction. Girolami et al. reported that presenting and re-presenting food with the oral motor tool (Nuk brush) was more effective in decreasing expulsions than using a spoon; however, success of the brush may be due to negative reinforcement [66]. In contrast, Gibbons et al. attributed the weaning of gastrostomy tube feeding and the elimination and tongue thrust in their subject to the use of the combined strategies, oral-motor and behavioral procedures [65]. Sharp and colleagues recruited three subjects that were all gastrostomy tube dependent and implemented a behavioral program with variable spoon presentations to assess its impact on reducing undesirable mealtime behaviors, mouth clean and expulsions [75]. The authors reported that the behavioral program was effective in decreasing inappropriate mealtime behaviors and increasing acceptance of bites; however, variable spoon presentations were necessary to reduce the level of food expulsion and increase mouth clean [75]. Penrod, Gardella, and Fernand found that antecedent-based strategies increased food consumption in the absence of escape extinction for the two subjects in their study with feeding disorders and ASD [72]. Levin et al. utilized redistribution, swallow facilitation, and swallow chaser techniques to increase mouth clean and decrease packing in children with ASD ( n = 2) [69].

3.9 Behavioral and sensory intervention

Four of the 23 studies utilized principles and techniques from sensory integration in combination with behavioral intervention strategies [59, 63, 70, 78]. Luiselli and Gleason combined sensory reinforcement in the form of a light, rocking motion, and texture fading to overcome chronic food refusal ( n = 1) [70]. Outcome measures included acceptance and nonacceptance behaviors of solid foods; at baseline, acceptance of solid foods was 0%. During treatment acceptance increased to above 80%. Acceptance decreased during month follow-ups, but never fell below 80%. As Luiselli’s et al. participant was a 4-year-old child with visual and hearing deficits, these findings support the notion of including sensory reinforcement techniques in treating food refusal particularly in children with documented sensory deficits [70].

Addison et al. combined individualized sensory integration therapy with escape extinction and noncontingent reinforcement as a way to increase acceptance and grams consumed and decrease inappropriate mealtime behaviors for two young children with poor nutritional intake and growth [59]. Results of functional analyses favored escape extinction plus noncontingent reinforcement for increasing acceptance, decreasing inappropriate behavior, and increasing amount of calories consumed [59]. Douglas and Harris reported on a large sample ( n = 73) of children treated in an outpatient-feeding clinic with a broad approach that included systematic desensitization, operant conditioning, family therapy, positive reinforcement, appetite manipulation, sensory play, and dietary advice to reduce the need for tube feeding, improve behavior during mealtimes and increase weight percentiles, grams consumed, and caregiver satisfaction. The authors reported success in outcome measures based on individualized tailoring of the vast therapeutic approaches utilized in their clinic [63].

Most recently, Suarez retrospectively reviewed the use of sensory integration and systematic desensitization in combination with escape extinction, positive reinforcement, and parent training within a home therapy program ( n = 4) [78]. Acceptance of non-preferred foods increased. All participants showed a positive trend in food acceptance over a relatively small number of treatment sessions (1–10) and were discharged after caregivers reported that they were able to facilitate their child taking at least 1 bite of every family food at every meal [78].

3.10 Behavioral and pharamceutical intervention

Two of the 23 studies reviewed the effects of pharmaceutical intervention in combination with behavioral therapy techniques in single subject designs [18, 60]. One of the two studies looked at the use of Risperidone in three children with feeding disorders [60]. While outcomes varied, all of the subjects ( n = 3) showed improvement in oral intake with at least partial reduction in calories administered via tube feeding [60]. Similarly, Davis et al. utilized pharmaceutical treatment (Gabapentin and/or amitriptyline, megesterol) plus hunger provocation to assess weight gain and percent of oral intake ( n = 9) [18]. Eight of the nine subjects maintained weight and never required additional tube feedings following treatment [18].

3.11 Behavioral, Oral Motor, & Sensory interventions from multidisciplinary teams

Five of the 23 articles reviewed intervention techniques that involved a multidisciplinary approach to improving feeding outcomes [61, 68, 71, 76, 79]. Clawson and colleagues utilized a large team of providers including occupational therapy, speech therapy, feeding technicians, dieticians, nutritionists, gastroenterologists, nursing staff, and a social worker to target four main components during the prescribed intervention that included oral motor exercises, structured meals, behavioral interventions and caregiver training [61]. In this small study of three children with Goldenhar Syndrome, the authors reported a 36% increase in acceptance of food and drink, 16% decrease in the incidence of expulsion during mealtime, approximately 88% increase in mouth clean, and a 56% decrease in inappropriate mealtime behaviors following discharge from this multidisciplinary feeding program [61].

Trabi et al. retrospectively examined the impact of a program that utilized hunger promotion by reduction of tube feeding in a supervised setting, including an intensive, noninvasive monitoring of the child’s medical condition and full support of the child’s capacity for autonomous food exploration and self-regulated intake on the time to wean from tube feeding in 221 long-term feeding tube dependent participants [79]. The program included occupational, speech, and psychodynamic therapy in addition to nutritional counseling for the participants and their families. Following treatment, approximately 92% of the subjects weaned completely from tube feeding and none of the subjects returned to tube feeding after completing treatment [79].

Mckirdy et al. reviewed oral intake for two subjects enrolled in school-based interdisciplinary intervention programs with a team of professionals that consisted of school based speech therapists, consultative medical speech therapists, classroom teachers and aides, physical therapists, occupational therapists, school nurses, program administrators, and caregivers [71]. At the end of treatment, one participant consumed all medications and liquids by mouth but remained on a self-restricted diet. The other participant achieved 100% oral intake and removal of nasogastric tube following treatment [71].

Shaw et al. retrospectively examined the oral intake of solid foods, weight gain, duration of meals, and percentage of self-feedings in a six-year-old boy with Down syndrome following participation in a program that combined oral motor techniques (including the Beckman Oral Motor Program) speech therapy, and occupational therapy with behavior modification including reinforcement and punishment as well as stimulant medication [76]. At discharge, the subject was reportedly eating thick soup-based mixtures with some texture added from mashed vegetables, meats and noodles, and yogurt with fruit pieces. There was an increase in lingual range of motion, but oral feeding skill was still limited to munching. Variety and duration of intake improved, and the subject was able to adequately maintain a healthy weight without the use of a high calorie supplement. At a six-month follow up, weight had increased in accordance with developmental expectations [76].

Laud et al. retrospectively reviewed a variety of dependent variables including acceptance, refusal behaviors, negative vocalizations, grams consumed, caregiver measures, food variety inventories, and caregiver satisfaction scores in a large sample of subjects ( n = 46) with diagnoses of ASD with or without co-occuring morbidity [68]. Behavioral and oral motor therapy implemented by an occupational therapist and a speech therapist included systematic meal sessions with individualized behavior protocols involving antecedent and consequence manipulation. Oral motor therapy was conducted to determine safety while eating and to perform eating and non-eating oral motor exercises. Significant differences were found in percentages of acceptance, refusal behaviors, negative vocalizations, and grams consumed from admission to discharge. Specifically, acceptance, refusal behaviors, and grams consumed increased significantly while negative vocalizations significantly decreased from admission to discharge. At three years following discharge, only 63% of the original sample could be contacted and 100% of those who could be contacted reported a significant increase in food acceptance and extinction of the structured therapeutic protocol [68].

Clinicians providing treatment to children with feeding and/or swallowing disorders should be aware of the levels of evidence available to support the various treatment modalities used in their recommended programs. The search strategy utilized for the current EBSR did not reveal any published literature that examined the effects of sensory therapies (exercises and activities that are designed to promote sensory integration such as using a brushing or vibration protocol, a weighted vest, or deep pressure to the joints) or pharmaceutical therapies (including any pharmaceutical agent prescribed by a physician for the documented purpose of improving volume of oral intake) applied singularly (clinical questions two and four) in a therapeutic environment on functional feeding outcomes (i.e. volume of intake, variety of intake, days to wean tube feeding, weight gain, or growth). This is not to imply that there is not clinical or anecdotal evidence to support the use of these modalities to improve functional feeding outcomes, but this EBSR establishes that the current level of evidence to support these types of treatments applied singularly to treat pediatric feeding and/or swallowing problems is phase I research (least robust level of evidence, see Table 6 for hierarchical levels of evidence) [81]. All of the quality features are summarized in Table 2 for each article included in this EBSR.

Clinical question one identified one published article that addressed the effect of oral motor interventions on functional oral feeding outcomes in children. [13, 21]. Although it was designed as a randomized controlled trial it was underpowered and suffered from significant loss of subjects to follow-up, making post treatment assessments impossible for nine subjects (only 55% of the subjects had both pre and post treatment analysis). As a result, the authors did not find significant differences between the control and experimental groups on the outcomes of weight gain or change in oral motor function and this study does not offer any direct support for oral motor therapy to benefit functional oral feeding outcomes. Based on the results of this EBSR, oral motor therapy applications are phase I research. Significant limitations of this study included inadequately described study protocol, p values neither reported nor calculable, and no effect sizes or confidence intervals reported or calculable (see Table 2) [21].

Of the 37 articles that addressed clinical question three, the majority (32/37) were determined to be phase one research reporting either 0/8 [27, 28, 37, 44], 1/8 [23, 24, 29, 30, 31, 36, 46, 48, 49, 55], 2/8 [26, 32, 33, 34, 35, 40, 41, 42, 43, 47, 51, 52, 54, 56, 57, 58], or 3/8 [50, 53] quality indicators. Limitations of the articles determined to be phase one research included small sample numbers (single subject or case controlled trials), with inadequately described protocols (insufficient detail for replication), utilizing convenience samples with inconsistently described evidence of treatment fidelity, and inconsistent reports of significance precision (effect size). The five remaining articles relevant to clinical question three were randomized controlled trials and determined to be phase four research [22, 25, 38, 39, 45]. None of the randomized controlled trials provided 8/8 quality markers, please see Table 2 for complete summary of included features.

The 23 studies identified by this EBSR relevant to clinical question five can be described as phase one research with all of them reporting two or less quality markers (seven studies reported 0/8 quality markers [18, 60, 71, 76, 77, 78, 79], five reported only 1/8 quality markers [59, 61, 62, 64, 75], and 11 reported 2/8 quality markers [63, 65, 66, 67, 68, 69, 70, 72, 73, 74, 80]. Only three of the 23 articles relevant to mixed modality interventions reported on the statistical significance of their findings [63, 68, 73].