An Empirically Supported Framework for Addressing Pediatric Nonalcoholic Fatty Liver Disease in Outpatient Care

Available online at www.sciencedirect.com

ScienceDirect Cognitive and Behavioral Practice 21 (2014) 446-455 www.elsevier.com/locate/cabp

An Empirically Supported Framework for Addressing Pediatric Nonalcoholic Fatty Liver Disease in Outpatient Care Rachel A. Annunziato, Fordham University and Mount Sinai School of Medicine Melissa Rubes and Chad Davis, Fordham University Samantha Rose, Recanati-Miller Transplant Institute, Mount Sinai Hospital Ronen Arnon, Mount Sinai School of Medicine and Recanati-Miller Transplant Institute, Mount Sinai Hospital In the wake of the pediatric obesity epidemic, the prevalence of nonalcoholic fatty liver disease (NAFLD) has been rising. Standard care for pediatric NAFLD includes dietary and physical activity guidelines, but such recommendations are difficult for families to implement. Clinicians in medical settings may be called upon to address NAFLD; cognitive behavioral techniques that have been effectively used for pediatric obesity are likely applicable. However, given the unique circumstances surrounding NAFLD, it may be helpful also to incorporate community-based and telehealth strategies when assisting families. This treatment development report offers a framework for integrating cognitive behavioral techniques within a community-based, telehealth approach to treating NAFLD within a medical setting. A series of illustrative cases is presented to depict exemplary considerations for implementing this intervention.

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ECAUSE of the growing pediatric obesity epidemic, children are now facing impairment in their liver functioning akin to that caused by alcohol abuse (Dunn & Schwimmer, 2008); this is called nonalcoholic fatty liver disease (NAFLD). Currently, approximately 40% to 70% of obese children have a diagnosis of NAFLD (Bellentani, Scaglioni, Marino, & Bedogni, 2010). NAFLD is associated with increased overall mortality compared to matched controls (Chalasani et al., 2012) and it may become the most common reason for liver transplantation by 2025 (Malik et al., 2009). Standard care for pediatric NAFLD consists of providing dietary guidance, including consultation with a nutritionist and physical activity recommendations. Recommendations are typically focused on promoting weight loss and changes in lifestyle management in congruence with current national guidelines regarding healthy eating and physical activity. Those patients with advanced histological changes are prescribed antioxidant therapy such as vitamin E. Children have regular clinic visits, where their liver chemistries are checked. Research on whether these recommendations result in clinically significant change is limited at this time but findings do suggest that when applied as instructed there are improvements in steatosis

Keywords: NAFLD; pediatric obesity; cognitive behavioral; community-based; telehealth

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and liver functions (Nobili et al., 2006; Nobili et al., 2008). However, a recent study found that families were unable to consistently implement U.S. dietary and physical activity guidelines (Centers for Disease Control and Prevention, 2012; United States Department of Agriculture, 2013); therefore, overall, patients did not lose weight (Kerkar et al., 2013). Furthermore, this study found that the psychosocial consequences of NAFLD seem to be worse than pediatric obesity alone. From both a medical and quality-of-life standpoint, establishing effective methods to help children with NAFLD lose weight is imperative. It is possible that effective treatments for pediatric obesity could be applied to children who also have a NAFLD diagnosis. A review on treatment options for pediatric obesity highlighted several components as key to successful weight management. These include cognitive behavioral strategies such as goal-setting, self-monitoring, stimulus control, and positive reinforcement (incentives) as well as creating a supportive family environment (Kirk, Scott, & Daniels, 2005). Yet, while shown to be efficacious, the effectiveness of such approaches in an outpatient clinical setting is largely unknown (Kirk, Scott, & Daniels, 2005). Children diagnosed with NAFLD are typically sent to medical settings with a pediatric gastroenterology or hepatology specialty and this location may not be close to home; therefore, not only are clinicians faced with the challenge of delivering services in the context of outpatient clinical care but also to families that they do not see regularly. Our goal was to develop a culturally congruent cognitive behavioral package that can feasibly be delivered to affected

A Framework For Addressing NAFLD families on or off-site of the medical setting. The Ecological Model of Childhood Overweight (Davison & Birch, 2001) recognizes that any effort to reduce childhood obesity must incorporate an understanding of the dynamic relations between the individual child and his/her family, school, neighborhood and larger social and cultural contexts. Within this framework, below we present a cognitive behavioral-based approach to addressing NAFLD in outpatient care.

Cultural and Ethnic Factors Influencing Obesity Management Cultural attitudes and behaviors contribute greatly to the etiology and maintenance of obesity. Norms within cultures influence child-feeding practices, preferences for physical activity and body image (Caprio et al., 2008). In addition to cultural norms, environmental variables specific to each community can impact weight control. Numerous studies have found associations between obesity and how much a neighborhood is saturated with fast food restaurants (Davis & Carpenter, 2009; Maddock, 2004), markets that provide access to affordable fruits and vegetables (Inagami, Cohen, Finch, & Asch, 2006; Morland, Diez Roux, & Wing, 2006), and areas conducive to physical activity (Giles-Corti, Macintyre, Clarkson, Pikora, & Donovan, 2003; Gordon-Larsen, Nelson, Page, & Popkin, 2006). Because the etiology and maintenance of obesity varies across cultural groups, weight-loss interventions need to be tailored according to the needs of its members. For this reason, community-based approaches to combat obesity are preferred because they allow investigators to identify cultural and environmental barriers to weight loss and harness resources within the community that would encourage weight loss (Economos, 2007).

Factors Influencing Community-Based Approaches for Treating Obesity All community-based interventions emphasize and value the social, cultural, and environmental factors influencing obesity. A systematic review of obesity prevention interventions in schools found that greater community partnership and engagement improved the success of these programs in achieving health-related goals (Krishnaswami, Martinson, Wakimoto, & Anglemeyer, 2012). Furthermore, parents play a key role in the success of community-based interventions (Koplan, Liverman, & Kraak, 2005). And, perhaps most important, it is necessary to achieve a high degree of youth buy-in if the effects of the intervention are to be sustained over time. Youth buy-in is cultivated by community-based participatory research (CBPR). CBPR entails a “partnership approach to research that equitably involves community members, organization representatives, and researchers

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in all aspects of the research process” (Israel, Eng, Schulz, & Parker, 2005, p. 5). Because CBPR incorporates community members in every step of the intervention process, from the initial design to the implementation and the evaluation of the program, it has the added benefit of designing interventions to meet the needs of people in the community, to be culturally relevant, and to be empowering to participants (Ginossar & Nelson, 2010), as well as the potential to result in lasting and sustainable changes in community health behaviors. CBPR studies promote and maintain youth buy-in when barriers to participation are removed (e.g., transportation issues), when there are incentives to participate (e.g., monetary, skills development, or improvements in their community), and when participation is perceived to be valued and meaningful to the project's goals (Vaughn, Wagner, & Jacquez, 2013).

Use of Telehealth in the Management of Pediatric Obesity For families living far away from tertiary care centers, accessing recommended services for pediatric obesity may not be feasible; this too may be a formidable challenge in treating NAFLD. The high rate of pediatric obesity among rural populations (Ogden, Carroll, Curtin, Lamb, & Flegal, 2010) has prompted exploration of innovative strategies for service provision like “telehealth” or “telemedicine.” Research has shown that telehealth does improve access to obesity services and decreases attrition for rural families (Irby, Boles, Jordan, & Skelton, 2012). Furthermore, telehealth for pediatric obesity appears to be equally acceptable to families as face-to-face care (Mulgrew, Shaikh, & Nettiksimmons, 2011). Few studies have examined outcomes including weight as well as physical activity and eating behavior. So far, the limited findings on objective outcomes are equivocal (Cohen, Irby, Boles, Jordan, & Skelton, 2012). But telehealth appears to be a feasible and acceptable way to deliver services.

The Application of Cognitive Behavioral Techniques to Address NAFLD Cognitive behavioral techniques are widely used as a part of evidence-based treatments for obesity (Jeffery et al., 2000; Kirk, Scott, & Daniels, 2005). Among children, data suggest that incorporating behavioral techniques and physical activity leads to successful short- and long-term results (Nemet et al., 2005; Nemet, Levi, Pantanowitz, & Eliakim, 2014). Drawing from the work of others, especially the Look AHEAD trial, our approach is to use a “toolkit” of cognitive behavioral strategies to be chosen based on individual family needs (The Look AHEAD Research Group, 2006). The Look AHEAD trial is investigating whether intentional weight loss leads to improved cardiovascular outcomes in over 5,000 individuals diagnosed with Type 2 diabetes. The behavioral arm of the Look AHEAD package prescribes the

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use of problem solving to identity barriers to successful weight loss (The Look AHEAD Research Group, 2006). Problem-solving therapy (Nezu, 2004) is a highly effective agent of behavioral change for patients with a wide range of health conditions (Malouff, Thorsteinsson, & Schutte, 2007) and is the backbone of our approach as a means to facilitate service procurement. The specific utility of problem-solving therapy for aiding in weight loss has been demonstrated (Perri et al., 2001).

Integrating Cognitive Behavioral Techniques Into a Community-Based, Telehealth Approach Figure 1 displays the integrative framework that inspired our methods. This community-based approach entailed connecting children and their parents to fitness programs located within their neighborhood. We sought out an array of programs and compiled an exhaustive list of resources for families. We attempted to match the needs and interests of the individual child with local and culturally sensitive community programs. The resource list was primarily physical activity focused. Interventions based solely on exercise have resulted in improvements in cardiovascular health and reductions in weight, triglycerides, and fasting glucose (Shaw, Gennat, O'Rourke, & Del Mar, 2006). Studies comparing exercise-only vs. diet-only have shown that exercise-only interventions have had greater success in helping men lose weight (King, Frey-Hewitt, Dreon, & Wood, 1989) and helped men and women keep weight off in the long term (Pronk &

Wing, 2012; Skender et al., 1996). In addition, exercise-only interventions consistently lower levels of visceral body fat even in the absence of weight loss (Lee et al., 2005), a finding that is particularly salient for NAFLD patients. Community-based approaches for treating obesity that focus on changing too many behaviors at once (diet, exercise, smoking, etc.) often fail to produce a significant amount of weight loss (Hall, Tunstall, Vila, & Duffy, 1992; Jeffery, 2012). We therefore hypothesize that our intervention, which encourages a singular behavior (physical activity), will prevent families from feeling overwhelmed at the idea of having to make several substantial lifestyle changes at one time. The first step we took during intervention development was to canvas the two communities served by a majority of NAFLD patients. To do so, we collaborated with other pediatric teams in order to learn if/whether they had any community connections in these areas. Then, we conducted a broad Internet search for additional locations. At that point, all options were entered into a database and the list was divided among team members to obtain further information. All sites were called to verify their contact information and to learn more about cost (if any), languages spoken, hours, etc. In the event that a site could not be reached, it was removed from the list. In total, it required approximately 30 hours to produce two comprehensive sets of resources (about 15 hours each). We expected that some families would require resources from other areas. Our initial undertaking helped us identify

Figure 1. Integrative framework utilized in the intervention.

A Framework For Addressing NAFLD where to search first and therefore subsequent efforts proceeded more quickly. For each patient from a different neighborhood, list generation takes about 1 to 2 hours. Figure 2 displays a verified list as given to a family. Drawing from the telehealth movement and cognitive behavioral practice, patients are given these resources and then followed by a coordinator who institutes a problem-solving approach (Nezu, 2004) to identifying barriers faced in securing and utilizing resources. Other studies by our group based in medical settings have shown that the utilization of a familiar coordinator (or interventionist) may be associated with improved health behaviors (Annunziato et al., 2013). Within this problem-solving framework, specific cognitive behavioral strategies are selected. Interventionists determine whether there are any barriers, generate solutions, and encourage selection of a solution that is evaluated during a later call. In the process of generating solutions, other cognitive behavioral strategies may be employed, such as goal setting, reframing, assertiveness training, and positive reinforcement, when appropriate. The interventionist communicates with families either by phone or email (off-site) or in clinic if requested. Of note, no families to date have been able to consistently meet with their interventionist in clinic. Team Members Our NAFLD team is composed of volunteers at different training levels. Table 1 describes each role and the corresponding time commitment. The protocol is run by a licensed clinical psychologist who also serves as an interventionist. A doctoral-level graduate student coordinates the project, manages resource procurement, and

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serves as an interventionist. An additional graduate student has assumed an interventionist role. An undergraduate level student assists with verification of resources. The study psychologist meets regularly with the interventionists for supervision and assigns a required reading, CBT for Chronic Illness and Palliative Care (Sage, Snowden, Chorlton, & Edeleanu, 2008). This text has a “toolkit” section that is referenced by interventionists. The medical director of Pediatric Liver/Liver Transplant provides oversight. A hepatology nurse also assists with medical queries. Procedure A medical team member first sees all patients with a NAFLD diagnosis at a routine outpatient visit. At our site, all other causes of abnormal liver enzymes and liver diseases are initially ruled out before making a NAFLD diagnosis. This process includes taking a clinical history, physical examination, blood work or laboratory studies, and possibly abdominal ultrasound. In addition, we try to rule out other causes for obesity, for example, low level of thyroid hormone-hypothyroidism and liver diseases. During this time, it is determined if the patient might benefit from our intervention. The medical team refers all families who understand the program and express interest in it. Patients and families are not referred if they do not understand or if the patient could not participate in physical activity. A study team member then explains to families that we are examining a program to assist families with finding physical activity resources; it is emphasized that participation is voluntary. To date, 33 children with NAFLD have been seen in clinic. Of these, 29 were referred to the program, four were not referred

North X Fitness Facilities Organization

Description

Phone #

Cost

Hours

Spanish

Free

Mon-Fri: After school

Y

Free

Mon-Fri: 2:30-6pm; Mon, Tues, Fri: 6-9pm (teens); Thurs: 6-9pm (ladies only); Sat: 9-5pm

Y

Basketball, soccer, dance, netball, track and field (ages 11-17)

Free

Mon, Wed, Fri: 6:308:30pm & Sat afternoons

Y

Swimming, tennis courts, basketball, baseball, softball

Free

Outdoor Pools: 11-7pm Field House: 7-4pm

Y

Dance, basketball, X Shape Up: Kickboxing; Zumba

Free

Y

A weight loss/ nutrition program developed by the Children’s Hospital at X and the XX Community Center (XXCC) Karate, basketball, archery

Free

Teen Program, Mon-Fri 6-9:30pm, Sat 12-3pm; Kickboxing, Sat: 12-1pm; Zumba, Mon: 6-7pm After school clinic: 4:30-6:30pm

Mon-Fri: 9-10pm

Y

Swimming, basketball courts, handball courts Basketball, dance, soccer, martial arts

Contact Name

Address

Free

Figure 2. Resource list of community-based fitness programs for participants and their families.

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Table 1

Resources allocated for NAFLD intervention Role

Responsibilities

Time commitment

Clinical Psychologist

✓ Oversee protocol ✓ Supervise interventionists ✓ Manage outcome assessment ✓ Oversee protocol ✓ Supervise team members ✓ Manage resource procurement ✓ Regularly contact families and implement protocol ✓ Liaison with other team members ✓ Meet w/ prospective patients & provide clearance ✓ Medical oversight of protocol ✓ Identification of patients ✓ Reiterate interventions to patients ✓ Addressing medical questions

5 hours/ week

Project Coordinator

Interventionists (3) Physician Hepatology nurse

and two declined participation. Among the four who were not referred, in two cases, the diagnosis of NAFLD was being reconsidered. In the third case, the patient was accompanied by his aunt rather than his primary caregiver. For ethical reasons as well as consideration of who the intervention should be delivered to, the medical director recommended that the patient return to clinic within a month accompanied by his caregiver. The last case was wheelchair bound with limited mobility and therefore reported difficulty participating in physical activity. Both families who refused stated that they did not have the time to participate in physical activity options. Once cleared for the intervention by medical personnel, patients immediately meet a member of our team. The team member describes our approach and provides the family with a list of resources. Both the medical and research team recommend physical activity and encourage usage of this list. If their neighborhood has not been canvassed, a list of resources is created and sent within a week. When possible, the team member who meets the family in clinic also serves as the interventionist. However, in order to balance caseloads, in some instances the interventionist is different. In this situation, the family is told at the initial visit who their interventionist will be and when to expect their call. Within a week, all families are called by their interventionist and this continues every 3 to 4 weeks. The interventionist records who they spoke to, the length of the call, and the content. During these calls, the interventionist checks in about physical activity use or barriers and generates corresponding solutions. For example, a common barrier encountered was concern over the time requirement for physical activity. In such cases, the interventionist may challenge automatic thoughts about lack of time for exercise and/or distortions about how much time it might require. If it appears that patients or parents are uncomfortable making calls about physical activity resources, the interventionist will

5 hours/ week

2 hours/ week 2 hours/ week 2 hours/ week

make an initial call and/or encourage and teach assertiveness. In some cases, positive reinforcement was discussed and employed by families, although interestingly, many patients expressed excitement over and enjoyment of their chosen exercise without the need for incentives. In all cases where barriers were reported, goal setting was utilized to help patients and families take initial steps towards increasing their physical activity. Depending on family preferences and the patient’s age, the interventionist may speak to the parent, the child, or both. Calls typically last for 10 to 15 minutes. However, especially when using a professional translation service, calls may be longer. Patients receive 4 to 6 phone calls before attending their next clinic appointment. During the course of the protocol, the interventionist continues to ask for youth and parent feedback in order to keep the child engaged and interested in the intervention. We recognize that youth buy-in is an essential component to the success of community-based programs. The interventionist attempts to remove any barriers to youth participation, such as transportation issues or scheduling conflicts. Participants are commended for their engagement in physical activity. The interventionist continually stresses how important and meaningful the child’s activities are to improving his/her health and well-being. Outcome Assessment All enrolled patients are asked to complete a brief outcome assessment battery consisting of a quality-of-life and physical activity measure. The Pediatric Quality of Life Inventory (PedsQL; Varni, Seid, & Kurtin, 2001) was chosen as it is a widely used, brief measure of quality of life with demonstrated psychometric properties among children, ages 8 to 17, with special health care needs. Scores on the PedsQL range from 0 to 100 with 100 indicating high quality of life. In a normative sample of healthy

A Framework For Addressing NAFLD children, mean total score and subscales scores were above 80 (Varni et al., 2001). The Physical Activity Questionnaire for Older Children (PAQ-C) or Adolescents (PAQ-A) is also a widely used measure of general physical activity levels from childhood to adolescence with demonstrated reliability and validity (Kowalski, Crocker, & Faulkner, 1997; Kowalski, Crocker, & Kowalski, 1997). Scores on the PAQ-C and PAQ-A range from 1 to 5, with 5 indicating high physical activity. In two validation studies of the PAQ-C, the mean scores were 3.23 and 3.35, respectively (Kowalski, Crocker, & Faulkner, 1997). In an adolescent validation sample, the mean score on the PAQ-A was 2.31 (Kowalski, Crocker, & Faulkner, 1997). Finally, a short acceptability survey was given as well to all patients in the intervention group.

Recommendations for Applying Cognitive Behavioral Strategies to Specific Presentations In the section below, we have selected three cases that illustrate salient themes and considerations that have been unearthed during the intervention. We first present an example of a case where the barriers to physical activity were quickly uncovered and the choice of cognitive behavioral strategies linked clearly to these obstacles. Second, we have learned that complex medical cases are common and we include an example of this. Third, the specific challenge of weight loss maintenance is presented. All three case examples were Hispanic and had public insurance, which echoes the majority of NAFLD patients seen at the study site. Case Example: Goal Setting in Action Case A is a 12-year-old Hispanic female who initiated our protocol with a BMI of 27.78. Her PedsQL score was 78.26 with a PAQ-A score of 3.01. She and her mother, Ms. A, reported feeling uncertain about starting physical activity due to time constraints, but she expressed excitement over trying tae kwon do. Ms. A mainly worried about whether this would conflict with school and homework. The interventionist working with this family focused on problem-solving barriers at the family level, such as finding appropriate activities after school and on weekends that would not interfere with Case A’s homework and extracurricular activities. In this context, problematic automatic thoughts about how physical activity might lead to disruptions in schoolwork were challenged and reframed. In addition, goal setting was utilized and Case A agreed to first try a tae kwon do class; this also required prompting assertiveness for both Case A and Ms. A (e.g., to take the initial step of scheduling the class). Over a series of four phone calls, where both Case A and her mother participated, we learned that Case A had been consistently attending classes which she described enjoying very much and reported no trouble balancing

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her schoolwork with her new physical activity. At her follow-up visit, Case A’s BMI was 25.84 with a PAQ-A score of 3.49; her PedsQL score had increased to 89.13. Case A reported being “very satisfied” with the program. Discussion A toolkit of cognitive behavioral strategies was employed to address the barriers to initiating physical activity expressed by Case A. In Case A, incentives did not seem applicable, given her strong motivation, but rather helping to mobilize the family to support her enthusiasm and willingness to attend tae kwon do classes was needed. Also of note, we have thus far, perhaps surprisingly, not detected any differences in how our male and female patients present in regards to psychological symptoms. Concerns about weight and body image have been quite uniform. Our pilot work suggested that girls with NAFLD may experience poorer self-esteem (Kerkar et al., 2013) but thus far, from a clinical standpoint, this has not been observed. Case Example: Medically Complex Patient Case B, a 17-year-old Hispanic male, was diagnosed with Trisomy 21. He received a heart transplant in 2010 due to congenital heart disease, status post repair with subsequent congestive heart failure. He only recently started seeing the Pediatric Liver/Liver Transplant team due to elevated liver enzymes. He was obese with a BMI of 30.73. Case B was overweight before his heart transplant but has gained weight since; it is unknown if this is related to the immunosuppressant medications that he is now taking. Immunosuppression, especially prednisone, can increase appetite and weight gain in patients post-transplant. Our team immediately contacted the cardiology team to learn whether Case B had any physical activity restrictions. His mother also was unsure about this. We learned right away that the Cardiology team strongly encouraged him to increase his activity level. Cardiology agreed to echo this message and both teams emphasized working together towards a common goal. But, Case B also faced significant challenges in taking up physical activity. Although he was enthusiastic about weight loss, his mother stated that he tired easily and they reported that walking was difficult and sometimes painful for him because his legs are different lengths. Case B did excitedly discuss and gesture how much he’d like to “pump iron.” Consequently, resources were located that would allow him to spend time in a gym setting. Ms. B was contacted over the course of four phone calls. The interventionist working with this family understood that Case B would follow his mother’s lead on this and he was eager to try some new things. However, his mother was appropriately distressed and overwhelmed about her son’s special health care needs. Therefore, the

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provision of resources that had been “vetted” already was very helpful to her but the telehealth sessions also addressed her problematic beliefs (e.g., “He will become too tired” or “It’s too cold for him to go out”) by reframing (“He may actually have more energy because of exercise”) and consistent psychoeducation about the benefits across his presenting problems as well as reasonable goal setting. Additionally, behavioral experiments were created whereby Case B’s mother asked him how he felt before and after light physical activity like lifting weights. On many occasions, Case B did report being tired, which was troubling for her. Therefore, the interventionist flexibly shifted to an emphasis on making sure that Case B at least participates in gym at school, which he denied feeling fatigued after, but also encouraged his mother to take a more active role in monitoring his food intake with the same vigilance as his physical activity. This shift in goal setting was reportedly helpful and his mother was able to work with school personnel to reduce overeating there. Case B has not been seen by the Pediatric Liver team but weight loss (3 pounds) was noted by the Cardiology team. Due to cognitive deficits associated with Trisomy 21, Case B was not asked to complete the outcome measures. However, Ms. B reported being “very satisfied” with the phone sessions. Discussion It is not uncommon to see medically complex cases with a diagnosis of NAFLD. We sought to frame weight loss as a goal across presentations. Furthermore, systemically, this family did not have a long-standing connection to the Pediatric Liver team, and we tried to work closely with the Cardiology team, who the family knew well. This case also illustrated the need for and challenge of cultivating a supportive family environment when it comes to pediatric weight loss. Problem solving was more focused on barriers salient to Ms. B than to Case B. In addition, the flexibility allowed for was extremely helpful when behavioral experiments did indeed reinforce concerns by Case B’s mother. Case Example: Hitting the Weight Maintenance Wall Case C is a 9-year-old Hispanic male diagnosed with NAFLD in early 2013. Although his BMI was in the “normal weight” range, it was increasing at every office visit and he was referred to our protocol in June 2013 with a BMI of 22.45. A team member met with Case C and his mother to discuss our intervention and see if it might be helpful to them. Case C’s mother is Spanish speaking and all meetings, in person or by telephone, have included an interpreter. Ms. C described how hard it has been for Case C to keep his weight down and the possibility of increasing physical activity was discussed. At the time, Case C’s

PedsQL score was an 86.96 and his PAQ-C score was a 3.15. Using a problem-solving framework, the interventionist and mother discussed appropriate incentives for his engagement in physical activity. Since Case C appeared to have “hit a wall” during weight maintenance, as is common (Jeffery et al., 2000), this seemed like an important time to consider the use of other motivating factors besides weight loss itself. In communications with Case C and his family, the interventionist learned that Case C wanted to learn how to play soccer so he could try out for his school’s soccer team. Case C occasionally played kickball with his friends after school, but he was nervous about trying out for the soccer team, because he was unsure of how to play. The interventionist spoke to Case C about attending a soccer clinic where he could improve his skills and gain the confidence to try out for the team. The interventionist was able to locate a soccer club, within walking distance from his home, which offered an 8-week program for beginners. The interventionist spoke with the clinic’s director and Case C’s family to help secure him a spot in the program. Case C’s motivation to join a soccer team and the encouragement from his family and the interventionist played a major role in helping him to increase his physical activity and to lose weight. During phone calls, Ms. C has reported that her son cannot stop practicing his soccer drills. Even when the 2-hour clinic was over, Ms. C reported that her son would stay late and go over the new things he learned with his friends. Ms. C stated that if it was “up to my son, he would be in clinic every day!” When the 8-week soccer program was over, Case C was eager to try out for his school’s soccer league in the fall. Ms. C was contacted over the course of six phone calls; most of these conversations were with her, given Case C’s age, but he was checked in with briefly as well. The interventionist provided substantial positive reinforcement, specifically praise and support, to Case C and to Ms. C. She continually stressed how important the child’s activities were to improving his/her health and well-being. At his follow-up visit 6 months later, Case C and his mother showcased his certificate of completion from the soccer clinic to the medical team. Case C reported feeling very satisfied with the program. His BMI was 21.61 with a PedsQL score of 88.04. Case C’s PAQ-C was considered invalid because he was sick over the past 7 days, therefore he will be asked to complete it again at his next visit. Discussion Case C was frustrated by hitting the weight maintenance wall and he was losing motivation. The interventionist focused on Case C’s interest in soccer and encouraged him to attend a nearby soccer clinic where he could practice and learn some basic drills. Our

A Framework For Addressing NAFLD approach was to make weight loss maintenance as simple and enjoyable as possible for Case C and his family. We thought that finding ways for Case C to learn how to play soccer and to offer positive reinforcement to his family would help him to increase his physical activity and overcome his weight maintenance difficulties.

Discussion The increase in NAFLD prevalence reflects a grim progression of the obesity epidemic. Just a few decades ago, it would have been unheard of for a child to be monitored for this disease. Personnel in medical settings are likely to now encounter NAFLD but may have a limited capacity to offer services beyond standard care, which appears difficult for families to consistently implement (Kerkar et al., 2013). Recommendations for addressing NAFLD are comparable to those for pediatric obesity; however, the context of treatment may vary greatly. Children with a NAFLD diagnosis are sent to specialists, who may be geographically distant, and such distance may contribute to families feeling less connected to the specialist than to their pediatrician. In addition, there may be more urgency to lose weight since children with NAFLD can develop liver fibrosis or cirrhosis. Therefore, an intervention, drawing from several evidence-based areas, was developed that would address the specific needs of this population. Namely, cognitive behavioral strategies were packaged within a community-based, telehealth model. The cases presented exemplify typical as well as more difficult scenarios that have been encountered thus far. Perhaps most importantly, this approach can generalize across a diverse range of patient presenting problems and backgrounds. Implementation of this protocol requires approximately 15 to 20 hours a week across personnel of different training backgrounds and levels. Medical expertise is necessary and the contribution of medical team members does not appear to greatly exceed the amount of time that delivering standard care to their NAFLD patients requires. A challenge faced during implementation of the protocol was that on some occasions, the interventionists fielded questions during calls that required consultation with the medical team. Therefore, patients and/or parents had to wait for an answer. In addition, despite the initial legwork completed to vet community resources, families and interventionists encountered changes in the information they initially received, which the intervention team had to resolve. Relatedly, new resources became available more frequently than was expected and personnel time was allocated to keep up with this. Of note, the activities required could certainly be delivered by a variety of personnel, including those from medicine, nursing, psychology, and social work, with either a part-time role or divided among team members as

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in our paradigm. Anecdotally, it does seem that experience with child/adolescent mental health and family dynamics may be helpful as has been the case concerning pediatric obesity (Kirk, Scott, & Daniels, 2005). Applications A goal when designing this approach was that it would have translational capabilities beyond pediatric NAFLD. For example, team members are working with Pediatric Endocrinology to see if this might be applicable and feasible to introduce in their setting. A recent survey of rural health care providers reported that the most helpful means for them to treat pediatric obesity would be readily accessible patient education materials, strategies to link patients with community treatment programs, and training in brief, focused counseling skills (Shaikh, Nettiksimmons, & Romano, 2011). The approach described here certainly encompasses these areas and perhaps could be generally used to assist clinicians in the management of pediatric obesity both in medical and rural settings. According to the American Academy of Pediatrics, physician recommendations and referral to community-based programs are encouraged for treating pediatric obesity within low-income families (American Academy of Pediatrics, 2003). Beyond the implications for medical staff, this approach may also be initiated by academic settings with strong community ties and need. Outcome Assessment Outcome assessment using a randomized-controlled design is underway to determine if this approach compared to standard care is associated with reductions in weight, improved liver functioning, quality of life, and increased physical activity as well as its acceptability. Because standard care appeared to be ineffective at our site (Kerkar et al., 2013), in this investigation the control condition has been augmented such that it also includes provision of an individualized community resource list. Based on preliminary power analyses, this protocol will be delivered to at least 34 families. Thus far, results suggest high acceptability with the intervention. However, some families have mentioned that the nutritional consultation component of standard care could be bolstered and more personalized; this suggestion is being addressed by the medical team. Medical team members have reported that the intervention has been helpful in regards to gathering information and supporting related challenges between visits. For example, in one case, a mother reported to the interventionist that her child’s teacher discovered that he was swapping his healthy lunches with his friends’. The interventionist was able to offer support and advice to her. An important consideration in the development of a telehealth approach is feasibility. Thus far there have been few difficulties

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reaching families; in fact, only two families assigned to the intervention arm have been challenging to contact. Conclusions Pediatric obesity alone is difficult to treat. Pilot work has illustrated some of the additional, specific challenges added when NAFLD develops. Because standard care cannot always be consistently implemented within families affected by NAFLD, an empirically based protocol to improve outcomes was developed, drawing on community-based, telehealth, and cognitive behavioral interventions. Research on effective community-based interventions for pediatric obesity emphasize that any effort to reduce childhood obesity must incorporate an understanding of the dynamic relations between the individual child and his/her family, neighborhood, and larger social and cultural contexts (Davison & Birch, 2001). There also needs to be a considerable amount of participant buy-in, such that youth feel empowered by the intervention and perceive their efforts as valuable and meaningful (Vaughn, Wagner, & Jacquez, 2013). To comply with the general recommendations of community-based obesity research, the interests of the individual child and his/her parents have been incorporated by connecting them to local, culturally relevant community programs. Research on telehealth supports the delivery of services via telephone and electronic devices to children and families that are not conveniently located near the tertiary care centers (Irby, Boles, Jordan, & Skelton, 2012). To comply with the auspices of telehealth programs, children and their parents have been contacted regularly via telephone or email to monitor and check in on their levels of physical activity. Cognitive behavioral theory includes many techniques that have been demonstrated and are relevant to the treatment of children with NAFLD. Grounded in cognitive behavioral theory, interventionists check in periodically with participants and their families to problem solve any issues related to health, scheduling, and transportation conflicts, to make sure that the child is still engaged and interested in the program, and to the help the child find a more appropriate activity, if necessary. The use of cognitive behavioral techniques also contributes to maintaining youth buy-in. Interventionists encourage and commend youth on efforts to stay physically fit by stressing the value and importance of the child’s active lifestyle and suggesting incentives where appropriate. Although centered on the specific circumstances surrounding addressing NAFLD in a medical setting, we are hopeful that this approach could be helpful more generally as well.

References American Academy of Pediatrics. (2003). Policy statement: Prevention of pediatric overweight and obesity. Pediatrics, 112, 424–430.

Annunziato, R. A., Baisley, M. C., Arrato, N., Barton, C., Henderling, F., Arnon, R., & Kerkar, N. (2013). Strangers headed to a strange land? Utilization of a transition coordinator to improve transfer from pediatric to adult service. The Journal of Pediatrics, 163(6), 1628–1633. Bellentani, S., Scaglioni, F., Marino, M., & Bedogni, G. (2010). Epidemiology of non-alcoholic fatty liver disease. Digestive Diseases, 28(1), 155–161. Caprio, S., Daniels, S. R., Drewnowski, A., Kaufman, F. R., Palinkas, L.A., Rosenbloom, A. L. (2008). Influence of race, ethnicity, and culture on childhood obesity: Implications for prevention and treatment. A consensus statement of Shaping America's Health and the Obesity Society. Diabetes Care, 31(11), 2211–2221. Centers for Disease Control and Prevention (2012, August 7). Physical Activity. Retrieved from http://www.cdc.gov/physicalactivity/ index.html. Chalasani, N., Younossi, Z., Lavine, J. E., Diehl, A. M., Brunt, E. M., Cusi, K., … Sanyal, A. J. (2012). The diagnosis and management of non-alcoholic fatty liver disease: Practice guideline by the American Association for the Study of Liver Diseases, American College of Gastroenterology, and the American Gastroenterological Association. Hepatology, 55(6), 2005–2023. Cohen, G. M., Irby, M. B., Boles, K., Jordan, C., & Skelton, J. A. (2012). Telemedicine and paediatric obesity treatment: Review of the literature and lessons learnt. Clinical Obesity, 2, 103–111. Davis, B., & Carpenter, C. (2009). Proximity of fast-food restaurants to schools and adolescent obesity. American Journal of Public Health, 99(3), 505–510. Davison, K. K., & Birch, L. L. (2001). Childhood overweight: A contextual model and recommendations for future research. Obesity Reviews, 2(3), 159–171. Dunn, W., & Schwimmer, J. B. (2008). The obesity epidemic and nonalcoholic fatty liver disease in children. Current Gastroenterology Reports, 10(1), 67–72. Economos, C. D. (2007). Community interventions: A brief overview and their application to the obesity epidemic. The Journal of Law, Medicine & Ethics, 35(1), 131–137. Giles-Corti, B., Macintyre, S., Clarkson, J. P., Pikora, T., & Donovan, R. J. (2003). Environmental and lifestyle factors associated with overweight and obesity in Perth, Australia. American Journal of Health Promotion, 18(1), 93–102. Ginossar, T., & Nelson, S. (2010). Reducing the health and digital divides: A model for using community-based participatory research approach to E-health interventions in low-income Hispanic communities. Journal of Computer-Mediated Communication, 15(4), 530–551. Gordon-Larsen, P., Nelson, M. C., Page, P., & Popkin, B. M. (2006). Inequality in the built environment underlies key health disparities in physical activity and obesity. Pediatrics, 117(2), 417–424. Hall, S. M., Tunstall, C. D., Vila, K. L., & Duffy, J. (1992). Weight gain prevention and smoking cessation: Cautionary findings. American Journal of Public Health, 82(6), 799–803. Inagami, S., Cohen, D. A., Finch, B. K., & Asch, S. M. (2006). You are where you shop. American Journal of Preventive Medicine, 31(1), 10–17. Irby, M. B., Boles, K., Jordan, C., & Skelton, J. A. (2012). TeleFIT: Adapting Multidisciplinary, Tertiary-Care Pediatric Obesity Clinic to Rural Populations. Telemedicine and e-Health, 18(3), 247–249. Israel, B. A., Eng, E., Schulz, A. J., & Parker, E. A. (2005). Methods in community-based participatory research for health. San Francisco: Jossey-Bass. Jeffery, R. W. (2012). Community programs for obesity prevention: The Minnesota Heart Health Program. Obesity Research, 3(S2), 283s–288s. Jeffery, R. W., Drenowski, A., Epstein, L. H., Stunkard, A. J., Wilson, G. T., Wing, R. R., & Hill, D. R. (2000). Long-Term Maintenance of Weight Loss: Current Status. Health Psychology, 19, 5–16. Kerkar, N., D’Urso, C., Van Nostrand, K., Kochin, I., Gault, A., Suchy, F., … Annunziato, R. A. (2013). Psychosocial outcomes for children with NAFLD over time and compared to obese children without NAFLD. Journal of Gastroenterology and Hepatology, 56(1), 77–82.

A Framework For Addressing NAFLD King, A. C., Frey-Hewitt, B., Dreon, D. M., & Wood, P. D. (1989). Diet vs exercise in weight maintenance: The effects of minimal intervention strategies on long-term outcomes in men. Archives of Internal Medicine, 149(12), 2741. Kirk, S., Scott, B. J., & Daniels, S. R. (2005). Pediatric obesity epidemic: treatment options. Journal of the American Dietetic Association, 105(5), 44–51. Krishnaswami, J., Martinson, M., Wakimoto, P., & Anglemeyer, A. (2012). Community-engaged interventions on diet, activity, and weight outcomes in U.S. schools: A systematic review. American Journal of Preventive Medicine, 43(1), 81–91. Koplan, J. P., Liverman, C. T., & Kraak, V. A. (2005). Preventing childhood obesity: Health in the balance. Washington, DC: The National Academies Press. Kowalski, K. C., Crocker, P. R. E., & Faulkner, R. A. (1997). Validation of the Physical Activity Questionnaire for Older Children. Pediatric Exercise Science, 9, 174–186. Kowalski, K. C., Crocker, P. R. E., & Kowalski, N. P. (1997). Convergent validity of the Physical Activity Questionnaire for Adolescents. Pediatric Exercise Science, 9, 342–352. Lee, S., Kuk, J. L., Davidson, L. E., Hudson, R., Kilpatrick, K., & Graham, T. E. (2005). Exercise without weight loss is an effective strategy for obesity reduction in obese individuals with and without type 2 diabetes. Journal of Applied Physiology, 99(3), 1220–1225. Look AHEAD Research Group (2006). The Look AHEAD study: a description of the lifestyle intervention and the evidence supporting it. Obesity (Silver Spring, Md.), 14(5), 737. Maddock, J. (2004). The relationship between obesity and the prevalence of fast food restaurants: State-level analysis. American Journal of Health Promotion, 19(2), 137–143. Malik, S. M., deVera, M. E., Fontes, P., Shaikh, O., & Ahmad, J. (2009). Outcome after liver transplantation for NASH cirrhosis. American Journal of Transplantation, 9, 782–793. Malouff, J. M., Thorsteinsson, E. B., & Schutte, N. S. (2007). The efficacy of problem solving therapy in reducing mental and physical health problems: A meta-analysis. Clinical Psychology Review, 27(1), 46–57. Morland, K., Diez Roux, A. V., & Wing, S. (2006). Supermarkets, other food stores, and obesity: The atherosclerosis risk in communities study. American Journal of Preventive Medicine, 30(4), 333–339. Mulgrew, K. W., Shaikh, U., & Nettiksimmons, J. (2011). Comparison of Parent Satisfaction with Care for Childhood Obesity Delivered Face-to-Face and by Telemedicine. Telemedicine and e-Health, 17(5), 383–387. Nemet, D., Barkan, S., Epstein, Y., Friedland, O., Kowen, G., & Eliakim, A. (2005). Short-and long-term beneficial effects of a combined dietary–behavioral–physical activity intervention for the treatment of childhood obesity. Pediatrics, 115(4), e443–e449. Nemet, D., Levi, L., Pantanowitz, M., & Eliakim, A. (2014). A combined nutritional-behavioral-physical activity intervention for the treatment of childhood obesity: A 7-year summary. Journal of Perinatal Medicine, 1–7. Nezu, A. M. (2004). Problem solving and behavior therapy revisited. Behavior Therapy, 35, 1–33.

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Nobili, V., Marcellini, M., Devito, R., Ciampalini, P., Piemonte, F., Camporcola, D., … Angulo, P. (2006). NAFLD in children: A prospective clinical-pathological study and effect of lifestyle. Hepatology, 44(2), 458–465. Nobili, V., Manco, M., Devito, R., Di Ciommo, V., Comparcola, D., Sartorelli, M. R., Piemonte, F., … Angulo, P. (2008). Lifestyle intervention and antioxidant therapy in children with nonalcoholic fatty liver disease: A randomized, controlled trial. Hepatology, 48(1), 119–128. Ogden, C. L., Carroll, M. D., Curtin, L. R., Lamb, M. M., & Flegal, K. M. (2010). Prevalence of high body mass index in US children and adolescents, 2007–2008. Journal of the American Medical Association, 303, 242–249. Perri, M. G., Nezu, A. M., McKelvey, W. F., Shermer, R. L., Renjilian, D. A., & Viegener, B. J. (2001). Relapse prevention training and problemsolving therapy in the long-term management of obesity. Journal of Consulting and Clinical Psychology, 69, 722–726. Pronk, N. P., & Wing, R. R. (2012). Physical activity and long‐term maintenance of weight loss. Obesity Research, 2(6), 587–599. Sage, N., Snowden, M., Chorlton, E., & Edeleanu, A. (2008). CBT for chronic illness and palliative care. New York: Wiley. Shaikh, U., Nettiksimmons, J., & Romano, P. (2011). Pediatric obesity management in rural clinics in california and the role of telehealth in distance education. The Journal of Rural Heath, 27, 263–269. Shaw, K., Gennat, H., O'Rourke, P., & Del Mar, C. B. (2006). Exercise for overweight or obesity.Cochrane Database of Systematic Reviews, 18(4), CD003817. Skender, M. L., Goodrick, G., Del Junco, D. J., Reeves, R. S., Darnell, L., & Gotto, A. M. (1996). Comparison of 2-year weight loss trends in behavioral treatments of obesity: Diet, exercise, and combination interventions. Journal of the American Dietetic Association, 96(4), 342–346. United States Department of Agriculture. (2013, December 11). Dietary Guidelines for Americans. Retrieved from http://www.cnpp.usda. gov/DietaryGuidelines.htm. Varni, J. W., Seid, M., & Kurtin, P. S. (2001). PedsQL (TM) 4.0: Reliability and validity of the pediatric quality of life Inventory (TM) Version 4.0 generic core scales in healthy and patient populations. Medical Care, 39(8), 800–812. Vaughn, L. M., Wagner, E., & Jacquez, F. (2013). A review of community-based participatory research in child health. The American Journal of Maternal-Child Nursing, 38(1), 48–53. This project was supported in part by a generous contribution from the Y H Mirzoeff & Sons Foundation Inc. Address correspondence to Rachel A. Annunziato, Ph.D., Department of Psychology, Fordham University, 441 E. Fordham Road, Bronx, NY 10458; e-mail: [email protected]. Received: March 2, 2013 Accepted: July 12, 2014 Available online 25 July 2014