Scurvy as a Sequela of Avoidant-Restrictive Food Intake Disorder in Autism: A Systematic Review.

OBJECTIVE: To document the clinical presentation of scurvy in children with autism spectrum disorder (ASD) and summarize the contemporary approaches to assessment and management in this population. Scurvy is a disease caused by vitamin C deficiency most often detected in populations at high risk for nutrition insufficiency (e.g., extreme poverty). Children with ASD and severe food selectivity consistent with avoidant-restrictive food intake disorder may also be at risk for scurvy. METHOD: We searched MEDLINE, CINAHL, and PsycINFO databases (1990-2018) in peer-reviewed journals for studies of children with ASD and scurvy. Inclusion criteria required confirmed diagnosis of ASD and scurvy in children (birth to 18 years) with a clear description of restrictive dietary patterns. Cases of scurvy due to other causes were excluded. We used a standardized protocol to independently code information; agreement between coders was high. RESULTS: The systematic search identified 20 case reports involving 24 children (mean age = 9 ± 3.5; 22 boys/2 girls). The eventual diagnosis of scurvy followed a wide range of negative diagnostic testing; treatment with ascorbic acid and/or a multivitamin resulted in rapid improvement. CONCLUSIONS: Symptoms of scurvy mimic other pediatric conditions (e.g., cancer). The range of diagnostic testing increased costs and healthcare risks (radiation, sedation) and delayed the diagnosis of scurvy. In children with ASD and severe food selectivity, a nutrition evaluation and laboratory testing are warranted before a more elaborate testing.

Feeding problems and gastrointestinal diseases in Down syndrome.

BACKGROUND AND METHOD: Feeding problems and gastrointestinal disorders are the most common anomalies in people with Down syndrome (DS) and have a significant impact on their daily life. This study lists the various anomalies on the basis of 504 references selected from a PubMed search in October 2018. RESULTS: The anomalies are grouped into three categories: anatomical anomalies: duodenal atresia and stenosis (3.9%), duodenal web and annular pancreas; aberrant right subclavian artery (12% of children with DS with cardiac anomaly); Hirschsprung's disease (2.76%); anorectal malformation (1.16%); congenital vascular malformations of the liver; orofacial cleft, bifid uvula (4.63%), and submucous orofacial cleft; esophageal atresia (0.5-0.9%); pyloric stenosis (0.3%); diaphragmatic hernia; malrotation of small intestine or duodenum inversum; omphalocele, gastroschisis or anomalies of the median line, anomalies of the umbilical vein; biological, immunological, and infectious anomalies: neonatal cholestasis (3.9%); neonatal hepatic fibrosis; Helicobacter pylori infection (75.8% in institutionalized children with DS, between 29.2 and 19.5% in non-institutionalized); non-alcoholic fatty liver disease (NAFLD; 82% in obese and 45% in non-obese); biliary lithiasis (6.9% under 3 years); celiac disease (6.,6%); geographical tongue (4%); hepatitis B virus sensitivity; autoimmune hepatitis and cholangitis; Crohn's disease, inflammatory bowel disease (IBD); pancreatitis; vitamin D deficiency (45.2% in Italy); functional disorders: suction, swallowing and chewing disorders (13 of 19 children with DS under 4 years); gastroesophageal reflux (47% in children with sleep apnea); achalasia (0,5% in adults); obesity (51.6% of males and 40.0% of females in Ireland) and overweight (32.0% and 14.8%); constipation (19.0%). Based on their practice, the authors insist on the following points: malformations are sometimes detected late (chronic vomiting after the introduction of food pieces, resistant constipation despite appropriate measures); prescription of preventive doses of vitamin D is advised; jaundice in a baby with DS may be retentional; in the event of transient leukemoid reaction it is vital to monitor liver function; the patient with geographic tongue must be reassured; for celiac serology there is no consensus on the staring age and the frequency, we propose every year from the age of 2; we advise to test people with DS for H. pylori infection if they are attending specialized institutions; abdominal ultrasounds must be systematic during the first months of life; detection of NAFLD is recommended; people with DS must be vaccinated against hepatitis B; breastfeeding is possible with maternal support; it is important to start speech therapy very early; feeding difficulties are often overlooked by the family and educators; gastroesophageal reflux is often pathological; preventing obesity must start from birth using body mass index for the general population; it is necessary to do everything for their meals to be joyful.

Modified-Bolus Placement as a Therapeutic Tool in the Treatment of Pediatric Feeding Disorders: Analysis From a Retrospective Chart Review.

Purpose Recognizing the need to advance the treatment of pediatric feeding disorders, an emerging area of inquiry focuses on therapeutic techniques that address expulsion and packing possibly associated with oral-motor dysfunction. In the current study, we documented the use of modified-bolus placement in the treatment of pediatric feeding disorders at an intensive, multidisciplinary day-treatment center over a 26-month period. Method The retrospective observational cohort study involved patients admitted for the assessment and treatment of chronic food refusal from August 2013 to October 2015. Results Patients (n = 23) who received modified bolus placement displayed moderate-to-severe oral-motor deficits. Use of modified-bolus placement was associated with reduced expulsion and packing of bites during meals, and treatment gains coincided with a threefold increase in grams consumed per meal. Conclusion Results provide additional support for the potential therapeutic benefits of enhancing bolus placement onto the tongue as a means to improve mealtime performance in children with pediatric feeding disorders when included as an adjunct to more traditional behavioral approaches.

Scurvy presenting with limp and weakness: a case report.

BACKGROUND: Scurvy is one of the oldest diseases known to mankind. Although presently rare in the developed world, scurvy was a common potentially fatal disease. In recent times, the most common risk factors for scurvy include alcoholism, low socioeconomic status, and severely poor nutrition or dietary restriction secondary to psychiatric illness or developmental disorders. Our case demonstrates the importance of having a high index of clinical suspicion of an uncommon disease in developed countries and emphasizes the necessity of a dietary screening that could potentially reduce extensive work-up in patients with nonspecific complaints. CASE PRESENTATION: We report a case of a 3-year-old previously healthy female originally seen in the rheumatology clinic for limp. She developed weakness and was admitted to the hospital for further evaluation. She underwent extensive diagnostic testing including blood work, magnetic resonance imaging, lumbar puncture, electromyogram, and nerve conduction studies. Ultimately, her vitamin C level returned undetectable. She had immediate and complete improvement upon starting vitamin C supplementation. CONCLUSIONS: Despite being developmentally appropriate, our patient's refusal to eat fruits or vegetables had limited her diet, emphasizing the importance of obtaining a diet history in a child presenting with an unknown diagnosis. In addition, our patient had no other characteristic features of scurvy, which further supports the need to consider this diagnosis in a child presenting with lower extremity weakness or abnormal gait.

Parental concern of feeding difficulty predicts poor growth status in their child.

BACKGROUND: Parents often express concerns about feeding difficulties in their child. We hypothesized that these parental concerns were associated with adverse growth status in early childhood. We aimed to determine the prevalence of such concerns and whether these concerns were associated with adverse growth status in early childhood. METHODS: We performed a cross-sectional study among healthy children aged 12-36 months attending three well-baby clinics in three urban areas in Malaysia and Singapore between December 2016 and February 2017. Parents were interviewed for concerns about their child's feeding and presence of behavioral and organic red flags for feeding difficulties. We defined growth faltering as weight-for-age < 3rd centile and short stature as height-for-age < 3rd centile according to World Health Organization Growth Standards. RESULTS: Of the 303 children studied (boys = 160, 52.8%; mean [± SD] chronological age at interview 21.3 [± 4.0] months), 13% (n = 38/292) had growth faltering and 19.5% (n = 50/256) had short stature. Overall, 36.3% (n = 110) of parents expressed concerns about their child's feeding behavior. Sixty-eight percent (n = 206) of parents reported presence of at least one behavioral and 18.5% (n = 56) had at least one organic red flag for feeding difficulties, respectively. 9.9% (n = 30) had both behavioral and organic red flags for feeding difficulties. Growth faltering was significantly associated with parental concern about feeding (odds ratio [OR] 3.049, p < 0.001), food refusal (OR 4.047, p < 0.001) and presence of at least one organic red flag (OR 2.625, p = 0.012). CONCLUSION: We found that parental concerns about their child's feeding to be common. Presence of parental concern, food refusal in the child and presence of organic red flags for feeding difficulties are associated growth faltering in early childhood.

Parent Training for Feeding Problems in Children With Autism Spectrum Disorder: Initial Randomized Trial.

Objective: Many children with autism spectrum disorder (ASD) have feeding and mealtime problems. To address these, we conducted a pilot randomized trial of a new 11-session, individually delivered parent training program that integrated behavioral strategies and nutritional guidance (PT-F). Methods: Forty-two young children (age: 2 to 7-11 years) with ASD and feeding problems were assigned to 11 sessions of PT-F intervention over 20 weeks or a waitlist control. Outcomes included attendance, parent satisfaction, therapist fidelity, and preliminary assessments of child and parent outcomes. Results: Of the 21 PT-F families, attendance was high (85%) as was parent satisfaction (94% would recommend to others). Treatment fidelity was also high (97%-therapist integrity; 94%-parent adherence). Compared with waitlist, children whose parents participated in PT-F showed significantly greater reductions on the two parent-completed primary outcomes (Brief Autism Mealtime Behavior Inventory-Revised; Twald = -2.79; p = .003; About Your Child's Eating; Twald = -3.58; p = .001). On the independent evaluator-completed secondary eating outcome, the Clinical Global Impression-Improvement, 48.8% of the participants in PT-F were rated as "responders" compared with 0% in waitlist (p = .006). General child disruptive behavior outcomes decreased more in PT-F but not significantly. Parent outcomes of caregiver stress showed nonsignificant trends favoring PT-F with moderate to small effect sizes. Conclusions: This trial provides evidence for feasibility, satisfaction, and fidelity of implementation of PT-F for feeding problems in young children with ASD. Feeding outcomes also appeared favorable and lends support for conducting a larger efficacy trial.

Failure to Replicate the Effects of the High-Probability Instructional Sequence on Feeding in Children With Autism and Food Selectivity.

Behavioral intervention has positive effects on feeding problems of children with autism and food selectivity (FS), and researchers have evaluated a variety of specific behavioral interventions. Confidence in the effects of some specific interventions on feeding such as the high-probability instructional sequence (HPS) is limited by a lack of replication. Therefore, we assessed the generality of the HPS by replicating the intervention in children with autism and FS. Contrary to prior research, the HPS did not improve feeding responses for three consecutive children enrolled in the study. We discuss the results in relation to publishing failures to replicate without experimental control in applied behavior analysis research.

Immunocognitive Model of Depression Secondary to Anxiety in Adolescents.

There is evidence that anxiety precedes the onset of depression and that rumination contributes to this risk pathway in adolescence. This study examined inflammatory biomarkers as mediators in a risk model of depressive symptoms secondary to anxiety symptoms among adolescents who ruminate. A sample of 140 adolescents (52% female, 54% African American, 40% Caucasian, 6% biracial, mean age at T1 = 16.5 years, SD = 1.2 years) provided blood samples on two visits (T1 and T2; mean time between T1 and T2 = 13.5 months, SD = 5.9 months). Self-report anxiety, depression, and rumination measures were given at T1 and the depression measure was given again at a third visit (T3, mean months since T1 = 26.0 months, SD = 9.0 months). Higher anxiety predicted more interleukin-6, but not more C-reactive protein, for adolescents with high levels of rumination. Moderated mediation analyses (N for analysis after removing cases with missing data and outliers = 86) indicated that interleukin-6, but not C-reactive protein, at T2 mediated the relationship between anxiety symptoms at T1 and depressive symptoms at T3, conditional on rumination. Anxiety and rumination interacted such that, as rumination increased, anxiety predicted greater inflammation and depressive symptoms. These results demonstrate that established cognitive vulnerabilities for the development of depressive symptoms secondary to anxiety symptoms in adolescence might indirectly operate though biological mechanisms such as inflammation. In addition to highlighting risk factors and potential treatment targets for depression, this study suggests a potential biological mechanism underlying the effects of psychotherapies that reduce rumination on negative affect (e.g., cognitive behavioral therapy).

Improvements in mealtime behaviors of children with special needs following a day-center-based behavioral intervention for feeding problems.

AIM: Feeding problems are documented in several developmental disabilities. This paper aims to present early results from the implementation of a day-center, multidisciplinary, intensive, behaviorally-based treatment package for feeding problems, delivered to children with developmental disabilities, namely Autism Spectrum Disorder (ASD) and Intellectual Disability (ID). METHODS: This is a quasi-experimental design study. Both an AB design (measures were administered at pre- and post-treatment times) and a comparison design were used; a follow-up was carried out after one year from the end of the treatment. A total of 18 children, 8 with ASD and 10 with ID, were included in the study. Body weight, count of food types and textures accepted, length of the meal, count of problem behaviour topographies, count of individuals with proper chewing and BAMBI-18 scores were the measures chosen to check the effectiveness of the treatment. RESULTS: Statistically significant differences between pre-, post-treatment and follow-up were found, with decreased problem behavior during meals and increased body weight, effective chewing and food variety (type and texture). CONCLUSIONS: Despite its relatively short duration, this treatment package appeared to be effective in improving behaviors at mealtime in both children with ASD and ID; if confirmed by future studies, the program could represent an interesting model for treating feeding problems in children with special needs, because it is implemented in a day service (with lower costs compared to residential or hospital services), takes advantage from family co-therapy, and can be integrated in a global psychoeducational program.

Riesgos asociados a la introducción directa de alimentos sólidos en lactantes / Risks associated with the direct introduction of solid food in infants

No disponible

Comparing behavioral treatment of feeding difficulties and tube dependence in children with cerebral palsy and autism spectrum disorder.

BACKGROUND: Feeding disorders are multifaceted with behavioral components often contributing to the development and continuation of food refusal. In these cases, behavioral interventions are effective in treating feeding problems, even when medical or oral motor components are also involved. Although behavioral interventions for feeding problems are frequently employed with children with autism, they are less commonly discussed for children with cerebral palsy. OBJECTIVE: The purpose of this study was to compare the effectiveness of using applied behavior analytic interventions to address feeding difficulties and tube dependence in children with autism and children with cerebral palsy. METHOD: Children ages 1 to 12 years who were enrolled in an intensive feeding program between 2003 and 2013, where they received individualized behavioral treatment, participated. RESULTS: Behavioral treatment components were similar across groups, predominately consisting of escape extinction (e.g., nonremoval of the spoon) and differential reinforcement. For both groups, behavioral treatment was similarly effective in increasing gram consumption and in decreasing refusal and negative vocalizations. A high percentage of individualized goals were met by both groups as well as high caregiver satisfaction reported. CONCLUSIONS: Behavioral interventions for food refusal are effective for children with cerebral palsy with behavioral refusal, just as they are for children with autism.

Prevalence of Auditory Problems in Children With Feeding and Swallowing Disorders.

Purpose: Although an interdisciplinary approach is recommended for assessment and management of feeding or swallowing difficulties, audiologists are not always included in the interdisciplinary team. The purpose of this study is to report the prevalence of middle ear and hearing problems in children with feeding and swallowing disorders and to compare this prevalence with that in typical children. Method: A total of 103 children were included in the study: 44 children with feeding and swallowing disorders and 59 children without any such disorders. Audiological examinations included case-history information, visualization of the ear canals through otoscopy, middle ear evaluation through tympanometry, and hearing screenings using an audiometer. Results: The odds of excessive cerumen (p = .0000, small effect size), middle ear dysfunction (p = .0148, small effect size), and hearing screening failure (p = .0000, large effect size) were 22.14%, 2.97%, and 13.5% higher, respectively, in children with feeding and swallowing disorders compared with typically developing children. Conclusion: The significantly higher prevalence of hearing problems in children with feeding and swallowing disorders compared with typically developing children suggests that inclusion of an audiologist on the interdisciplinary team is likely to improve overall interventional outcomes for children with feeding and swallowing disorders.

Secretive eating among youth with overweight or obesity.

PURPOSE: Secretive eating, characterized by eating privately to conceal being seen, may reflect eating- and/or body-related shame, be associated with depression, and correlate with binge eating, which predicts weight gain and eating disorder onset. Increasing understanding of secretive eating in youth may improve weight status and reduce eating disorder risk. This study evaluated the prevalence and correlates of secretive eating in youth with overweight or obesity. METHODS: Youth (N = 577) presented to five research/clinical institutions. Using a cross-sectional design, secretive eating was evaluated in relation to eating-related and general psychopathology via linear and logistic regression analyses. RESULTS: Secretive eating was endorsed by 111 youth, who were, on average, older than youth who denied secretive eating (mean age = 12.07 ± 2.83 versus 10.97 ± 2.31). Controlling for study site and age, youth who endorsed secretive eating had higher eating-related psychopathology and were more likely to endorse loss of control eating and purging than their counterparts who did not endorse secretive eating. Groups did not differ in excessive exercise or behavioral problems. Dietary restraint and purging were elevated among adolescents (≥13y) but not children (<13y) who endorsed secretive eating; depression was elevated among children, but not adolescents, who endorsed secretive eating. CONCLUSIONS: Secretive eating may portend heightened risk for eating disorders, and correlates of secretive eating may differ across pediatric development. Screening for secretive eating may inform identification of problematic eating behaviors, and understanding factors motivating secretive eating may improve intervention tailoring.

Disturbed eating behaviors in adolescents with type 1 diabetes. How to screen for yellow flags in clinical practice?

BACKGROUND: Adolescents with type 1 diabetes are at an increased risk of disturbed eating behaviors (DEBs). OBJECTIVE: The aims of this study are to (i) explore the prevalence of DEBs and associated 'yellow flags', and (ii) establish concordance between adolescents-parents and adolescents-clinicians with respect to DEBs. METHODS: Adolescents (11-16 yr) and parents completed questionnaires. A stepwise approach was used to assess DEBs: only adolescents whose answers raised psychological yellow flags for DEBs completed the Diabetes Eating Problems Scale - Revised and questions from the AHEAD study. Parents and clinicians shared their observations regarding possible DEBs. Kruskal-Wallis tests, post hoc Mann-Whitney U test, and chi-squared tests were utilized to examine clinical yellow flags. Cohen's kappa was used to assess concordance. RESULTS: Of 103 adolescents participated (51.5% girls), answers of 47 (46.5%) raised psychological yellow flags, indicating body and weight concerns. A total of 8% scored above cut-off for DEBs. Clinical yellow flags were elevated glycated hemoglobin A1c (p = 0.004), older age (p = 0.034), dieting frequency (p = 0.001), reduced quality of life (p = 0.007), less diabetes self-confidence (p = 0.015), worsened diabetes management (p < 0.001), and body dissatisfaction (p < 0.001). Body Mass Index (BMI) z-scores and gender were no yellow flags. Concordance between parents and adolescents was slight (k = 0.126 and 0.141), and clinicians and adolescents was fair (k = 0.332). DISCUSSION: Half of the adolescents reported body and weight concerns, less than 1 in 10 reported DEBs. Screening for yellow flags for DEBs as a part of clinical routine using a stepwise approach and early assistance is recommended to prevent onset or deterioration of DEBs.

Reliability and validity of a tool to measure the severity of tongue thrust in children: the Tongue Thrust Rating Scale.

This study aimed to develop a scale called Tongue Thrust Rating Scale (TTRS), which categorised tongue thrust in children in terms of its severity during swallowing, and to investigate its validity and reliability. The study describes the developmental phase of the TTRS and presented its content and criterion-based validity and interobserver and intra-observer reliability. For content validation, seven experts assessed the steps in the scale over two Delphi rounds. Two physical therapists evaluated videos of 50 children with cerebral palsy (mean age, 57·9 ± 16·8 months), using the TTRS to test criterion-based validity, interobserver and intra-observer reliability. The Karaduman Chewing Performance Scale (KCPS) and Drooling Severity and Frequency Scale (DSFS) were used for criterion-based validity. All the TTRS steps were deemed necessary. The content validity index was 0·857. A very strong positive correlation was found between two examinations by one physical therapist, which indicated intra-observer reliability (r = 0·938, P < 0·001). A very strong positive correlation was also found between the TTRS scores of two physical therapists, indicating interobserver reliability (r = 0·892, P < 0·001). There was also a strong positive correlation between the TTRS and KCPS (r = 0·724, P < 0·001) and a very strong positive correlation between the TTRS scores and DSFS (r = 0·822 and r = 0·755; P < 0·001). These results demonstrated the criterion-based validity of the TTRS. The TTRS is a valid, reliable and clinically easy-to-use functional instrument to document the severity of tongue thrust in children.

Pediatric rumination subtypes: A study using high-resolution esophageal manometry with impedance.

BACKGROUND: The differential diagnosis of intractable reflux in children includes rumination syndrome, but confirming the diagnosis using antroduodenal manometry is invasive, is costly, and requires anesthesia. High-resolution esophageal manometry with impedance (HRM-MII) overcomes these limitations, and the goal of this study is to validate the use of HRM-MII as a diagnostic tool for rumination and to describe the subtypes of pediatric rumination. METHODS: We reviewed the HRM-MII tracings of 21 children presenting with symptoms of intractable reflux in whom rumination was being considered. Patients underwent a standard and post-prandial HRM-MII. Peak intraluminal esophageal pressures, baseline gastric and thoracic pressures, and the timing of the R wave relative to LES relaxations and bolus flow were recorded. Chi-square analyses were used for comparison of proportions and means were compared using t-tests or non-parametric equivalent. KEY RESULTS: Forty-one (55.5%) primary and 33 (44.5%) secondary rumination episodes were seen. Three types of primary rumination were identified: i) LES relaxation without retrograde flow preceding the R wave (51% of episodes); ii) LES relaxation after the R wave (20% of episodes); and iii) R waves with no LES relaxation (29% of episodes). Eleven patients had rumination episodes with a peak gastric pressure <30 mm Hg. A total of 44 (60%) rumination episodes occurred during the standard HRM-MII, and 30 (40%) occurred during or after the meal. CONCLUSIONS & INFERENCES: HRM-MII can accurately diagnose rumination in children. We identify three types of primary rumination which may provide insight into therapeutic response.

Rumination Syndrome and Dental Erosions in Children.

Rumination syndrome is the effortless regurgitation of recently ingested food with subsequent reswallowing or spitting out. Dental erosion (DE) affects 2% to 5% of the population. DE is defined as loss of tooth structure by a chemical process that does not involve bacteria. Our objective was to compare the frequency of DE among children with rumination syndrome with healthy controls. We enrolled 30 patients 4 to 21 years of age diagnosed with rumination syndrome, and 30 age- and sex-matched healthy control subjects. Patients were evaluated by pediatric dentists for presence of DE with Taji et al a validated grading system. Patients with rumination were more likely to have DE (P < 0.001). Of patients with rumination syndrome, 23 (77%) had DE, compared with 4 (13%) control subjects. DEs are more frequent in patients with rumination syndrome.

Effects of emotional acceptance and rumination on media-induced body dissatisfaction in anorexia and bulimia nervosa.

INTRODUCTION: Body dissatisfaction is an important risk and maintaining factor for eating disorders. The aim of the present study was to experimentally test the effects of two emotion regulation strategies - acceptance and rumination - on media-induced body dissatisfaction in eating disorders. METHOD: After watching pictures of thin models, women with anorexia nervosa (AN; n = 39) and bulimia nervosa (BN; n = 39) were encouraged to either use emotional acceptance or rumination to cope with their feelings. Body dissatisfaction and mood were repeatedly assessed. RESULTS: Acceptance significantly improved body dissatisfaction in women with BN. Rumination led to a significant increase of body dissatisfaction in both eating disorder groups. Results were independent from mood changes. DISCUSSION: Findings highlight the importance ruminative thinking may have in the aggravation of dissatisfaction with the own body in AN and BN. Results suggest that emotional acceptance is a useful strategy to regulate body dissatisfaction after exposure to thin-ideal media in BN.

Gastrointestinal and nutritional problems in neurologically impaired children.

The current increasing survival of children with severe central nervous system damage has created a major challenge for medical care. Gastrointestinal and nutritional problems in neurologically impaired children have been recently recognized as an integral part of their disease, often leading to growth failure and worsened quality of life for both children and caregivers. Nutritional support is essential for the optimal care of these children. Undernourished handicapped children might not respond properly to intercurrent diseases and suffer unnecessarily. On the other hand, restoring a normal nutritional status results in a better quality of life in many. The easiest and least invasive method to increase energy intake is to improve oral intake. However, oral intake can be maintained as long as there is no risk of aspiration, the child is growing well and the time required to feed the child remains within acceptable limits. When oral intake is unsafe, insufficient or too time consuming, enteral nutrition should be initiated. Damage to the developing central nervous system may result in significant dysfunction in the gastrointestinal tract and is reflected in impairment in oral-motor function, rumination, gastro-oesophageal reflux (GER), with or without aspiration, delayed gastric emptying and constipation. These problems can all potentially contribute to feeding difficulty in disabled children, carrying further challenging long-term management issues.