Psychological and social impacts on carers of children with a gastrostomy: a systematic review.

To determine the psychological and social impacts of a gastrostomy in childhood on carers and families. A systematic search of OVID, Medline and Embase was undertaken using the subject headings and word variants for 'gastrostomy', 'children' and 'carers'. Studies included were those describing the impact of gastrostomies in children on family and carers, published in English. 564 articles were identified. After exclusion of duplicates, abstract and full text screening, 25 were included. Carer anxiety increases in the period leading up to, and for a short period following gastrostomy insertion. 3-6 months following gastrostomy insertion, anxiety reduced (reduced State-Trait Anxiety Inventory scores), carer quality of life improved (higher Quality of Life Scale scores), and carer satisfaction with the child's gastrostomy increased (improved Satisfaction Questionnaire with Gastrostomy Feeding scores). Reported changes in carer quality of life in the longer term following a child's gastrostomy insertion were mixed. The social and psychological burden on caregivers of a gastrostomy in childhood varies over time. There is evidence that paediatric gastrostomies have positive effects on carers' psychological and social well-being; however, aspects of carers' quality of life remain impaired. Carer education and support are vital to reduce the burden placed on carers.

Complete Resection of Necrotic Bowel Improves Survival in NEC Without Compromising Enteral Autonomy.

AIMS: Controversy persists regarding operative strategy for necrotising enterocolitis (NEC). Some surgeons advocate resecting all necrotic bowel, whilst others defunction with a stoma, leaving diseased bowel in situ to preserve bowel length. We reviewed our institutional experience of both approaches. METHODS: Neonates undergoing laparotomy for NEC May 2015-2019 were identified. Data extracted from electronic records included: demographics, neonatal Sequential Organ Failure Assessment (nSOFA) score at surgery, operative findings, and procedure performed. Neonates were assigned to two groups according to operative strategy: complete resection of necrotic bowel (CR) or necrotic bowel left in situ (LIS). Primary outcome was survival, and secondary outcome was enteral autonomy. Outcomes were compared between groups. RESULTS: Fifty neonates were identified. Six were excluded: 4 with NEC totalis and 2 with no visible necrosis or histological confirmation of NEC. Of the 44 remaining neonates, 27 were in the CR group and 17 in the LIS group. 32 neonates survived to discharge (73%). On univariate analysis, survival was associated with lower nSOFA score (P = 0.003), complete resection of necrotic bowel (OR 9.0, 95% CI [1.94-41.65]), and being born outside the surgical centre (OR 5.11 [1.23-21.28]). On Cox regression multivariate analysis, complete resection was still strongly associated with survival (OR 4.87 [1.51-15.70]). 28 of the 32 survivors (88%) achieved enteral autonomy. There was no association between operative approach and enteral autonomy (P = 0.373), or time to achieve this. CONCLUSION: Complete resection of necrotic bowel during surgery for NEC significantly improves likelihood of surviving without negatively impacting remaining bowel function. LEVEL OF EVIDENCE: III.

Assessing micro- vs macro-costing approaches for treating appendicitis in children with appendicectomy or non-operatively.

OBJECTIVES: We conducted a health economic sub-study within a feasibility RCT comparing a non-operative treatment pathway as an alternative to appendicectomy for the treatment of uncomplicated acute appendicitis in children. The objectives were to understand and assess data collection tools and methods and to determine indicative costs and benefits assessing the feasibility of conducting a full economic evaluation within the definitive trial. METHODS: We compared different methods of estimating treatment costs including micro-costing, hospital administrative data (PLICS) and health system (NHS) reference costs. We compared two different HRQoL instruments (CHU-9D and EQ-5D-5L) in terms of data completeness and sensitivity to change over time, including potential ceiling effects. We also explored how the timing of data collection and duration of the analysis could affect QALYs (Quality Adjusted Life Years) and the results of the cost-utility analysis (CUA) within the future RCT. RESULTS: Using a micro-costing approach, the total per treatment costs were in alignment with hospital administrative data (PLICS). Average health system reference cost data (macro-costing using NHS costs) could potentially underestimate these treatment costs, particularly for non-operative treatment. Costs incurred following hospital discharge in the primary care setting were minimal, and limited family borne costs were reported by parents/carers. While both HRQoL instruments performed relatively well, our results highlight the problem of ceiling effect and the importance of the timing of data collection and the duration of the analysis in any future assessment using QALYs and CUA. CONCLUSIONS: We highlighted the importance of obtaining accurate individual-patient cost data when conducting economic evaluations. Our results suggest that timing of data collection and duration of the assessment are important considerations when evaluating cost-effectiveness and reporting cost per QALY. CLINICAL TRIAL REGISTRATION: Current Controlled Trials ISRCTN15830435.

The Community Cost of Maintaining Gastrostomies in Pediatric Patients.

The use of gastrostomies in children is increasing and their need for long-term management following insertion presents a potentially significant financial and resource challenge to local healthcare systems. Objectives: The aim of this study was to determine the yearly cost of maintaining a gastrostomy in a pediatric patient. Methods: A retrospective, bottom-up cost-analysis was conducted in a cohort of 180 patients with gastrostomies aged 0-19 years. One in 5 patients were randomly selected for individual cost analysis (n = 36). Their electronic health record was interrogated from the period of March 1, 2019-March 1, 2020. Costs included in the analysis were staff contact time from the community nursing and nutrition teams, and equipment costs. Results: The mean cost of maintaining a pediatric gastrostomy across all ages was £709.87 (SD 403.18) per year. Mean annual cost varied by age, underlying diagnosis, and gastrostomy device, but this variation was only statistically significant for the type of device, with Mic-Key buttons having a mean annual cost of £834.66 (SD 307.85), Mini buttons £799.06 (SD 395.01), and percutaneous endoscopic gastrostomy tubes £279.34 (SD 297.45; P = 0.004). Conclusion: The mean cost of maintaining a gastrostomy in a pediatric patient is just over £700 per year. The cost is the highest as a child enters adulthood. Button devices have higher maintenance costs compared with percutaneous endoscopic gastrostomy tubes.

Point Prevalence of Gastrostomy in a Paediatric Population.

OBJECTIVE: The aim of the study was to determine the prevalence of gastrostomy in a paediatric population. METHODS: A population-based cross-sectional point prevalence study of paediatric gastrostomy was performed. Patients included were ages 0 to 19 years, living within 3 inner-city London boroughs; Southwark, Lambeth, and Lewisham. Patients were identified as having a gastrostomy in situ via Home Enteral Nutrition (HEN) and community nursing databases. Electronic healthcare records were scrutinised to confirm current use of a gastrostomy. The main outcome measures were the point prevalence of gastrostomy in the paediatric population (gastrostomies/100,000 children), primary diagnosis, indication underlying gastrostomy insertion, and age at insertion. RESULTS: The total population studied was 946,709, of whom 213,920 were of age 0 to 19 years. Of these, 179 had a gastrostomy in situ giving a point prevalence for gastrostomy in the paediatric population of 83.7 (95% confidence interval [CI]: 71.4-96.0)/100,000 children. This varied between age groups: 0 to 4 years: 79.6 (57.3-102.0)/100,000, 5 to 9 years: 116.3 (88.7-143.9)/100,000, 10 to 14: years 87.9 (61.9-113.9)/100,000 and 15 to 19: years 41.4 (22.1-60.1)/100,000. The most common primary diagnoses were neurological disorders (57.1%), and structural abnormalities (16.2%). Unsafe swallow was the most common indication (61%), followed by nutritional or fluid supplementation (28.6%), and behavioural reasons (8.7%). The majority (85.1%) of gastrostomies were inserted under the age of 2 years. CONCLUSIONS: This is the first UK population-based study of paediatric gastrostomy, identifying a point prevalence of 84/100,000 children. The peak prevalence is in children ages 5 to 9 years. Gastrostomy insertion after a child reaches school age is uncommon.

Application of the Clavien-Dindo classification to a pediatric surgical network.

INTRODUCTION: A comprehensive validated system to evaluate surgical complications is required in our specialty to facilitate comparison and audit. The Clavien-Dindo (CD) classification of post-surgical complications was originally described in an adult general surgical setting in 1992 and has become widely used. We aimed to apply this to a pediatric surgical setting. METHODS: Data were collected on emergency and elective surgical activity together with complications in a prospective audit over a recent 4-month period in three geographical conjoined regional pediatric surgical units (including two major trauma centres). Briefly the CD classification codes complications according to degree of harm and magnitude of intervention required [I - V (death) with III and IV sub-divided according to whether general anesthesia was needed]. Length of stay and mode of admission were recorded. Data are given as median (range). Non-parametric comparison was used, and a p value of <0.05 was regarded as significant. RESULTS: During the period JULY - OCT 2018 (inclusive), there were 1822 admissions (elective, n = 1186: emergency, n = 636) and 1556 operations (elective, n = 1189, and of these 393 were urological). There were 69 patient complications: CDI (n = 7), CD-II (n = 19), CD-IIIa (n = 4), CD-IIIb (n = 28), CD-IV (n = 4), CD-V (n = 7). Deaths were principally in neonates and due to NEC (n = 6) at 2.5 (1-140) days post-operatively. There was a single post-traumatic death in an adolescent. LOS was 9 (0-217) days in CD I-IV. The incidence of any complication was 4.4%, of serious complication (defined as ≥CD III) 2.6% (A = 2.1%, B = 2.0%, and C = 3.2%: p = 0.16), and of death 0.45%. The most frequent complications were wound infection (n = 12) and post-appendicectomy collections/abscess (n = 10). CONCLUSIONS: This appears to be the 1st report of the C-D classification in a general pediatric surgery network and can be considered a benchmark. The risk of death or serious harm is very low in such a practice. TYPE OF STUDY: Prospective Cohort Study. LEVEL OF EVIDENCE: IIb.

Prenatal detection of esophageal atresia: A systematic review and meta-analysis.

INTRODUCTION: The primary aim of this systematic review was to quantify the diagnostic performance of ultrasound, magnetic resonance imaging and amniotic fluid analysis in detecting esophageal atresia prenatally. The secondary aim was to explore the accuracy of individual imaging signs in identifying this anomaly. MATERIAL AND METHODS: MEDLINE, Embase and Cochrane databases were searched. The quality of studies was assessed using the revised tool for the quality assessment of diagnostic accuracy studies. Summary estimates of sensitivity, specificity, positive and negative likelihood ratios, and diagnostic odds ratio for the predictive accuracy of ultrasound, magnetic resonance imaging and amniotic fluid analysis in detecting esophageal atresia were computed using the hierarchical summary receiver operating characteristic or DerSimonian-Laird random-effect model, according to the number of studies included in each analysis. PROSPERO registration number: CRD42017055828. RESULTS: Twenty studies (73 246 fetuses, 1760 affected by esophageal atresia) were included. Overall, prenatal ultrasound had a sensitivity of 31.7%. Only two studies reported all data for diagnostic accuracy; based on these studies, prenatal ultrasound had a sensitivity of 41.9%, a specificity of 99.9%, a positive likelihood ratio of 88.1, a negative likelihood ratio of 0.58 and a diagnostic odds ratio of 153.7. Prenatal ultrasound correctly identified 77.9% of cases with esophageal atresia and 21.9% esophageal atresia with an associated tracheo-esophageal fistula. Polyhydramnios was present in 56.3% of cases affected by esophageal atresia, and a small or absent stomach was identified in 50.0% cases. When performed following a suspicious ultrasound, fetal magnetic resonance imaging had an good overall diagnostic accuracy for esophageal atresia, with a sensitivity of 94.7%, a specificity of 89.3%, a positive likelihood ratio of 8.8, a negative likelihood ratio of 0.06 and a diagnostic odds ratio of 149.3. Finally, amniotic fluid analysis with an esophageal atresia index ≥3 had a sensitivity of 89.9% and a specificity of 99.6% in detecting esophageal atresia. CONCLUSIONS: Ultrasound alone is a poor diagnostic tool for identifying esophageal atresia prenatally, and has a high rate of false positive diagnoses. Magnetic resonance imaging and amniotic fluid analysis have high diagnostic accuracy for esophageal atresia. We would recommend their use following a suspicious ultrasound.

Surgical Site Infection in a Tertiary Neonatal Surgery Centre.

INTRODUCTION: Surgical site infection (SSI) is a key performance indicator to assess the quality of surgical care. Incidence and risk factors for SSI in neonatal surgery are lacking in the literature. AIM: To define the incidence of SSI and possible risk factors in a tertiary neonatal surgery centre. MATERIALS AND METHODS: This is a prospective cohort study of all the neonates who underwent abdominal and thoracic surgery between March 2012 and October 2016. The variables analyzed were gender, gestational age, birth weight, age at surgery, preoperative stay in neonatal intensive care unit, type of surgery, length of stay, and microorganisms isolated from the wounds. Statistical analysis was done with chi-square, Student's t- or Mann-Whitney U-tests. A logistic regression model was used to evaluate determinants of risk for SSI; variables were analyzed both with univariate and multivariate models. For the length of hospital stay, a logistic regression model was performed with independent variables. RESULTS: A total of 244 neonates underwent 319 surgical procedures. The overall incidence of SSIs was 43/319 (13.5%). The only statistical differences between neonates with and without SSI were preoperative stay (<4 days vs. ≥4 days, p < 0.01) and length of hospital stay (<30 days vs. ≥30 days, p < 0.01). A pre-operative stay longer than 4 days was associated with almost three times increased risk of SSI (odds ratio [OR] 2.96, 95% confidence interval [CI] 1.05-8.34, p = 0.0407). Gastrointestinal procedures were associated with more than ten times the risk of SSI compared with other procedures (OR 10.17, 95% CI 3.82-27.10, p < 0.0001). Gastroschisis closure and necrotizing enterocolitis (NEC) laparotomies had the highest incidence SSI (54% and 62%, respectively). The risk of longer length of hospital stay after SSI was more than three times higher (OR = 3.36, 95%CI 1.63-6.94, p = 0.001). CONCLUSION: This is the first article benchmarking the incidence of SSI in neonatal surgery in the United Kingdom. A preoperative stay ≥4 days and gastrointestinal procedures were independent risk factors for SSI. More research is needed to develop strategies to reduce SSI in selected neonatal procedures.

Laparoscopic Appendectomy for Chronic Right Iliac Fossa Pain: Correlating Histology with Outcome.

BACKGROUND: Chronic right iliac fossa (CRIF) pain in children is associated with repeated hospital attendance, inconclusive investigations, and missed school days. There is increasing evidence for the role of laparoscopic appendectomy for the management of CRIF pain. However, currently there is no substantial evidence to correlate histological changes identified in the appendixes removed with resolution of pain. METHOD: This study collected prospective data for a single surgeon's series of laparoscopic appendectomy performed for CRIF pain between 2003 and 2014. RESULTS: Forty patients with a median age of 13 years (range 5-19 years) underwent laparoscopic appendectomy for CRIF pain. Twenty-nine patients (73%) had macroscopically normal appendixes, but histological changes were identified in 28 patients (70%). Other findings at laparoscopy included gynecological pathology (ovarian/para-ovarian cysts), unfixed mobile caecum, and a well-defined peritoneal band extending from the caecum to the anterolateral abdominal wall (caeco-peritoneal band). Patients were followed up for a median of 2 months (range 1-15 months). All patients reported resolution of CRIF pain. DISCUSSION: Laparoscopic appendectomy appears to be an effective treatment for CRIF pain, while also providing the opportunity to diagnose and treat potential sources of CRIF pain other than the appendix. Consistent with the literature, a small number of patients appear to have resolution of pain, despite no histological change being identified in the appendix removed and no other pathology having been identified. The same histological changes commonly seen have also been demonstrated in appendixes removed incidentally. Caution therefore should be applied when interpreting the significance of histological changes seen.

Can the Presence of Crystalluria Predict Stone Formation in Patients with Cystinuria?

OBJECTIVES: To determine the feasibility of crystalluria as a biomarker for stone disease in patients with cystinuria. PATIENTS AND METHODS: All patients attending a multidisciplinary cystinuria clinic provided early morning urine (EMU) and clinic urine (CU) samples for crystal measurement over a 2-year period (August 1, 2010, to July 31, 2012). Association between presence of crystals, presence of stone(s), and new stone growth (NSG) was determined using the chi-square test. Crystal numbers in EMU and CU were compared in patients with stones/NSG and no stones/stable disease using the Mann-Whitney U test. RESULTS: There was a statistically significant difference between the presence of crystalluria and presence of stones for CU (chi-square test = 5.86, df = 1, p = 0.02) but not EMU (chi-square test = 1.92, df = 1, p = 0.17) and between the presence of crystalluria and NSG for CU (chi-square test = 8.10, df = 1, p = 0.004) but not EMU (chi-square test = 1.32, df = 1, p = 0.25). Patients with stones and NSG have higher levels of crystalluria in CU than patients with no stones or stable disease (stones, median = 41, interquartile range [IQR] = 600 vs median = 0, IQR = 21, p = 0.01; NSG, median = 49, IQR = 525 vs median = 0, IQR = 40, p = 0.01). CONCLUSION: The presence of crystalluria in CU samples is associated with the presence of stones. Crystalluria is comparable to ultrasound and may serve as a useful adjunct to predict whether a patient with cystinuria has stones, which could guide the frequency of clinic review and imaging.

The genetic diversity of cystinuria in a UK population of patients.

OBJECTIVE: To examine the genetic mutations in the first UK cohort of patients with cystinuria with preliminary genotype/phenotype correlation. PATIENTS AND METHODS: DNA sequencing and multiplex ligation-dependent probe amplification (MLPA) were used to identify the mutations in 74 patients in a specialist cystinuria clinic in the UK. Patients with type A cystinuria were classified into two groups: Group M patients had at least one missense mutation and Group N patients had two alleles of all other types of mutations including frameshift, splice site, nonsense, deletions and duplications. The levels of urinary dibasic amino acids, age at presentation of disease, number of stone episodes and interventions were compared between patients in the two groups using the Mann-Whitney U-test. RESULTS: In all, 41 patients had type A cystinuria, including one patient with a variant of unknown significance and 23 patients had type B cystinuria, including six patients with variants of unknown significance. One patient had three sequence variants in SLC7A9; however, two are of unknown significance. Three patients had type AB cystinuria. Three had a single mutation in SLC7A9. No identified mutations were found in three patients in either gene. There were a total of 88 mutations in SLC3A1 and 55 mutations in SLC7A9. There were 23 pathogenic mutations identified in our UK cohort of patients not previously published. In patients with type A cystinuria, the presence of a missense mutation correlated to lower levels of urinary lysine (mean [SE] 611.9 [22.65] vs 752.3 [46.39] millimoles per mole of creatinine [mM/MC]; P=0.02), arginine (194.8 [24.83] vs 397.7 [15.32] mM/MC; P<0.001) and ornithine (109.2 [7.40] vs 146.6 [12.7] mM/MC; P=0.02). There was no difference in the levels of urinary cystine (182.1 [8.89] vs 207.2 [19.23] mM/MC; P=0.23). CONCLUSIONS: We have characterised the genetic diversity of cystinuria in a UK population including 23 pathogenic mutations not previously published. Patients with at least one missense mutation in SLC3A1 had significantly lower levels of lysine, arginine, and ornithine but not cystine than patients with all other combinations of mutations.