Benjamin Allin

Outcomes at one-year post anastomosis from a national cohort of infants with oesophageal atresia.

Background and Aims We aimed to provide a contemporaneous assessment of outcomes at one-year post oesophageal atresia/tracheoesophageal fistula (OA-TOF) repair, focussing particularly on post-operative complications. It is generally accepted that oesophageal stricture is the most common complication and causes significant morbidity. We also aimed to assess the efficacy of prophylactic anti-reflux medication (PARM) in reducing stricture formation. Method A prospective, multi-centre cohort study of all infants live-born with oesophageal atresia in the United Kingdom and Ireland in 2008/9 was performed, recording clinical management and outcomes at one year. The effect of PARM on stricture formation in infants with the type-c anomaly was assessed using logistic regression analysis. Results 151 infants were live-born with oesophageal atresia in the defined reporting period, 126 of whom had the type-c anomaly. One-year follow-up information was returned for 105 infants (70%); the mortality rate was 8.6% (95% CI 4.7–14.3%). Post-operative complications included anastomotic leak (5.4%), recurrent fistula (3.3%) and oesophageal stricture (39%). Seventy-six (60%) of those with type-c anomaly were alive at one-year with returned follow-up, 57(75%) of whom had received PARM. Of these, 24 (42%) developed a stricture, compared to 4 (21%) of those who had not received PARM (adjusted odds ratio 2.60, 95% CI 0.71–9.46, p = 0.147). Conclusions This study provides a benchmark for current outcomes and complication rates following OA-TOF repair, with oesophageal stricture causing significant morbidity. The use of PARM appeared ineffective in preventing strictures. This study creates enough doubt about the efficacy of PARM in preventing stricture formation to warrant further investigation of its use with a randomised controlled trial.

Laparoscopic assistance for primary transanal pull-through in Hirschsprung's disease: a systematic review and meta-analysis.

Objective To compare outcomes following totally transanal endorectal pull-through (TTERPT) versus pull-through with any form of laparoscopic assistance (LAPT) for infants with uncomplicated Hirschsprungs disease. Design Systematic review and meta-analysis. Setting Five hospitals with a paediatric surgical service. Participants 405 infants with uncomplicated Hirschsprungs disease. Interventions TTERPT versus LAPT. Primary and secondary outcome measures Primary outcomes: mortality, postoperative enterocolitis, faecal incontinence, constipation, unplanned laparotomy or stoma formation, and injury to abdominal viscera. Secondary outcomes Haemorrhage requiring transfusion of blood products, abscess formation, intestinal obstruction, intestinal ischaemia, enteric fistula formation, urinary incontinence or retention, impotency and duration of procedure. Results Five eligible studies comprising 405 patients were identified from 2107 studies. All studies were retrospective case series, with variability in outcome assessment quality and length of follow-up. Operative duration was 50.29 min shorter with TTERPT (95% CI 39.83 to 60.74, p<0.00001). There were no significant differences identified between TTERPT and LAPT for incidence of postoperative enterocolitis (OR=0.78, 95% CI 0.44 to 1.38, p=0.39), faecal incontinence (OR=0.44, 95% CI 0.09 to 2.20, p=0.32) or constipation (OR=0.84, 95% CI 0.32 to 2.17, p=0.71). Conclusions This meta-analysis did not find any evidence to suggest a higher rate of enterocolitis, incontinence or constipation following TTERPT compared with LAPT. Further long-term comparative studies and multicentre data pooling are needed to determine whether a purely transanal approach offers any advantages over a laparoscopically assisted approach to rectosigmoid Hirschsprungs disease. Trial registration number PROSPERO registry- CRD42013005698.

Challenges of improving the evidence base in smaller surgical specialties, as highlighted by a systematic review of gastroschisis management.

Objective To identify methods of improving the evidence base in smaller surgical specialties, using a systematic review of gastroschisis management as an example. Background Operative primary fascial closure (OPFC), and silo placement with staged reduction and delayed closure (SR) are the most commonly used methods of gastroschisis closure. Relative merits of each are unclear. Methods A systematic review and meta-analysis was performed comparing outcomes following OPFC and SR in infants with simple gastroschisis. Primary outcomes of interest were mortality, length of hospitalization and time to full enteral feeding. Results 751 unique articles were identified. Eight met the inclusion criteria. None were randomized controlled trials. 488 infants underwent OPFC and 316 underwent SR. Multiple studies were excluded because they included heterogeneous populations and mixed intervention groups. Length of stay was significantly longer in the SR group (mean difference 8.97 days, 95% CI 2.14–15.80 days), as was number of post-operative days to complete enteral feeding (mean difference 7.19 days, 95%CI 2.01–12.36 days). Mortality was not statistically significantly different, although the odds of death were raised in the SR group (OR 1.96, 95%CI 0.71–5.35). Conclusions Despite showing some benefit of OPFC over SR, our results are tempered by the low quality of the available studies, which were small and variably reported. Coordinating research through a National Paediatric Surgical Trials Unit could alleviate many of these problems. A similar national approach could be used in other smaller surgical specialties.

A UK wide cohort study describing management and outcomes for infants with surgical Necrotising Enterocolitis.

The Royal College of Surgeons have proposed using outcomes from necrotising enterocolitis (NEC) surgery for revalidation of neonatal surgeons. The aim of this study was therefore to calculate the number of infants in the UK/Ireland with surgical NEC and describe outcomes that could be used for national benchmarking and counselling of parents. A prospective nationwide cohort study of every infant requiring surgical intervention for NEC in the UK was conducted between 01/03/13 and 28/02/14. Primary outcome was mortality at 28-days. Secondary outcomes included discharge, post-operative complication, and TPN requirement. 236 infants were included, 43(18%) of whom died, and eight(3%) of whom were discharged prior to 28-days post decision to intervene surgically. Sixty infants who underwent laparotomy (27%) experienced a complication, and 67(35%) of those who were alive at 28 days were parenteral nutrition free. Following multi-variable modelling, presence of a non-cardiac congenital anomaly (aOR 5.17, 95% CI 1.9–14.1), abdominal wall erythema or discolouration at presentation (aOR 2.51, 95% CI 1.23–5.1), diagnosis of single intestinal perforation at laparotomy (aOR 3.1 95% CI 1.05–9.3), and necessity to perform a clip and drop procedure (aOR 30, 95% CI 3.9–237) were associated with increased 28-day mortality. These results can be used for national benchmarking and counselling of parents.

NETS 1HD : study protocol for development of a core outcome set for use in determining the overall success of Hirschsprung’s disease treatment

BackgroundUse of core outcome sets in research has been proposed as a method for countering the problems caused by heterogeneity of outcome measure reporting. Heterogeneity of outcome measure reporting occurs in Hirschsprung’s disease (HD) research and is limiting the development of a robust evidence base to support clinical practice.MethodsCandidate outcome measures have been identified through a systematic review. These outcome measures will form the starting point for a three-phase online Delphi process to be carried out in parallel by three panels of experts. Panel 1 is a neonatal panel; panel 2 is a non-neonatal panel; and panel 3 is a lay panel. In round 1, experts will be asked to score the previously identified outcome measures from 1 to 9 based on how important they think the measures are in determining the overall success of their/their child’s/their patient’s HD. In round 2, experts will be presented with the same list of outcome measures and graphical representations of how their panel scored that outcome in round 1. They will be asked to re-score the outcome measure, taking into account how important other members of their panel felt it to be. In round 3, experts will again be asked to re-score each outcome measure, but this time they will receive a graphical representation of the distribution of scores from all three panels, which they should take into account when re-scoring. Following round 3 of the Delphi process, 40 experts will be invited to attend a face-to-face consensus meeting. Participants will be invited in a purposive manner to obtain balance between the different panels. Results of the Delphi process will be discussed, and outcomes will be re-scored. Outcome measures where >70% of participants at the meeting scored it 7–9 and <15% scored it 1–3 will form the core outcome set.DiscussionDevelopment of a core outcome set will help to reduce heterogeneity of outcome measure reporting in HD. This will increase the quality of research taking place and ultimately improve care provided to infants with HD.

Variability of outcome reporting in Hirschsprung’s Disease and gastroschisis: a systematic review

Heterogeneity in outcome reporting limits identification of gold-standard treatments for Hirschsprung’s Disease(HD) and gastroschisis. This review aimed to identify which outcomes are currently investigated in HD and gastroschisis research so as to counter this heterogeneity through informing development of a core outcome set(COS). Two systematic reviews were conducted. Studies were eligible for inclusion if they compared surgical interventions for primary treatment of HD in review one, and gastroschisis in review two. Studies available only as abstracts were excluded from analysis of reporting transparency. Thirty-five HD studies were eligible for inclusion in the review, and 74 unique outcomes were investigated. The most commonly investigated was faecal incontinence (32 studies, 91%). Seven of the 28 assessed studies (25%) met all criteria for transparent outcome reporting. Thirty gastroschisis studies were eligible for inclusion in the review, and 62 unique outcomes were investigated. The most commonly investigated was length of stay (24 studies, 80%). None of the assessed studies met all criteria for transparent outcome reporting. This review demonstrates that heterogeneity in outcome reporting and a significant risk of reporting bias exist in HD and gastroschisis research. Development of a COS could counter these problems, and the outcome lists developed from this review could be used in that process.

Oesophageal atresia with no distal tracheoesophageal fistula: Management and outcomes from a population-based cohort

PURPOSE To describe the incidence and outcomes to one-year in infants born with oesophageal atresia (OA) with no distal tracheoesophageal fistula within a population cohort. METHODS A subgroup analysis of a prospective multicentre population cohort study was undertaken describing the outcomes of infants with OA and no tracheoesophageal fistula, (type A) and those with only an upper pouch fistula, (type B). MAIN RESULTS Twenty-one of 151 infants in the whole cohort were diagnosed with type A or B oesophageal atresia (14%). Fifteen were type A (71%) and six type B (29%). Infants with type B had a shorter gap length than those with type A: 2.5 vertebral bodies (2-3) vs. 5 (4-6) (p=0.008). All infants with type B OA underwent oesophageal anastomosis, 83% (n=5) as the primary procedure. All infants with type A, underwent staged management. Six (40%) had delayed primary anastomosis and eight required oesophageal replacement (53%). One infant died prior to reconstruction. The median time to delayed primary anastomosis in infants with type A or B OA was 82days (75-89days) (n=7). The median time to oesophageal replacement was 94days (89-147days) (n=8). Median length of stay for infants with type A or B OA from first operation to first discharge was 101days (31-123days). CONCLUSIONS Infants with type B OA had a shorter gap length and all were managed with oesophageal anastomosis. OA with no distal tracheoesophageal fistula is uncommon at a population level and frequently has a complex course. LEVEL OF EVIDENCE Rating: II.

NETS(1HD) study: development of a Hirschsprung's disease core outcome set

Objective The objective of this study was to develop a Hirschsprung’s disease (HD) core outcome set (COS). Methods Candidate outcomes were identified from a systematic review and stakeholder nomination. A three-phase Delphi process and consensus meeting were used to prioritise candidate outcomes based on scores assigned by stakeholder participants using a nine-point scale. In phases two and three, participants were shown graphical representations of their panel’s scores and all panels’ scores respectively for each outcome from the previous phase. After the third phase, outcomes prioritised by two or three panels were taken forward to the consensus meeting. The COS was formed from the 10 highest scoring outcomes meeting the threshold for inclusion (≥70% 7–9 and <15% 1–3). Results Eighty-nine stakeholders (82%) completed all three phases of the Delphi process. Seventy-four outcomes were assessed in phase one of the Delphi process, the following 10 of which met criteria for inclusion in the COS: (1) death with cause specified, (2) long-term faecal incontinence, (3) long-term voluntary bowel movements without need for enemas, or rectal or colonic irrigation, (4) long-term psychological stress for the individual with Hirschsprung’s disease, (5) long-term urinary incontinence, (6) objective score of quality of life, (7) objective score of bowel function, (8) unplanned reoperation, (9) >need for a permanent stoma, (10) enterocolitis. Conclusions This HD COS is formed of 10 outcomes deemed important by key stakeholders. Use of this COS in research will reduce outcome reporting heterogeneity and increase our ability to identify gold standard treatments for HD.

Enterolithotomy for the treatment of large bowel obstruction secondary to gallstones

We describe the case of an 81-year-old woman with large bowel-obstruction caused by an impacted gallstone. An 81-year-old, Caucasian, fully independent woman without significant comorbidities presented with absolute constipation, faecal vomiting and abdominal pain. Abdominal radiography revealed dilated small bowel, and a subsequent contrast CT demonstrated a 2.5 cm gallstone in the sigmoid colon. This is believed to have entered the transverse colon via a cholecyst-colonic fistula, and then migrated to a section of sigmoid colon affected by diverticular disease, where it became impacted. Two sigmoidoscopic removals were attempted but were unsuccessful as the gallstones size prevented removal with an endoscopic basket. A laparotomy was performed and the stone extracted via a sigmoid enterotomy. No covering stoma was formed, and following 48 h on intensive trauma unit and a short ward-based stay for rehabilitation, the patient was discharged home and is currently doing well.

Developing a set of consensus indicators to support maternity service quality improvement: using Core Outcome Set methodology including a Delphi process

To develop a core metric set to monitor the quality of maternity care.