Paediatric testicular torsion in low- and middle-income countries: an OxPLORE scoping study.

BACKGROUND: Literature on paediatric surgical conditions in low- and middle-income countries (LMICs) remains limited. As a common emergency, timely treatment of testicular torsion acts as a benchmark of adequate emergency service delivery in paediatric surgery. This scoping study aims to synthesise all existing literature on paediatric testicular torsion in LMICs. METHODS: A database search was conducted by the OxPLORE global paediatric surgery research group to identify studies containing the terms 'testicular torsion' or 'acute scrotum' originating from LMICs. A thematic analysis was applied to the results of the search and the quality of evidence was appraised for all included articles. RESULTS: This review included 17 studies with 1798 patients. All studies originated from middle-income countries and the majority (76%) had sample sizes smaller than 100 patients. All studies were appraised as providing less than adequate evidence. Included studies identified long delays to treatment and highlighted ongoing debates on the value of scoring systems and Doppler ultrasonography in diagnosing torsion. Major heterogeneity in surgical approaches to treatment of testicular torsion in children was also observed. CONCLUSIONS: Literature on paediatric testicular torsion in LMICs is scarce and heterogeneous. Prospective, multi-centre research on the management of this common paediatric surgical emergency is urgently required.

Transitional Care in Patients With Hirschsprung Disease: Those Left Behind.

BACKGROUND: The long-term effects of Hirschsprung disease are clinically variable. An improved understanding of challenges patients may face as adults can help inform transitional care management. OBJECTIVE: To explore the outcomes and transitional care experiences in adult patients with Hirschsprung. DESIGN: Cohort study. SETTING: Single center. PATIENTS: All patients treated for Hirschsprung between 1977 and 2001 (aged older than 18 years at the time of survey distribution in July 2018-2019). Eligible patients were sent validated multidomain surveys and qualitative questions regarding their transitional care. MAIN OUTCOME MEASURES: Status of transitional care, bowel function, and quality-of-life assessment. Qualitative analysis of transitional care experience. RESULTS: Of 139 patients, 20 had received transition care (10 had at least 1 visit but had been discharged and 10 were receiving ongoing follow-up). These patients had inferior bowel function and quality-of-life scores at follow-up. Twenty-three patients (17%) had issues with soiling at the time of discharge, and 7 patients received transitional care. Of these 23 patients, 9 (39%) had a normal Bowel Function Score (17 or more), 5 (22%) had a poor score (less than 12), and 1 had since had a stoma formation. Eighteen patients (13%) had active moderate-severe issues related to bowel function, only 5 had been transitioned, and just 2 remained under ongoing care. Importantly, when these patients were discharged from our pediatric center, at a median age of 14 (interquartile range, 12-16) years, 10 of 17 patients had no perceptible bowel issues, suggesting a worsening of function after discharge. LIMITATIONS: The retrospective design and reliance on clinical notes to gather information on discharge status as well as patient recall of events. CONCLUSIONS: There remains a small but significant proportion of Hirschsprung patients for whom bowel function either remains or becomes a major burden. These results support a need to better stratify patients requiring transitional care and ensure a clear route to care if their status changes after discharge. See Video Abstract . ATENCIN DE TRANSICIN EN PACIENTES CON ENFERMEDAD DE HIRSCHSPRUNG, LOS QUE SE QUEDAN ATRS: ANTECEDENTES:Los efectos a largo plazo de la enfermedad de Hirschsprung son clínicamente variables. Una mejor comprensión de los desafíos que los pacientes pueden enfrentar cuando sean adultos puede ayudar a informar la gestión de la atención de transición.OBJETIVO:Explorar los resultados y las experiencias de atención de transición en pacientes adultos con Hirschsprung.DISEÑO:Estudio de cohorte.AJUSTE:Unico centro.PACIENTES:Todos los pacientes tratados por Hirschsprung 1977-2001 (edad >18 años en el momento de la encuesta, Julio de 2018-2019). A los pacientes elegibles se les enviaron encuestas multidominio validadas, así como preguntas cualitativas sobre su atención de transición.PRINCIPALES MEDIDAS DE RESULTADOS:Estado de la atención de transición, función intestinal y evaluación de la calidad de vida. Análisis cualitativo de la experiencia de cuidados transicionales.RESULTADOS:De 139 pacientes, 20 habían recibido atención de transición (10 tuvieron al menos una visita pero habían sido dados de alta y 10 estaban recibiendo seguimiento continuo). Estos pacientes tenían puntuaciones inferiores de función intestinal y calidad de vida en el seguimiento. Veintitrés (17%) pacientes tuvieron problemas para ensuciarse en el momento del alta y 7 recibieron atención de transición. De estos, 9/23 (39%) tenían una puntuación de función intestinal normal (≥17), 5/23 (22%) tenían una puntuación baja (<12) y un paciente había tenido desde entonces una formación de estoma. Dieciocho (13%) pacientes tenían problemas activos de moderados a graves relacionados con la función intestinal, solo cinco habían realizado la transición y solo 2 permanecían bajo atención continua. Es importante destacar que cuando estos pacientes fueron dados de alta de nuestro centro pediátrico, a una edad promedio de 14 [RIQ 12-16] años, 10/17 no tenían problemas intestinales perceptibles, lo que sugiere un empeoramiento de la función después del alta.LIMITACIONES:El diseño retrospectivo y la dependencia de notas clínicas para recopilar información sobre el estado del alta, así como el recuerdo de los eventos por parte del paciente.CONCLUSIÓN:Sigue existiendo una proporción pequeña pero significativa de pacientes con Hirschsprung para quienes la función intestinal permanece o se convierte en una carga importante. Estos resultados respaldan la necesidad de estratificar mejor a los pacientes que requieren atención de transición y garantizar una ruta clara hacia la atención si su estado cambia después del alta. ( Traducción-Dr. Yesenia Rojas-Khalil ).

Vertical and horizontal gene transfer shaped plant colonization and biomass degradation in the fungal genus Armillaria.

The fungal genus Armillaria contains necrotrophic pathogens and some of the largest terrestrial organisms that cause tremendous losses in diverse ecosystems, yet how they evolved pathogenicity in a clade of dominantly non-pathogenic wood degraders remains elusive. Here we show that Armillaria species, in addition to gene duplications and de novo gene origins, acquired at least 1,025 genes via 124 horizontal gene transfer events, primarily from Ascomycota. Horizontal gene transfer might have affected plant biomass degrading and virulence abilities of Armillaria, and provides an explanation for their unusual, soft rot-like wood decay strategy. Combined multi-species expression data revealed extensive regulation of horizontally acquired and wood-decay related genes, putative virulence factors and two novel conserved pathogenicity-induced small secreted proteins, which induced necrosis in planta. Overall, this study details how evolution knitted together horizontally and vertically inherited genes in complex adaptive traits of plant biomass degradation and pathogenicity in important fungal pathogens.

The impact of the COVID-19 pandemic on paediatric surgical volumes in Africa: A retrospective observational study.

BACKGROUND: The aim of this study is to investigate the impact that COVID-19 had on the pattern and trend of surgical volumes, urgency and reason for surgery during the first 6 months of the pandemic in sub-Saharan Africa. METHODS: This retrospective facility-based study involved collection of paediatric operation data from operating theatre records across 5 hospitals from 3 countries: Zimbabwe, Zambia and Nigeria over the first half of 2019 and 2020 for comparison. Data concerning diagnosis, procedure, anaesthesia, grade, speciality, NCEPOD classification and indication was collected. The respective dates of enactment of cancellation policies in each country were used to compare changes in weekly median surgical case volume before cancellation using the Wilcoxon Sign-Rank Test. RESULTS: A total of 1821 procedures were recorded over the study period. Surgical volumes experienced a precipitous drop overall from a median of 100 cases/week to 50 cases/week coinciding with cancellation of surgical electives. Median accumulated weekly procedures before COVID-related cancellation were significantly different from those after cancellation (p = 0.027). Emergency surgery fell by 23.3% while electives fell by 78,9% (P = 0.042). The most common primary indication for surgery was injury which experienced a 30.5% drop in number of procedures, only exceeded by congenital surgery which dropped 34.7%. CONCLUSIONS: The effects of surgical cancellations during the covid-19 pandemic are particularly devastating in African countries where the unmet need and surgical caseload are high. Continued cancellations that have since occurred will cause similar drops in surgical case volume that these health systems may not have the resilience to recover from. LEVEL OF EVIDENCE: Level II.

Birth prevalence of anorectal malformations in England and 5-year survival: a national birth cohort study.

OBJECTIVE: To determine the birth prevalence, maternal risk factors and 5-year survival for isolated and complex anorectal malformations. DESIGN: National birth cohort using hospital admission data and death records. SETTING: All National Health Service England hospitals. PATIENTS: Live-born singletons delivered from 2002 through 2018, with evidence in the first year of life of a diagnosis of an anorectal malformation and repair during a hospital admission, or anorectal malformation recorded on the death certificate. Cases were further classified as isolated or complex depending on the presence of additional anomalies. MAIN OUTCOME MEASURES: Birth prevalence of anorectal malformations per 10 000 live births, risk ratios for isolated and complex anorectal malformation by maternal, infant and birth characteristics, and 5-year survival. RESULTS: We identified 3325 infants with anorectal malformations among 9 474 147 live-born singletons; 61.7% (n=2050) of cases were complex. Birth prevalence was 3.5 per 10 000 live births (95% CI 3.4 to 3.6). Complex anorectal malformations were associated with maternal age extremes after accounting for other sociodemographic factors. Compared with maternal ages 25-34 years, the risk of complex anorectal malformations was 31% higher for ≥35 years (95% CI 17 to 48) and 13% higher for ≤24 years (95% CI 0 to 27). Among 2376 anorectal malformation cases (n=1450 complex) born from 2002 through 2014, 5-year survival was lower for complex (86.9%; 95% CI 85.1% to 88.5%) than isolated anorectal malformations (98.2%; 95% CI 97.1% to 98.9%). Preterm infants with complex anorectal malformations had the lowest survival (73.4%; 95% CI 68.1% to 78.0%). CONCLUSIONS: Differences in maternal risk factors for isolated and complex anorectal malformations may reflect different underlying mechanisms for occurrence. Five-year survival is high but lowest for preterm children with complex anorectal malformations.

An OxPLORE Initiative Evaluating Children's Surgery Resources Worldwide: A Cross-sectional Implementation of the OReCS Document.

BACKGROUND: The Global Initiative for Children's Surgery (GICS) group produced the Optimal Resources for Children's Surgery (OReCS) document in 2019, listing standards of children's surgical care by level of healthcare facilities within low resource settings. We have previously created and piloted an audit tool based on the OReCS criteria in a high-income setting. In this study, we aimed to validate its use in identifying gaps in children's surgery provision worldwide. METHODS: Our OReCS audit tool was implemented in 10 hospitals providing children's surgery across eight countries. Collaborators were recruited via the Oxford Paediatrics Linking Our Research with Electives (OxPLORE) international network of medical students and trainees. The audit tool measured a hospital's current capacity for children's surgery. Data were analysed firstly to express the percentage of 'essential' criteria met for each specialty. Secondly, the 'OxPLORE method' was used to allocate each hospital specialty a level based on procedures performed and resources available. A User Evaluation Tool (UET) was developed to obtain feedback on the ease of use of the tool. RESULTS: The percentage of essential criteria met within each category varied widely between hospitals. The level given to hospitals for subspecialties based on OReCS criteria often did not reflect their self-defined level. The UET indicated the audit tool was practicable across multiple settings. CONCLUSIONS: We recommend the use of the OReCS criteria to identify areas for local hospital improvement and inform national children's surgical plans. We have made informed suggestions to increase usability of the OReCS audit tool.

Association between timing of re-introduction of enteral feeding and short-term outcomes following laparotomy for necrotising enterocolitis.

PURPOSE: To investigate the relationship between timing of re-introduction of feeds following surgery for Necrotising Enterocolitis (NEC) and important early outcomes. METHODS: Secondary analysis of prospectively collected data from paediatric surgical units in UK/Ireland of infants who underwent laparotomy for NEC between 01/03/2013 and 28/02/2014. Multivariable logistic regression analysis was used to compare the relationship of early (≤ 7 days) and later (8-27 days) re-introduction of feeding after surgery on death or need for PN at 28 days, correcting for known cofounders. RESULTS: 41/143 infants (29%) received early and 102/143 infants (71%) had delayed reintroduction of feeding. Infants in the early feeding group had a higher gestational age at birth, higher proportion of growth restriction, lower inotrope requirement, and weremore likely to have undergone primary anastomosis. Following adjustment there was no statistically significant difference detected in the rate of death or need for PN at 28 days, adjusted OR 0.4 (95% CI 0.2-1.1), noting the limited statistical power of this comparison. CONCLUSIONS: There is no evidence from this study to support a minimum period of 7 days nil by mouth post laparotomy for infants with NEC. Early feed reintroduction following laparotomy for NEC is safe in appropriate cases. LEVEL OF EVIDENCE: Level II - Treatment Study Group; Prospective comparative study.

Multicenter Study of 342 Anorectal Malformation Patients: Age, Gender, Krickenbeck Subtypes, and Associated Anomalies.

INTRODUCTION: Published studies based on Krickenbeck classification of anorectal malformations (ARMs) are still insufficient to assess the global as well as regional relative incidence of different ARM subtypes, gender distribution, and associated anomalies. The primary purpose of this study was to provide an estimate of those in Global Initiative for Children's Surgery (GICS) research group. MATERIALS AND METHODS: We collected ARM data prospectively for 1 year from four institutes of different geographic locations. A total of 342 patients were included in this study (195, 126, 11, and 10 from Bangladesh, Iran, Papua New Guinea, and Oxford, United Kingdom, respectively). RESULTS: Overall male to female ratio was 1:1. The most frequent ARM subtype was perineal fistula (23.7% = 81/342). About 48.5% (166/342) patients had at least one associated anomaly. Cardiac and genitourinary systems were the most commonly affected systems, 31.6% (108/342) and 18.4% (63/342), respectively. These organ-systems were followed by anomalies of vertebral/spinal (9.9% = 34/342), musculoskeletal (4.4% = 15/342), and gastrointestinal/abdominal (3.2% = 11/342) systems. Rectovesical fistula had the highest percentage (96.4% = 27/28) of associated anomalies. About 18.1% (62/342) patients had multiple anomalies. ARMs (both isolated and with associated anomalies) occurred equally in males and females. Comparison between patients from Bangladesh and Iran showed differences in relative incidence in ARM subtypes. In addition, Iranian patient group had higher percentage of associated anomalies compared with Bangladeshi (73 vs. 35.4%). CONCLUSION: Our study provides important insights about ARM subtypes, gender distribution and associated anomalies based on Krickenbeck classification especially from Bangladesh and Iran.

Biliary atresia: a scientometric analysis of the global research architecture and scientific developments.

Biliary atresia (BA) is a rare cholangiopathy of largely unknown etiology and unpredictable outcome. There has been an increasing number of BA-related publications, which may challenge researchers to determine their actual scientific value. This study aimed to evaluate the global research activity and developments relating to BA using a combination of scientometric methodologies and visualization tools. A comprehensive search strategy for the Web of Science™ database was designed to obtain bibliographic data on scientific BA publications for the timespan 1900-2018. Research output of countries, institutions, individual authors and collaborative networks was analyzed. Semi-qualitative research measures including citation rate and h-index were assessed. Choropleth mapping and network diagrams were used to visualize results. In total, 4,459 publications on BA were identified (88.5% in English), originating from 63 countries. The largest number was published by the USA (n = 991; 22.2%), Japan (n = 667; 15.0%) and the UK (n = 294; 6.6%). The USA combined the highest number of cooperation articles (n = 140). The most productive collaborative network was established between the USA and Canada (n = 17). Scientific papers from the UK received the highest average citation rate (16.7), whereas the USA had the highest country-specific h-index (59). Eighty-eight (2.0%) items were published under the auspices of multicenter consortiums and registries. The most productive institutions and authors were based in the USA, the UK, Japan, France, Canada and Taiwan. BA-related research has constantly been progressing, becoming more multidisciplinary but with main research endeavors concentrated in a few high-income countries. Studies into pathogenesis of BA remain uncommon, but are sorely needed to foster true scientific progress with this rare disease. Hence, international collaborative and translational research should be strengthened to allow further evolution in this field.

Choledochal malformations: global research, scientific advances and key controversies.

Choledochal malformations (CMs) represent a spectrum of relatively rare and complex congenital anomalies, characterized by abnormal dilatation of the biliary tract in the absence of any acute obstruction. Today, almost 20% of CMs can be detected in-utero using maternal ultrasonography. Formal scientometric analysis was used to identify where modern CM research is taking place and perhaps where our attention should be directed in the future. Thus, this article offers a comprehensive review of recent scientific advances relating to CMs including the current understanding of etiology and classification, whilst also discussing key controversies such as risk of malignant transformation and the role of newer modalities of surgical treatment. Although laparoscopic excision of CMs and biliary reconstruction is nowadays feasible and safe, care should be taken before dispensing with standard open techniques, which have minimal complication rates and proven long-term benefit.

Correction to: Choledochal malformations: global research, scientific advances and key controversies.

The last sentence in the first paragraph under subheading "Minimally invasive treatment" was incorrect in the original publication.

Learning from lawsuits: Ten-years of NHS litigation authority claims against 11 surgical specialities in England.

INTRODUCTION: Medico-legal claims are a drain on NHS resources and promote defencive practice. The litigious burden of surgery in England has not been previously described. This paper describes trends over ten years of claims made against the NHS across 11 surgical specialities. MATERIALS AND METHODS: Data were requested for all claims received by the NHS Litigation Authority (NHSLA) from 2004 to 2014. Surgical specialities included cardiothoracic, general, neurosurgery, obstetric, oral and maxillofacial (OMFS), orthopaedic, otorhinolaryngology, paediatric, plastic, urology and vascular surgery. A literature review of peer-reviewed publications was performed with search terms 'NHSLA' and 'Surgery'. RESULTS: The NHS paid out approximately £1.5 billion across 11 surgical specialities from 2004 to 2014. Orthopaedic, obstetric and general surgery received the largest number of claims per year, and paediatric surgery the least. The mean time from registration of claim with the NHSLA to settlement was 25.5 months (range 17.8 months-35 months). Neurosurgery was responsible for the highest average amount paid per claim, and OMFS the lowest. Failure/delay in treatment and/or diagnosis and failure to warn/adequately consent were the three leading types of claim. 806 never events were successfully claimed for during the ten-year period. DISCUSSION AND CONCLUSION: Sharing information and good practice should be a priority for surgical professionals. Lessons learnt from medico-legal claims are transferrable in strategic planning. This pan-speciality report has demonstrated considerable burden on the NHS and should promote improvement in practice on an individual level in addition to providing systems based recommendations to NHS and international organisations.

A native promoter and inclusion of an intron is necessary for efficient expression of GFP or mRFP in Armillaria mellea.

Armillaria mellea is a significant pathogen that causes Armillaria root disease on numerous hosts in forests, gardens and agricultural environments worldwide. Using a yeast-adapted pCAMBIA0380 Agrobacterium vector, we have constructed a series of vectors for transformation of A. mellea, assembled using yeast-based recombination methods. These have been designed to allow easy exchange of promoters and inclusion of introns. The vectors were first tested by transformation into basidiomycete Clitopilus passeckerianus to ascertain vector functionality then used to transform A. mellea. We show that heterologous promoters from the basidiomycetes Agaricus bisporus and Phanerochaete chrysosporium that were used successfully to control the hygromycin resistance cassette were not able to support expression of mRFP or GFP in A. mellea. The endogenous A. mellea gpd promoter delivered efficient expression, and we show that inclusion of an intron was also required for transgene expression. GFP and mRFP expression was stable in mycelia and fluorescence was visible in transgenic fruiting bodies and GFP was detectable in planta. Use of these vectors has been successful in giving expression of the fluorescent proteins GFP and mRFP in A. mellea, providing an additional molecular tool for this pathogen.

Contemporary pediatric surgical training in the UK.

INTRODUCTION: Pediatric surgical trainees worldwide face pressures from expansion of programs and training positions, subspecialization, regionalization, restrictions of working hours, and rigid training criteria. The era of apprenticeship training has long gone, and surgical education needs to be responsive and adapt to newer challenges. The aim of this study was to examine the teaching provision component of pediatric surgical training in the UK. METHOD: A national teaching survey was sent to UK pediatric surgery trainees in 2010 and compared to results of a repeat survey in 2015. Analysis was carried out to compare type of teaching, trends in teaching delivery, quality, and attendance over time. RESULTS: Regional variability was noted in teaching programs. Both provision of educational activities and ability to attend teaching improved between 2010 and 2015. Despite this, overall trainee satisfaction remained low, with 50% and 52% of respondents describing their teaching as "good" or "excellent" in 2010 and 2015, respectively (P=0.84). Seventy-five percent of centers provided simulation training, and 25% of respondents had regional teaching provided. Survey response rate was comparable between 2010 and 2015. CONCLUSION: Variability in national educational provision was observed. We suggest regular national audit of educational activity and responsive adaption to external pressures on training if competent surgeons are to be the product of contemporary pediatric surgery training programs.

Surgical Success in Chronic Pancreatitis: Sequential Endoscopic Retrograde Cholangiopancreatography and Surgical Longitudinal Pancreatojejunostomy (Puestow Procedure).

Introduction Chronic pancreatitis (CP) can be a cause of recurrent, severe, disabling abdominal pain in children. Surgery has been suggested as a useful therapy, although experience is limited and the results unpredictable. We reviewed our experience of a two-stage protocol-preliminary endoscopic retrograde cholangiopancreatography (ERCP) and duct stenting, and if symptoms resolved, definitive surgical decompression by longitudinal pancreatojejunostomy (LPJ) (Puestow operation). Patients and Methods This is a single-center, retrospective review of children with established CP who underwent an LPJ between February 2002 and September 2012. A questionnaire was completed (incorporating visual analog scale pain and lifestyle scores) to assess functional outcome. Data are expressed as median (range). Results In this study, eight (M:F ratio of 4:4) children underwent an LPJ and one female child had a more limited pancreatojejunostomy anastomosis following preliminary ERCP and stent placement where possible. Diagnoses included hereditary pancreatitis (n = 3), idiopathic or structural pancreatitis (n = 5), and duct stricture following radiotherapy (n = 1). Median duct diameter presurgery was 5 (4-11) mm. Endoscopic placement of a Zimmon pancreatic stent was possible in six with relief of symptoms in all. Median age at definitive surgery was 11 (range, 7-17) years with a median postoperative stay of 9 (range, 7-12) days and a follow-up of 6 (range, 0.5-12) years. All children reported markedly reduced episodes of pain postprocedure. One developed diabetes mellitus, while three had exocrine deficiency (fecal elastase < 200 µg/g) requiring enzyme supplementation. The child with limited LPJ had symptomatic recurrence and required restenting and further surgery to widen the anastomosis to become pain free. Conclusion ERCP and stenting provide a therapeutic trial to assess possible benefit of a definitive duct drainage procedure. LPJ-the modified Puestow operation was safe and complication-free with good medium-term relief of symptoms. We were not able to identify a consistent etiology-associated outcome.