Research prioritization in paediatric orthopaedics and the impact on funding.

In 2017, the British Society for Children's Orthopaedic Surgery engaged the profession and all relevant stakeholders in two formal research prioritization processes. In this editorial, we describe the impact of this prioritization on funding, and how research in children's orthopaedics, which was until very recently a largely unfunded and under-investigated area, is now flourishing. Establishing research priorities was a crucial step in this process.

Protecting my injured child: a qualitative study of parents' experience of caring for a child with a displaced distal radius fracture.

BACKGROUND: Childhood fractures can have a significant impact on the daily lives of families affecting children's normal activities and parent's work. Wrist fractures are the most common childhood fracture. The more serious wrist fractures, that can look visibly bent, are often treated with surgery to realign the bones; but this may not be necessary as bent bones straighten in growing children. The children's radius acute fracture fixation trial (CRAFFT) is a multicentre randomised trial of surgery versus a cast without surgery for displaced wrist fractures. Little is known about how families experience these wrist fractures and how they manage treatment uncertainty. This study aimed to understand families' experience of this injury and what it is like to be asked to include their child in a clinical trial. METHODS: Nineteen families (13 mothers, 7 fathers, 2 children) from across the UK participated in telephone interviews. Interviews were audio recorded, transcribed and analysed using reflexive thematic analysis. RESULTS: Our findings highlight parents' desire to be a good parent through the overarching theme "protecting my injured child". To protect their child after injury, parents endeavoured to make the right decisions about treatment and provide comfort to their child but they experienced ongoing worry about their child's recovery. Our findings show that parents felt responsible for the decision about their child's treatment and their child's recovery. They also reveal the extent to which parents worried about the look of their child's wrist and their need for reassurance that the wrist was healing. CONCLUSION: Our findings show that protecting their child after injury can be challenging for parents who need support to make decisions about treatment and confidently facilitate their child's recovery. They also highlight the importance of providing information about treatments, acknowledging parents' concerns and their desire to do the right thing for their child, reassuring parents that their child's wrist will heal and ensuring parents understand what to expect as their child recovers.

Pay for performance and hip fracture outcomes: an interrupted time series and difference-in-differences analysis in England and Scotland.

AIMS: Hip fractures are associated with high morbidity, mortality, and costs. One strategy for improving outcomes is to incentivize hospitals to provide better quality of care. We aimed to determine whether a pay-for-performance initiative affected hip fracture outcomes in England by using Scotland, which did not participate in the scheme, as a control. MATERIALS AND METHODS: We undertook an interrupted time series study with data from all patients aged more than 60 years with a hip fracture in England (2000 to 2018) using the Hospital Episode Statistics Admitted Patient Care (HES APC) data set linked to national death registrations. Difference-in-differences (DID) analysis incorporating equivalent data from the Scottish Morbidity Record was used to control for secular trends. The outcomes were 30-day and 365-day mortality, 30-day re-admission, time to operation, and acute length of stay. RESULTS: There were 1 037 860 patients with a hip fracture in England and 116 594 in Scotland. Both 30-day (DID -1.7%; 95% confidence interval (CI) -2.0 to -1.2) and 365-day (-1.9%; 95% CI -2.5 to -1.3) mortality fell in England post-intervention when compared with outcomes in Scotland. There were 7600 fewer deaths between 2010 and 2016 that could be attributed to interventions driven by pay-for-performance. A pre-existing annual trend towards increased 30-day re-admissions in England was halted post-intervention. Significant reductions were observed in the time to operation and length of stay. CONCLUSION: This study provides evidence that a pay-for-performance programme improved the outcomes after a hip fracture in England. Cite this article: Bone Joint J 2019;101-B:1015-1023.

Dopamine receptor D4 (DRD4) polymorphisms with reduced functional potency intensify atrophy in syndrome-specific sites of frontotemporal dementia.

OBJECTIVE: We aimed to understand the impact of dopamine receptor D4 (DRD4) polymorphisms on neurodegeneration in patients with dementia. We hypothesized that DRD4dampened-variants with reduced functional potency would be associated with greater atrophy in regions with higher receptor density. Given that DRD4 is concentrated in anterior regions of the limbic and cortical forebrain we anticipated genotype effects in patients with a more rostral pattern of neurodegeneration. METHODS: 337 subjects, including healthy controls, patients with Alzheimer's disease (AD) and frontotemporal dementia (FTD) underwent genotyping, structural MRI, and cognitive/behavioral testing. We conducted whole-brain voxel-based morphometry to examine the relationship between DRD4 genotypes and brain atrophy patterns within and across groups. General linear modeling was used to evaluate relationships between genotype and cognitive/behavioral measures. RESULTS: DRD4 dampened-variants predicted gray matter atrophy in disease-specific regions of FTD in anterior cingulate, ventromedial prefrontal, orbitofrontal and insular cortices on the right greater than the left. Genotype predicted greater apathy and repetitive motor disturbance in patients with FTD. These results covaried with frontoinsular cortical atrophy. Peak atrophy patterned along regions of neuroanatomic vulnerability in FTD-spectrum disorders. In AD subjects and controls, genotype did not impact gray matter intensity. CONCLUSIONS: We conclude that DRD4 polymorphisms with reduced functional potency exacerbate neuronal injury in sites of higher receptor density, which intersect with syndrome-specific regions undergoing neurodegeneration in FTD.

What is the incidence of late detection of developmental dysplasia of the hip in England?: a 26-year national study of children diagnosed after the age of one.

AIMS: The aim of this study was to establish the incidence of developmental dysplasia of the hip (DDH) diagnosed after one-year of age in England, stratified by age, gender, year, and region of diagnosis. PATIENTS AND METHODS: A descriptive observational study was performed by linking primary and secondary care information from two independent national databases of routinely collected data: the United Kingdom Clinical Practice Research Datalink and Hospital Episode Statistics. The study examined all children from 1 January 1990 to 1 January 2016 who had a new first diagnostic code for DDH aged between one and eight years old. RESULTS: The incidence of late-diagnosed DDH was 1.28 per 1000 live births. Within the study population, 754 children were identified with a diagnosis of DDH after one-year of age. Of all late diagnoses, 536 (71.1%) were detected between one to two years of age. There were 608 female patients (80.6%) and 146 male patients (19.4%), giving a female-to-male ratio of 4.2:1. Distribution was evenly spread throughout England. CONCLUSION: The incidence of late-diagnosed DDH has not been reduced from that reported 35 years ago, prior to the introduction of the national selective screening programme for DDH. Cite this article: Bone Joint J 2019;101-B:281-287.

Temporal trends and survivorship of total hip arthroplasty in very young patients: a study using the National Joint Registry data set.

AIMS: The aim of this study was to describe temporal trends and survivorship of total hip arthroplasty (THA) in very young patients, aged ≤ 20 years. PATIENTS AND METHODS: A descriptive observational study was undertaken using data from the National Joint Registry (NJR) for England, Wales, Northern Ireland and the Isle of Man between April 2003 and March 2017. All patients aged ≤ 20 years at the time of THA were included and the primary outcome was revision surgery. Descriptive statistics were used to summarize the data and Kaplan-Meier estimates calculated for the cumulative implant survival. RESULTS: A total of 769 THAs were performed in 703 patients. The median follow-up was 5.1 years (interquartile range (IQR) 2.6 to 7.8). Eight patients died and 35 THAs were revised. The use of metal-on-metal (MoM) bearings and resurfacing procedures declined after 2008. The most frequently recorded indications for revision were loosening (20%) and infection (20%), although the absolute risk of these events occurring was low (0.9%). Factors associated with lower implant survival were MoM and metal-on-polyethylene (MoP) bearings and resurfacing arthroplasty ( vs ceramic-on-polyethylene (CoP) and ceramic-on-ceramic (CoC) bearings, p = 0.002), and operations performed by surgeons who undertook few THAs in this age group as recorded in the NJR ( vs those with five or more recorded operations, p = 0.030). Kaplan-Meier estimates showed 96% (95% confidence interval (CI) 94% to 98%) survivorship of implants at five years. CONCLUSION: Within the NJR, the overall survival for very young patients undergoing THA exceeded 96% during the first five postoperative years. In the absence of studies that can better account for differences in the characteristics of the patients, surgeons should consider the association between early revision and the type of implant, the number of THAs performed in these patients, and the bearing surface when performing THA in very young patients. Cite this article: Bone Joint J 2018;100-B:1320-9.

A consensus exercise identifying priorities for research into clinical effectiveness among children's orthopaedic surgeons in the United Kingdom.

Aims: High-quality clinical research in children's orthopaedic surgery has lagged behind other surgical subspecialties. This study used a consensus-based approach to identify research priorities for clinical trials in children's orthopaedics. Methods: A modified Delphi technique was used, which involved an initial scoping survey, a two-round Delphi process and an expert panel formed of members of the British Society of Children's Orthopaedic Surgery. The survey was conducted amongst orthopaedic surgeons treating children in the United Kingdom and Ireland. Results: A total of 86 clinicians contributed to both rounds of the Delphi process, scoring priorities from one (low priority) to five (high priority). Elective topics were ranked higher than those relating to trauma, with the top ten elective research questions scoring higher than the top question for trauma. Ten elective, and five trauma research priorities were identified, with the three highest ranked questions relating to the treatment of slipped capital femoral epiphysis (mean score 4.6/ 5), Perthes' disease (4.5) and bone infection (4.5). Conclusion: This consensus-based research agenda will guide surgeons, academics and funders to improve the evidence in children's orthopaedic surgery and encourage the development of multicentre clinical trials. Cite this article: Bone Joint J 2018;100-B:680-4.

A case control study to determine the association between Perthes' disease and the recalled use of tobacco during pregnancy, and biological markers of current tobacco smoke exposure.

AIMS: It is well established that there is a strong association between Perthes' disease and worsening socioeconomic deprivation. It has been suggested that the primary determinant driving this association is exposure to tobacco smoke. This study aimed to examine this hypothesis. PATIENTS AND METHODS: A hospital case-control study (n = 149/146) examined the association between tobacco smoke exposure and Perthes' disease, adjusting for area-level socioeconomic deprivation. Tobacco smoke exposure was assessed by parental questionnaire of smoking habits during pregnancy, and by quantitative assay of current exposure using the urinary cotinine-creatinine ratio, which is a widely used and validated measure of tobacco smoke exposure. RESULTS: The odds of Perthes' disease significantly increased with reported in utero exposure after adjustment for socioeconomic deprivation (maternal smoking odds ratio (OR) 2.06, 95% confidence interval (CI) 1.17 to 3.63; paternal smoking OR 2.09, 95% CI 1.26 to 3.46). The cotinine-creatinine ratio was significantly greater in cases, OR 1.63 (95% CI 1.09 to 2.43), suggesting a greater 'dose' of current tobacco exposure. CONCLUSION: An association exists between tobacco smoke exposure and Perthes' disease but we remain unable to disentangle the association with socioeconomic deprivation. Cite this article: Bone Joint J 2017;99-B:1102-8.

Regionalisation of trauma care in England.

AIMS: We aimed to determine whether there is evidence of improved patient outcomes in Major Trauma Centres following the regionalisation of trauma care in England. PATIENTS AND METHODS: An observational study was undertaken using the Trauma Audit and Research Network (TARN), Hospital Episode Statistics (HES) and national death registrations. The outcome measures were indicators of the quality of trauma care, such as treatment by a senior doctor and clinical outcomes, such as mortality in hospital. RESULTS AND CONCLUSION: A total of 20 181 major trauma cases were reported to TARN during the study period, which was 270 days before and after each hospital became a Major Trauma Centre. Following regionalisation of trauma services, all indicators of the quality of care improved, fewer patients required secondary transfer between hospitals and a greater proportion were discharged with a Glasgow Outcome Score of "good recovery". In this early post-implementation analysis, there were a number of apparent process improvements (e.g. time to CT) but no differences in either crude or adjusted mortality. The overall number of deaths following trauma in England did not change following the national reconfiguration of trauma services. Evidence from other countries that have regionalised trauma services suggests that further benefits may become apparent after a period of maturing of the trauma system. Cite this article: Bone Joint J 2016;98-B:1253-61.

The lognormal age of onset distribution in Perthes' disease: an analysis from a large well-defined cohort.

AIMS: To explore the of age of onset distribution for Perthes' disease of the hip, with particular reference to gender, laterality and conformity to the lognormal distribution. PATIENTS AND METHODS: A total of 1082 patients were identified from the Liverpool Perthes' Disease Register between 1976 and 2010, of which 992 had the date of diagnosis recorded. In total, 682 patients came from the geographical area exclusively served by Alder Hey Hospital, of which 673 had a date of diagnosis. Age of onset curves were analysed, with respect to the predefined subgroups. RESULTS: The age of onset demonstrated a positive skew with a median of 5.8 years (interquartile range 4.6 to 7.5). Disease onset was a mean five months earlier in girls (p = 0.01) and one year earlier in those who went on to develop bilateral disease (p < 0.001). There was no difference in the age of onset between geographical districts with differing incidence rates. The entire dataset (n = 992) conformed to a lognormal distribution graphically and with the chi-squared test of normality (p = 0.10), but not using the Shapiro-Wilk test (p = 0.01). The distribution for the predefined geographical subgroup (n = 673) conformed well to a lognormal distribution (chi-squared p = 0.16, Shapiro-Wilk p = 0.08). Given the observed lognormal distribution it was assumed that Perthes' disease followed on incubation period consistent with a point-source disease exposure. The incubation period was further examined using Hirayama's method, which suggested that the disease exposure may act in the prenatal period. CONCLUSION: The age of onset in Perthes' disease conforms to a lognormal distribution, which allows comparisons with infectious disease epidemiology. Earlier onset in girls and those who develop bilateral disease may offer clues to understanding the aetiological determinants of the disease. The analysis suggests that an antenatal aetiological determinant may be responsible for disease. TAKE HOME MESSAGE: Perthes' disease age of onset conforms to a lognormal model, which is most typical of infectious diseases. The shape of the distribution suggests that an aetiological trigger in the pre-natal period may be an important determinant of disease. Cite this article: Bone Joint J 2016;98-B:710-14.

Quality of care for patients with a fracture of the hip in major trauma centres: a national observational study.

AIMS: In this study, we aimed to determine whether designation as a major trauma centre (MTC) affects the quality of care for patients with a fracture of the hip. PATIENTS AND METHODS: All patients in the United Kingdom National Hip Fracture Database, between April 2010 and December 2013, were included. The indicators of quality that were recorded included the time to arrival on an orthopaedic ward, to review by a geriatrician, and to operation. The clinical outcomes were the development of a pressure sore, discharge home, length of stay, in-hospital mortality, and re-operation within 30 days. RESULTS: There were 289 466 patients, 49 350 (17%) of whom were treated in hospitals that are now MTCs. Using multivariable logistic and generalised linear regression models, there were no significant differences in any of the indicators of the quality of care or clinical outcomes between MTCs, hospitals awaiting MTC designation and non-MTC hospitals. CONCLUSION: These findings suggest that the regionalisation of major trauma in England did not improve or compromise the overall care of elderly patients with a fracture of the hip. TAKE HOME MESSAGE: There is no evidence that reconfiguring major trauma services in England disrupted the treatment of older adults with a fracture of the hip.

'Big data' reporting guidelines: how to answer big questions, yet avoid big problems.

The extent and depth of routine health care data are growing at an ever-increasing rate, forming huge repositories of information. These repositories can answer a vast array of questions. However, an understanding of the purpose of the dataset used and the quality of the data collected are paramount to determine the reliability of the result obtained. This Editorial describes the importance of adherence to sound methodological principles in the reporting and publication of research using 'big' data, with a suggested reporting framework for future Bone & Joint Journal submissions.

Standardised effect sizes in clinical research: how to compare shoulder surgeons with hip surgeons.

The Bone & Joint Journal provides the latest evidence to guide the clinical practice of orthopaedic surgeons. The benefits of one intervention compared with another are presented using outcome measures; some may be specific to a limb or joint and some are more general health-related quality of life measures. Readers will be familiar with many of these outcome measures and will be able to judge the relative benefits of different interventions when measured using the same outcome tool; for example, different treatments for pain in the knee measured using a particular knee score. But, how should readers compare outcomes between different clinical areas using different outcome measures? This article explores the use of standardised effect sizes.

Designing clinical trials in trauma surgery: overcoming research barriers.

The surgical community is plagued with a reputation for both failing to engage and to deliver on clinical research. This is in part due to the absence of a strong research culture, however it is also due to a multitude of barriers encountered in clinical research; particularly those involving surgical interventions. 'Trauma' amplifies these barriers, owing to the unplanned nature of care, unpredictable work patterns, the emergent nature of treatment and complexities in the consent process. This review discusses the barriers to clinical research in surgery, with a particular emphasis on trauma. It considers how barriers may be overcome, with the aim to facilitate future successful clinical research. Cite this article: Bone Joint Res 2014;3:123-9.

The epidemiology of transient synovitis in Liverpool, UK.

BACKGROUND: The epidemiology of transient synovitis is poorly understood, and the aetiology is unknown, although a suggestion of a viral association predominates. PURPOSE: This population-based study investigated the epidemiology in order to formulate aetiological theories of pathogenesis. PATIENT AND METHODS: Cases in Merseyside were identified between 2004 and 2009. Incidence rates were determined and analysed by age, sex, season and region of residence. Socioeconomic deprivation scores were generated using the Index of Multiple Deprivation, allocated by postcode. Poisson confidence intervals were calculated and Poisson regression was used to check for trends. RESULTS: Two hundred and fifty-nine cases were identified over 5.5 years. The annual incidence was 25.1 (95 % CI 22.1-28.5) per 100,000 0-14 year-olds. Male to female ratio was 3.2:1 (p < 0.001). Mean age at presentation was 5.4 years (95 % CI 5.0-5.8), which demonstrated a near-normal distribution. No relationship was identified between seasonality and incidence (p = 0.64). A correlation was identified with socioeconomic deprivation in Merseyside: incidence rate ratio 1.16 (95 % CI 1.06-1.26, p < 0.001), although further analysis within the subregion of Liverpool did not confirm this finding (p = 0.35). CONCLUSIONS: The normal distribution for age at disease presentation suggests a specific disease entity. The absence of seasonality casts some doubt on the popular theory of a viral aetiology. The absence of a consistent socioeconomic gradient in both Merseyside and Liverpool challenges a previous suggestion of an association with Perthes' disease. This paper provides ecological evidence that may challenge existing aetiological theories, though transient synovitis remains an enigma.

Delayed surgery in displaced paediatric supracondylar fractures: a safe approach? Results from a large UK tertiary paediatric trauma centre.

BACKGROUND: Displaced paediatric supracondylar humeral fractures pose a significant risk of neurovascular injury and consequently have traditionally been treated as a surgical emergency. Recently, the need for emergency surgery has been questioned. We wished to analyse our experience at a large UK tertiary paediatric trauma centre. METHODS: A retrospective case note review was performed on patients with Gartland Grades 2 and 3 supracondylar fractures observed in a 2-year period from July 2008 to July 2010. We divided children into those treated before 12 h (early surgery) and after 12 h (delayed surgery). Analysis was undertaken using Fisher's exact test. RESULTS: Of the 137 patients, 115 were included in the study; median time-to-surgery was 15:30 (range 2:45-62:50); thirty-nine children were treated before 12 h and 76 patients after. In the early surgery group, three children (7.7%) developed a superficial pin-site infection, four children (10.3%) required open reduction, five children (12.8%) sustained an iatrogenic nerve injury, and two children (5%) required reoperation. In the delayed surgery group, one child (1.3%) had a superficial pin-site infection, four children (5.3%) required open reduction, seven children (9.2%) sustained an iatrogenic nerve injury, and two children (2.6%) reoperation. Bivariate analysis of our data using Fisher's exact t test revealed no statistically significant difference between early and delayed surgery groups with regard to infection rates (p = 0.1), iatrogenic nerve injury (p = 0.53) or need for open reduction (p = 0.44). CONCLUSION: Our results indicate that delayed surgery appears to offer a safe management approach in the treatment of displaced supracondylar fractures, but it is important that cases are carefully evaluated on an individual basis. These results indicate that patient transfer to a specialist paediatric centre, often with consequent surgical delay, is a safe management option and also negates the obligation to carry out these procedures at night.