Multimodal Outcomes of Early Open Extended Midline Strip Craniectomy With Bilateral Microbarrel Staving for Correction of Isolated Nonsyndromic Sagittal Synostosis.

AIM: Sagittal synostosis is the most common craniosynostosis. The aim of surgery is to preserve cognitive function and to correct the morphologic changes. In our unit, the authors offer strip craniectomy and microbarrel staving (SMB) performed at 16 to 22 weeks. This study aims to ascertain the 7-year outcome of scaphocephaly correction by SMB. The authors investigate whether the improvement in cranial index (CI) is sustained over time, and report speech and language and neurodevelopmental outcomes. METHODS: All nonsyndromic patients with sagittal synostosis who underwent SMB between 2009 and 2012 were identified from a prospectively created database that recorded anthropometric measurements, quality metrics, and neurodevelopmental outcomes. RESULTS: A total of 27 patients were analyzed. The mean CI preoperatively was 67.5% (±3.5), and at 7 years 78% (±4.8). Regarding neurodevelopmental outcomes at 7 years, SDQ responses did not highlight any concerns for social, emotional, or behavioral outcomes. The vast majority of those who completed a WASI-II assessment performed within the average ranges in terms of cognitive ability. In terms of speech and language 70% of subjects demonstrated receptive language within normal level (WNL). Seventy-seven percent of subjects demonstrated expressive language WNL. CONCLUSIONS: In conclusion, after 7 years of follow-up, early open extended SMB is a safe and effective method for correction of isolated nonsyndromic isolated sagittal synostosis. Across the neurodevelopmental measures and assessments, the children in the current study appeared to perform within expected ranges. Speech and language problems are more prevalent than in the general population.

Guidance to inform research recruitment processes for studies involving critically ill patients.

Clinical research in intensive care units (ICUs) is essential for improving treatments for critically ill patients. However, invitations to participate in clinical research in this situation pose numerous challenges. Studies are frequently initiated within a narrow time window when patients are often unconscious and unable to consent. Consultations or consent discussions must therefore be held with consultees or representatives, usually the patient's relatives. Conversations about research participation in this setting may be difficult, as relatives are often overwhelmed and may feel uneasy about making decisions on behalf of their relatives. In some circumstances, legislation allows doctors to act as consultees or representatives to enrol patients in research. However, there is little good quality evidence on UK stakeholders' perspectives to inform how recruitment is carried out in ICU studies. The Perspectives Study collected evidence on the views of over 1400 stakeholders, including patients, relatives and healthcare practitioners, many of whom had first-hand experience of ICU treatment and research. This evidence was used to inform good practice guidance on recruitment of critically ill patients to research. Established social science methods and empirical ethics were employed to reflect the interests of stakeholders and justify recommendations. This guidance aims to bridge the gap between the legal frameworks and the realities of ICU studies and to ensure that research recruitment processes reflect the views of patients and families. Researchers and an expert Advisory Group brought different perspectives to interpreting the evidence to develop the guidance. In this article we present guidance for future ICU studies.

Surgery for Craniosynostosis: Developing a Psychosocial Booklet for Families.

Parents of children with craniosynostosis may experience psychological distress, particularly during intensive periods of treatment. Yet, recent research indicates parents may not be accessing the support they need to cope with common challenges. The aim was to develop an evidence-based booklet to promote psychological health in families undergoing surgery and to assess the acceptability of the booklet among the craniosynostosis community. Researchers collaborated with UK charity Headlines Craniofacial Support, 5 parent representatives, and 2 specialist clinical psychologists to develop the booklet based on data obtained from online focus groups, individual interviews, and written accounts. The draft booklet was attached to an online acceptability survey and distributed to a broader group of parents and multidisciplinary craniofacial specialists for feedback. A total of 52 complete responses to the online acceptability survey were received, and acceptability (measured by the number of respondents who would recommend the booklet) was 100%. All respondents supported the UK-wide distribution of the booklet. Evidence for psychological intervention in the craniofacial field remains scarce, and specialist teams may be under-resourced to effectively screen and support families. It is hoped this booklet can begin to address the gap in psychological support for families affected by craniosynostosis.

The Craniofacial Collaboration UK: Developmental Outcomes in 7- and 10-Year-Old Children With Sagittal Synostosis.

The Craniofacial Collaboration UK (CC-UK) protocol is a shared agreement across the 4 UK Highly Specialist Craniofacial Centres (HSCCs) to conduct robust neurodevelopmental and psychosocial clinical screening for children with craniosynostosis. This agreement allows for the analysis of outcomes of a homogenous sample of children with single suture craniosynostosis (SSC), a frequent limitation of the existing research. The current study is the latest analysis of CC-UK data on behavioral, cognitive, and psychosocial outcomes. The focus of this analysis is 7- and 10-year-olds with nonsyndromic sagittal synostosis (SS) who have undergone primary corrective surgery and completed routine clinical screening at 1 of the 4 HSCCs since the introduction of the CC-UK protocol. Due to changes in clinical pathways, only data from 3 HSCCs is included to preserve homogeneity. Results show that the majority of children with SS fall within the average range across behavioral and neurodevelopmental domains. A notable exception was a task involving perceptual reasoning and visuomotor skills (Block Design). Although this difference was small and the mean score remained within the average range, it suggests some increased risk of subtle difficulty with such skills for children with SS. Across other measures, there was no consistent evidence of any significantly increased risk of poorer outcomes, in line with findings of previous CC-UK papers. Understanding the psychological phenotype of SS is a key research priority for parents and clinicians, and the current study is another step toward achieving this goal.

A review of core outcome sets (COS) developed for different settings finds there is a subset of outcomes relevant for both research and routine care.

OBJECTIVES: To compare the outcomes selected for the same condition in core outcome sets (COSs) for research with those in COS for the routine care setting. METHODS: A sample of COS was created from the most frequent five health areas within previous systematic reviews of COS for research and COS for routine care. Outcomes were extracted and categorized using an outcome taxonomy. Frequency of outcome domains included within COS were analyzed in subgroups according to research or care setting, patient involvement in COS development and health area. Matched sets of COS were created, where at least one research COS and one routine care COS exist for the same health condition, to identify the outcomes that were recommended for both settings. A similar process was used for a subset of paired COS matched in scope for both intervention and population as well as health condition. RESULTS: The sample of COS comprised: 246 COS for research only, 76 COS for routine care only and 55 COS for both research and routine care. Across the 18 sets matched by health condition the median number (range) of outcomes included in both research COS and routine care COS was 6 (3-15), with differences noted across health areas. For the 11 paired COS matched by scope and health condition, the corresponding figures were 2 (2-8). Across all settings, COS that did not include patients as participants were less likely to include life impact outcomes. CONCLUSION: Within a given health condition, a small number of core outcomes were found to be relevant for both research and care, offering a meaningful starting point for linking research and real-world evaluation.

The overlap between randomised evaluations of recruitment and retention interventions: An updated review of recruitment (Online Resource for Recruitment in Clinical triAls) and retention (Online Resource for Retention in Clinical triAls) literature.

BACKGROUND: The Online Resource for Recruitment in Clinical triAls (ORRCA) and the Online Resource for Retention in Clinical triAls (ORRCA2) were established to organise and map the literature addressing participant recruitment and retention within clinical research. The two databases are updated on an ongoing basis using separate but parallel systematic reviews. However, recruitment and retention of research participants is widely acknowledged to be interconnected. While interventions aimed at addressing recruitment challenges can impact retention and vice versa, it is not clear how well they are simultaneously considered within methodological research. This study aims to report the recent update of ORRCA and ORRCA2 with a special emphasis on assessing crossover of the databases and how frequently randomised studies of methodological interventions measure the impact on both recruitment and retention outcomes. METHODS: Two parallel systematic reviews were conducted in line with previously reported methods updating ORRCA (recruitment) and ORRCA2 (retention) with publications from 2018 and 2019. Articles were categorised according to their evidence type (randomised evaluation, non-randomised evaluation, application and observation) and against the recruitment and retention domain frameworks. Articles categorised as randomised evaluations were compared to identify studies appearing in both databases. For randomised studies that were only in one database, domain categories were used to assess whether the methodological intervention was likely to impact on the alternate construct. For example, whether a recruitment intervention might also impact retention. RESULTS: In total, 806 of 17,767 articles screened for the recruitment database and 175 of 18,656 articles screened for the retention database were added as result of the update. Of these, 89 articles were classified as 'randomised evaluation', of which 6 were systematic reviews and 83 were randomised evaluations of methodological interventions. Ten of the randomised studies assessed recruitment and retention and were included in both databases. Of the randomised studies only in the recruitment database, 48/55 (87%) assessed the content or format of participant information which could have an impact on retention. Of the randomised studies only in the retention database, 6/18 (33%) assessed monetary incentives, 4/18 (22%) assessed data collection location and methods and 3/18 (17%) assessed non-monetary incentives, all of which could have an impact on recruitment. CONCLUSION: Only a small proportion of randomised studies of methodological interventions assessed the impact on both recruitment and retention despite having a potential impact on both outcomes. Where possible, an integrated approach analysing both constructs should be the new standard for these types of evaluations to ensure that improvements to recruitment are not at the expense of retention and vice versa.

"When I was Younger, My Story Belonged to Everyone Else": Co-production of Resources for Adults Living with Craniosynostosis.

OBJECTIVE: Despite growing recognition that congenital craniofacial conditions have lifelong implications, psychological support for adults is currently lacking. The aim of this project was to produce a series of short films about living with craniosynostosis in adulthood, alongside a psychoeducational booklet. DESIGN: The resources were developed using multiple focus groups and meetings attended by researchers, patient representatives, a leading charitable organisation, an award-winning film production company, clinicians, and other experts in the field. RESULTS: An online mixed-methods survey was developed based on prior work to request feedback on the acceptability and utility of the resources from the craniosynostosis community. While data collection to evaluate the resources is ongoing, preliminary results (n = 36) highlight an acceptability rating of 100%. CONCLUSIONS: The resources developed represent a step forward in addressing the unmet information and support needs of adults with craniosynostosis and highlight the benefits of co-production in research.

The Craniofacial Collaboration UK: Developmental Outcomes in 7- and 10-Year-Old Children With Metopic Synostosis.

The Craniofacial Collaboration (CC-UK) is a shared initiative across the Psychology teams attached to 4 highly specialized craniofacial centers in the United Kingdom. The CC-UK aims to address key limitations in the existing craniofacial literature by analyzing data for homogenous samples of children with craniosynostosis. This article presents the fifth wave of CC-UK data collection, focused on 7- and 10-year olds who have undergone primary corrective surgery for metopic synostosis (MS). Data for children with sagittal synostosis and MS have previously been presented at 3 and 5 years. This paper continues to build on this with consideration to older age groups, presenting the first CC-UK analysis of cognitive assessment data using the Wechsler Abbreviated Scale of Intelligence-Second Edition. Results show that the majority of children with MS fall within the average ranges across behavioral and neurodevelopmental domains. However, several domains indicated a trend of heightened concern when compared with normative data, particularly for parent-reported outcomes, suggesting that there may be some subtle difficulties for children with MS. Consideration of how these findings compare with that of previous CC-UK analyses is explored. Further, implications for clinical practice and future research are considered, with the need for longitudinal analyses, as well as data from multiple perspectives (eg, school, parents, and self) at older age points to establish patterns over time. Through collaboration across the highly specialized craniofacial centers, the CC-UK hopes to work toward this goal moving forward.

Potential Anomalous Findings on the Cerberus Subtest of the TEACh 2 in a Pediatric Neuro-Oncology Cohort.

INTRODUCTION: The Test of Everyday Attention for Children 2 (TEA-Ch 2) is a rigorously tested measure of attention, often used in pediatric neuro-oncology settings. Data from one Primary Treatment Centre found a high proportion of children scored in the highest range on the Cerberus subtest. This brief report attempts to answer the question: Does the Cerberus subtest of the TEA-Ch 2 provide outlying scores in the pediatric neuro-oncology population? METHODS: Data representing 62 Cerberus assessments from four primary treatment centers were analyzed. RESULTS: Data showed a substantially higher level of performance on the Cerberus subtest compared to other TEA-Ch2 subtests. Scores were not only higher than expected relative to children's performance on other subtests but also higher than would be expected in the general population. DISCUSSION: Within our data, performance on the Cerberus subset of the TEA-Ch 2 yields somewhat questionable data from which to draw conclusions regarding sustained attentional ability in a pediatric neuro-oncology cohort.

Measuring the success of programmes of care for people living with dementia: a protocol for consensus building with consumers to develop a set of Core Outcome Measures for Improving Care (COM-IC).

INTRODUCTION: The Core Outcome Measures for Improving Care (COM-IC) project aims to deliver practical recommendations on the selection and implementation of a suite of core outcomes to measure the effectiveness of interventions for dementia care. METHODS AND ANALYSIS: COM-IC embeds a participatory action approach to using the Alignment-Harmonisation-Results framework for measuring dementia care in Australia. Using this framework, suitable core outcome measures will be identified, analysed, implemented and audited. The methods for analysing each stage will be codesigned with stakeholders, through the conduit of a Stakeholder Reference Group including people living with dementia, formal and informal carers, aged care industry representatives, researchers, clinicians and policy actors. The codesigned evaluation methods consider two key factors: feasibility and acceptability. These considerations will be tested during a 6-month feasibility study embedded in aged care industry partner organisations. ETHICS AND DISSEMINATION: COM-IC has received ethical approval from The University of Queensland (HREC 2021/HE001932). Results will be disseminated through networks established over the project, and in accordance with both the publication schedule and requests from the Stakeholder Reference Group. Full access to publications and reports will be made available through UQ eSpace (https://espace.library.uq.edu.au/), an open access repository hosted by The University of Queensland.