The central role of mindful parenting in child's emotional regulation and human flourishing: a blueprint perspective.

This article provides an innovative perspective of emotional-regulation and human flourishing which acknowledges the fundamental role of early parent-child experiences in shaping brain structure and functioning involved in emotional regulation and the central role of mindful parenting in facilitating emotional regulation in both parent and child (co-regulation). In this perspective paper the author underlines not only the central role of emotions and emotional regulation in human development and flourishing, but also the importance of maternal mental health, mindfulness, and a connected supportive community during pregnancy and postnatally in facilitating emotional regulation in both the caregiver and the infant and thus promoting secure attachment. The role of alloparenting and how we evolved to share childrearing is introduced, and emotional regulation is described not as an individual phenomenon but a relational embodied process. The associations between right brain functioning, mindfulness and secure attachment, all leading to emotional regulation, wellbeing, and resilience are described. Sharing findings and perspectives offer an opportunity for insights and reflection upon what strategies could be created to promote relational emotional regulation and wellbeing in early life, thus human flourishing leading to a peaceful society.

Comparison of The Results of Sponsored Genetic Testing Panels for Inherited Retinal Diseases.

Background/Objectives: Gene therapy's emergence has made molecular diagnosis for inherited retinal diseases clinically significant. Free genetic testing panels have improved testing access in clinical practice, yet the interpretation of results, especially variants of unknown significance (VUS), remains challenging and requires expertise. This study shares our experience in utilizing sponsored IRD panel tests by Invitae and Blueprint Genetics (BG), reporting their positivity rates, and comparing their reclassification of variants through amendments. Methods: This retrospective study analyzed genetic test reports from patients who underwent testing via Invitae or BG panels. A positive test was determined if there was a pathogenic mutation in an autosomal dominant gene, two pathogenic mutations in an autosomal recessive gene, or a pathogenic mutation in an X-linked gene in a male patient. Results: The testing positivity rates were 34.9% for Invitae (n = 109) and 42.1% for BG (n = 107). Invitae had more pathogenic variants per report (0.87 vs. 0.58 variants, p = 0.0038) and issued more amendments than BG (0.54 vs. 0.03 amendments; p < 0.01). Of the Invitae variant classification changes, 66.2% switched a VUS to benign. In the BG group, 75% of variant reclassifications changed a VUS to pathogenic. As a result of the Invitae amendments, 88% did not change the overall report result. Conclusions: While free-of-charge genetic testing panels offer valuable insights for diagnosing IRD, limitations such as low diagnostic yield and variant classification discrepancies persist between Invitae and BG. VUS should not be considered pathogenic in the clinical decision-making process. Careful interpretation of genetic testing is required.

The Information Technology (IT) Infrastructure of the Multicenter Archipelago of Ovarian Cancer Research Biobank: A Potential Blueprint for Other Biobanks.

Objective: Biobanks play a crucial role in fundamental and translational research by storing valuable biomaterials and data for future analyses. However, the design of their information technology (IT) infrastructures is often customized to specific requirements, thereby lacking the ability to be used for biobanks comprising other (types of) diseases. This results in substantial costs, time, and efforts for each new biobank project. The Dutch multicenter Archipelago of Ovarian Cancer Research (AOCR) biobank has developed an innovative, reusable IT infrastructure capable of adaptation to various biobanks, thereby enabling cost-effective and efficient implementation and management of biobank IT systems. Methods and Results: The AOCR IT infrastructure incorporates preexisting biobank software, mainly managed by Health-RI. The web-based registration tool Ldot is used for secure storage and pseudonymization of patient data. Clinicopathological data are retrieved from the Netherlands Cancer Registry and the Dutch nationwide pathology databank (Palga), both established repositories, reducing administrative workload and ensuring high data quality. Metadata of collected biomaterials are stored in the OpenSpecimen system. For digital pathology research, a hematoxylin and eosin-stained slide from each patient's tumor is digitized and uploaded to Slide Score. Furthermore, adhering to the Findable, Accessible, Interoperable, and Reusable (FAIR) principles, genomic data derived from the AOCR samples are stored in cBioPortal. Conclusion: The IT infrastructure of the AOCR biobank represents a new standard for biobanks, offering flexibility to handle diverse diseases and types of biomaterials. This infrastructure bypasses the need for disease-specific, custom-built software, thereby being cost- and time-effective while ensuring data quality and legislative compliance. The adaptability of this infrastructure highlights its potential to serve as a blueprint for the development of IT infrastructures in both new and existing biobanks.

Developing and testing a produce prescription implementation blueprint to improve food security in a clinical setting: a pilot study protocol.

BACKGROUND: Food insecurity is common in the United States, especially in Rhode Island, where it affects up to 33% of residents. Food insecurity is associated with adverse health outcomes and disproportionally affects people from minoritized backgrounds. Produce prescription programs, in which healthcare providers write "prescriptions" for free or reduced cost vegetables, have been used to address food insecurity and diet-related chronic disease. Although there is growing evidence for the effectiveness of produce prescription programs in improving food security and diet quality, there have been few efforts to use implementation science methods to improve the adoption of these programs. METHODS: This two-phase pilot study will examine determinants and preliminary implementation and effectiveness outcomes for an existing produce prescription program. The existing program is funded by an Accountable Care Organization in Rhode Island and delivered in primary care practices. For the first phase, we conducted a formative evaluation, guided by the Consolidated Framework for Implementation Research 2.0, to assess barriers, facilitators, and existing implementation strategies for the produce prescription program. Responses from the formative evaluation were analyzed using a rapid qualitative analytic approach to yield a summary of existing barriers and facilitators. In the second phase, we presented our formative evaluation findings to a community advisory board consisting of primary care staff, Accountable Care Organization staff, and staff who source and deliver the vegetables. The community advisory board used this information to identify and refine a set of implementation strategies to support the adoption of the program via an implementation blueprint. Guided by the implementation blueprint, we will conduct a single-arm pilot study to assess implementation antecedents (i.e., feasibility, acceptability, appropriateness, implementation climate, implementation readiness), implementation outcomes (i.e., adoption), and preliminary program effectiveness (i.e., food and nutrition security). The first phase is complete, and the second phase is ongoing. DISCUSSION: This study will advance the existing literature on produce prescription programs by formally assessing implementation determinants and developing a tailored set of implementation strategies to address identified barriers. Results from this study will inform a future fully powered hybrid type 3 study that will use the tailored implementation strategies and assess implementation and effectiveness outcomes for a produce prescription program. TRIAL REGISTRATION: Clinical trials: NCT05941403 , Registered June 9, 2023.

A blueprint for health technology assessment capacity building: lessons learned from Malta.

OBJECTIVES: The development and strengthening of health technology assessment (HTA) capacity on the individual and organizational level and the wider environment is relevant for cooperation on HTAs. Based on the Maltese case, we provide a blueprint for building HTA capacity. METHODS: A set of activities were developed based on Pichler et al.'s framework and the starting HTA capacity in Malta. Individual level activities focused on strengthening epidemiological and health economic skills through online and in-person training. On the organizational level, a new HTA framework was developed which was subsequently utilized in a shadow assessment. Awareness campaign activities raised awareness and support in the wider environment where HTAs are conducted and utilized. RESULTS: The time needed to build HTA capacity exceeded the planned two years accommodating the learning progress of the assessors. In addition to the planned trainings, webinars supplemented the online courses, allowing for more knowledge exchange. The advanced online course was extended over time to facilitate learning next to the assessors' daily tasks. Training sessions were added to implement the new economic evaluation framework, which was utilized in a second shadow assessment. Awareness by decision-makers was achieved with reports, posters, and an article on the current and developing HTA capacity. CONCLUSIONS: It takes time and much (hands-on) training to build skills for conducting complex assessment such as HTAs. Facilitating exchange with knowledgeable parties is crucial for succeeding as well as the buy-in of local managers motivating staff. Decision-makers need to be on-boarded for the continued success of HTA capacity building.

Artificial Intelligence Sensing: Effective Flavor Blueprinting of Tea Infusions for a Quality Control Perspective.

Tea infusions are the most consumed beverages in the world after water; their pleasant yet peculiar flavor profile drives consumer choice and acceptance and becomes a fundamental benchmark for the industry. Any qualification method capable of objectifying the product's sensory features effectively supports industrial quality control laboratories in guaranteeing high sample throughputs even without human panel intervention. The current study presents an integrated analytical strategy acting as an Artificial Intelligence decision tool for black tea infusion aroma and taste blueprinting. Key markers validated by sensomics are accurately quantified in a wide dynamic range of concentrations. Thirteen key aromas are quantitatively assessed by standard addition with in-solution solid-phase microextraction sampling followed by GC-MS. On the other hand, nineteen key taste and quality markers are quantified by external standard calibration and LC-UV/DAD. The large dynamic range of concentration for sensory markers is reflected in the selection of seven high-quality teas from different geographical areas (Ceylon, Darjeeling Testa Valley and Castleton, Assam, Yunnan, Azores, and Kenya). The strategy as a sensomics-based expert system predicts teas' sensory features and acts as an AI smelling and taste machine suitable for quality controls.

Staged esthetic crown lengthening: Classification and guidelines for periodontal-restorative therapy.

OBJECTIVE: This article presents technical guidelines for perio-restorative esthetic crown lengthening, along with a discussion of the biologic rationale. A classification system is proposed to assist in treatment planning and sequencing the surgical and restorative phases. CLINICAL CONSIDERATIONS: When esthetic crown lengthening is performed as an adjunct to restorative therapy, the surgical approach must be determined by the anticipated position of the restorative margins. The removal of sufficient bone to achieve the desired clinical crown length and preserve the supracrestal gingival tissue dimensions is facilitated by the use of a surgical guide fabricated according to the design of the restorations. A staged approach allows sequencing the provisional restoration to minimize unesthetic sequelae during the healing period. Inadequate bone resection and/or alteration of the soft tissue dimensions results in delayed healing, leading to coronal gingival rebound and biologic width impingement. CONCLUSION: The identification and preservation of appropriate restorative and biologic landmarks is essential for success in pre-prosthetic esthetic crown lengthening treatment. A staged approach improves the esthetic management during the postsurgical healing and maturation period. CLINICAL SIGNIFICANCE: A restorative driven classification system for sequencing and staging adjunctive esthetic crown lengthening procedures is presented. Technical guidelines to enhance gingival margin predictability are suggested, accompanied by relevant evidence. In addition, wound healing timelines following gingival and osseous resection are provided.

Predicting the next pandemic: VACCELERATE ranking of the WorldHealth Organization's Blueprint forAction toPreventEpidemics.

INTRODUCTION: The World Health Organization (WHO)'s Research and Development (R&D) Blueprint for Action to Prevent Epidemics, a plan of action, highlighted several infectious diseases as crucial targets for prevention. These infections were selected based on a thorough assessment of factors such as transmissibility, infectivity, severity, and evolutionary potential. In line with this blueprint, the VACCELERATE Site Network approached infectious disease experts to rank the diseases listed in the WHO R&D Blueprint according to their perceived risk of triggering a pandemic. VACCELERATE is an EU-funded collaborative European network of clinical trial sites, established to respond to emerging pandemics and enhance vaccine development capabilities. METHODS: Between February and June 2023, a survey was conducted using an online form to collect data from members of the VACCELERATE Site Network and infectious disease experts worldwide. Participants were asked to rank various pathogens based on their perceived risk of causing a pandemic, including those listed in the WHO R&D Blueprint and additional pathogens. RESULTS: A total of 187 responses were obtained from infectious disease experts representing 57 countries, with Germany, Spain, and Italy providing the highest number of replies. Influenza viruses received the highest rankings among the pathogens, with 79 % of participants including them in their top rankings. Disease X, SARS-CoV-2, SARS-CoV, and Ebola virus were also ranked highly. Hantavirus, Lassa virus, Nipah virus, and henipavirus were among the bottom-ranked pathogens in terms of pandemic potential. CONCLUSION: Influenza, SARS-CoV, SARS-CoV-2, and Ebola virus were found to be the most concerning pathogens with pandemic potential, characterised by transmissibility through respiratory droplets and a reported history of epidemic or pandemic outbreaks.

Blueprinting of Assessment for the Lower Limb in Undergraduate Anatomy Curriculum.

BACKGROUND:  Competency-based medical education is followed by most of the medical schools in India. Health universities are changing their question paper pattern and practical assessment pattern to ensure valid and reliable evaluation of student learning but assessment of entire curriculum competencies is a difficult task to achieve. Assessment blueprinting can provide a guiding map for balanced and rational assessment. It ensures similar exam content from year to year (over- or under-representing a topic can be avoided). The assessment blueprint provides ideas of appropriate formats for the knowledge/skills being assessed. The study aims to observe facilitators' perceptions and develop an assessment blueprint for the anatomy curriculum. METHODS:  This educational mixed-method study was conducted in the Department of Anatomy of All India Institute of Medical Sciences, Jodhpur, India. Perceptions of 13 facilitators of the Anatomy Department about assessment blueprinting were observed on a five-point Likert scale. Topics of the lower limb were identified based on the Undergraduate Curriculum Volume-1 document provided by the National Medical Council of the Government of India. Weightage to each topic was given on the basis of anatomical importance and clinical significance at the undergraduate level. Marks for the topic as per relative weightage were calculated. The blueprint was prepared and validated by the subject expert of the institute. An example question paper template was prepared as per the assessment blueprint. RESULTS:  In the present study, all 13 (100%) participants agreed that there is a need for a valid assessment framework to maintain the reliability of assessment, 11 (85%) participants agreed that an assessment blueprint can provide a valid tool for assessment, and 10 (77%) participants agreed that assessments are not planned as per learning domain requirement, and planning of assessment as per blueprint requires more time and resource. Of the participants, eight (62%) agreed that all topics or competencies are not included while setting the question paper. The blueprint for assessment of the lower limb provided an idea of high-weightage and low-weightage topics and the suitable types of questions for these topics. CONCLUSION: Assessment is the key element of competency-based medical education. For a complete, valid, and reliable assessment, a blueprint for assessment is the need of the hour. Assessment blueprinting in this study showed that the anterior thigh region, hip joint, knee joint, and vascular and nerve supply of the lower limb are topics with maximum weightage for the lower limb in the medical anatomy undergraduate curriculum. It can help in reducing under or over-representation of content. With the help of a blueprint, the examiners can assess the majority of content without any intentional or unintentional bias.

Blueprint designing and validation for competency-based curriculum for theory assessment in community medicine.

Background: Blueprint provides a base for assessment by assigning proportionate weightage to various content areas and helps the paper setter to construct a uniform and valid assessment. This study aimed to design and validate a blueprint for theory in Community Medicine as per the new curriculum for Medical Undergraduates in India. Methods: Blueprint in community medicine was designed by assigning impact score (I) and frequency score (F) for the competencies. Blueprint was validated using the Content Validity Index (CVI), and inter-rater agreement for subject experts using Fleiss' kappa statistics was calculated. Feedback from faculty and students was obtained afterward to assess the postimplementation response. Results: Blueprint was designed by an expert group where impact score and frequency score were assigned to 146 competencies in the theory of Community Medicine. In Delphi survey I, 63.2% of subject experts responded, while in Delphi survey II, a response rate of 58.3% was achieved. Value of the Fleiss' Kappa test for an inter-rater agreement was 0.68, i.e. "substantial agreement," while CVI among the raters came out to be 0.86, i.e. overall valid assessment. Feedback of faculty (n = 11) suggested that the blueprint was helpful and standardized the paper setting, whereas feedback from students (n = 138) depicted that it helped in preparing for exams, and they would recommend it to other students. Conclusion: Validated blueprint by consensus of subject experts has impact score and frequency score along with topic-wise distribution of marks for the convenience of faculty and its utility is well proven among learners too.

Investigating the availability of 3D-printed firearm designs on the clear web.

The release of the plans of the 3D-printed Liberator firearm sparked a wave of new designs from creators worldwide, resulting in an extensive collection of 3D-printed firearm plans, in particular blueprints, and parts available for almost unrestricted download on the internet. Identifying and categorizing the diverse range of 3D-printed firearms and components pose a challenge due to the abundance of designs available. Between 2021 and April 2023, data was collected on over 2,100 3D-printed firearm plans. While blueprints of fully 3D-printed firearms initially dominated the scene, hybrid designs and parts kit completions / conversions (PKC) have gained popularity for their improved reliability and performance. The now highly networked community offers considerable support with detailed instructions and procedures, providing precise guidance for construction. This systematic classification, grouping and structuration of the recorded data on the Clear Web supported the identification of patterns of the main threat trends related to 3D-printed firearms.

A risk score model based on lipid metabolism-related genes could predict response to immunotherapy and prognosis of lung adenocarcinoma: a multi-dataset study and cytological validation.

BACKGROUND: Lipid metabolism is a key factor in tumorigenesis and drug resistance, and models related to lipid metabolism have shown potential to predict survival and curative effects of adjuvant therapy in various cancers. However, the relationship between lipid metabolism and prognosis and treatment response of lung adenocarcinoma (LUAD) are still unclear. METHODS: We enrolled seven bulk RNA-sequence datasets (GSE37745, GSE19188, GSE30219, GSE31547, GSE41271, GSE42127, and GSE72094) from the GEO database and one single-cell RNA-sequencing dataset (GSE117570) from the TISCH2 database. Non-negative matrix factorization (NMF) was utilized to construct the risk score model based on lipid score calculated by GSVA algorithm. Phs000452.v3, PMID: 26359337, PMID: 32472114, PRJEB23709 datasets were used to test the response to immunotherapy. Drug sensitivity analysis was assessed according to the GDSC database, and immunotherapy response was evaluated using the Wilcoxon test. Cellular function assays including clone formation, EDU assays and flow cytometry were implemented to explore the phenotype alteration caused by the knockdown of PTDSS1, which is one of key gene in risk score model. RESULTS: We analyzed both bulk and single-cell RNA sequencing data to establish and validate a risk score model based on 18 lipid metabolism-related genes with significant impact on prognosis. After divided the patients into two groups according to risk score, we identified differences in lipid-related metabolic processes and a detailed portrait of the immune landscapes of high- and low-risk groups. Moreover, we investigated the potentials of our risk score in predicting response to immunotherapy and drug sensitivity. In addition, we silenced PTDSS1 in LUAD cell lines, and found that the proliferation of the cells was weakened, and the apoptosis of the cells was increased. CONCLUSION: Our study highlights the crucial roles of lipid metabolism in LUAD and provides a reliable risk score model, which can aid in predicting prognosis and response to immunotherapy. Furthermore, we investigated the roles of PTDSS1 in LUAD carcinogenesis, which showed that PTDSS1 regulated proliferation and apoptosis of LUAD cells.

Analysis of the Health Examination Service Process Using Service Blueprint: Focus on the Older Adult Patient in South Korea.

As the older adult population grows, the paradigm of aging is shifting from simply living longer to living longer while maintaining health. This shift has led to a transformation in healthcare from passive to proactive approaches, emphasizing disease prevention. Health examination services have seen significant growth as they transition from being solely diagnostic processes to crucial tools for disease prevention. This study focuses on the health examination service industry, particularly in the context of the older adult population, and aims to develop a service blueprint to identify challenges and solutions in utilizing these services. The research employs the service blueprint methodology to map out the health examination service process comprehensively. The distinction is made between customer interactions and internal procedures that are observable and those that are not. Through a comprehensive analysis of the service process, it is possible to identify potential instances of customer unhappiness. These instances primarily occur during the initial interaction between older clients and the service, as well as when they receive their examination findings. There are several factors that contribute to discontent among individuals, namely the insufficient comprehension of the needs of the aged by service providers and the provision of substandard facilities. The study suggests strategies to improve customer satisfaction, such as dedicated personnel for older adult assistance, specialized education for staff, and better facilities tailored for older adult patients. Furthermore, the research highlights the significance of effectively addressing both areas of critical discontent and elements that enhance satisfaction in the process of service design. This research provides a detailed analysis of health examination services for the older adult, highlighting opportunities for improvement through enhanced customer experiences and specialized services.

A service blueprint approach to prioritize operational improvements in a new outpatient clinic.

As a US-based health care system, Mayo Clinic faced considerable challenges opening a new affiliated outpatient facility in the UK at the beginning of the COVID-19 pandemic, which severely affected patient volumes and staffing. As COVID-19 restrictions were eased, the clinic had to prioritize gradual improvements to reestablish service while using resources responsibly. To assist in understanding the current state and to isolate challenges, we elected to develop a service blueprint. We describe how we did this during the COVID-19 pandemic with the use of both face-to-face and virtual services. In many industries, service blueprints are used to help with the design, delivery, and management of new and established services. Although they share some features with value stream mapping, service blueprints often focus on human tasks and the customer's service experience, while value stream maps emphasize information or product flows and capabilities. Several themes for prioritized improvement efforts were identified for future work. In addition, the service blueprint workshops led to a much better understanding of how each person's work affected the other team members and the patient experience. We learned that service blueprints are an efficient way to identify and anticipate critical operational interdependencies and team dynamics that will affect the patient experience when building new clinical services.

Invasive Breast Cancer with HER2 ≥4.0 and <6.0: Risk Classification and Molecular Typing by a 21-Gene Expression Assay and MammaPrint Plus BluePrint Testing.

Purpose: To investigate the HER2 status and clinicopathological features in invasive breast cancer with HER2 ≥4.0 and <6.0, which has always been controversial. Methods: Forty breast cancer cases with HER2 ≥4.0 and <6.0 by fluorescence in situ hybridization (FISH) were collected and classified into two groups based on the HRE2/CEP17 ratio (Group A: ≥2.0, n=22; Group B: <2.0, n=18). Clinicopathological characteristics, HER2 status, risk classification, and molecular typing were further analyzed and compared by 21-Gene expression assay and MammaPrint plus BluePrint test. Results: The majority of cases in both groups were invasive carcinoma (NOS), with histological grade II, HR+, Ki-67 ≥20%, HER2 2+, and a high risk of recurrence, although younger patients and lymph node metastases were more common in Group A. Surprisingly, all HR+ breast cancers in both groups were classified as luminal-type, HR- cases were all basal-type or unknown, and the index of HER2 in all cases was <0.000 using the BluePrint test, which indicated that HER2 status should be negative. Furthermore, the level of HER2 mRNA expression in all cases of both groups was <10.7, which was defined as HER2 negative by the 21-Gene expression assay. In addition, 10 patients of Group A received anti-HER2 neoadjuvant therapy; only one patient with HR- achieved Grade 5 based on the Miller-Payne system, whereas none of the patients achieved pathological complete response (pCR) based on the Residual Cancer Burden system. Conclusion: Group A breast cancer, which has always been unquestionably diagnosed as HER2 amplification, was more likely to be HER2 negative and derived less benefit from anti-HER2 neoadjuvant chemotherapy. Group A breast cancer should be distinguished from classical HER2-positive breast cancers when assessing HER2 FISH, and a larger cohort of Group A patients should be included in further studies.

Developing a Blueprint for Theoretical Assessment of Biochemistry of Phase I MBBS Students.

Here, we have systematically developed a blueprint for the biochemistry theory assessment of phase I MBBS students in India which we have been using for both formative and summative assessments for the past 2 academic years. The blueprint has ensured the content validity and construct reliability and fairness of the assessment.

Artificial Intelligence decision-making tools based on comprehensive two-dimensional gas chromatography data: the challenge of quantitative volatilomics in food quality assessment.

Effective investigation of food volatilome by comprehensive two-dimensional gas chromatography with parallel detection by mass spectrometry and flame ionization detector (GC×GC-MS/FID) gives access to valuable information related to industrial quality. However, without accurate quantitative data, results transferability over time and across laboratories is prevented. The study applies quantitative volatilomics by multiple headspace solid phase microextraction (MHS-SPME) to a large selection of hazelnut samples (Corylus avellana L. n = 207) representing the top-quality selection of interest for the confectionery industry. By untargeted and targeted fingerprinting, performant classification models validate the role of chemical patterns strongly correlated to quality parameters (i.e., botanical/geographical origin, post-harvest practices, storage time and conditions). By quantification of marker analytes, Artificial Intelligence (AI) tools are derived: the augmented smelling based on sensomics with blueprint related to key-aroma compounds and spoilage odorant; decision-makers for rancidity level and storage quality; origin tracers. By reliable quantification AI can be applied with confidence and could be the driver for industrial strategies.

Building the foundation for a community-generated national research blueprint for inherited bleeding disorders: facilitating research through infrastructure, workforce, resources and funding.

BACKGROUND: The National Hemophilia Foundation (NHF) conducted extensive, inclusive community consultations to guide prioritization of research in coming decades in alignment with its mission to find cures and address and prevent complications enabling people and families with blood disorders to thrive. RESEARCH DESIGN AND METHODS: With the American Thrombosis and Hemostasis Network, NHF recruited multidisciplinary expert working groups (WG) to distill the community-identified priorities into concrete research questions and score their feasibility, impact, and risk. WG6 was charged with identifying the infrastructure, workforce development, and funding and resources to facilitate the prioritized research. Community input on conclusions was gathered at the NHF State of the Science Research Summit. RESULTS: WG6 detailed a minimal research capacity infrastructure threshold, and opportunities to enable its attainment, for bleeding disorders centers to participate in prospective, multicenter national registries. They identified challenges and opportunities to recruit, retain, and train the diverse multidisciplinary care and research workforce required into the future. Innovative collaborative approaches to trial design, resource networking, and funding to surmount obstacles facing research in rare disorders were elucidated. CONCLUSIONS: The innovations in infrastructure, workforce development, and resources and funding proposed herein may contribute to facilitating a National Research Blueprint for Inherited Bleeding Disorders.

Research is critical to advancing the diagnosis and care of people with inherited bleeding disorders (PWIBD). This research requires significant infrastructure, including people and resources. Hemophilia treatment centers (HTC) need many different skilled care professionals including doctors, nurses, and other providers; also statisticians, data managers, and other experts to process patients' clinical information into research. Attracting diverse qualified professionals to the clinical and research work requires long-term planning, recruiting individuals in training programs and retaining them as they become experts. Research infrastructure includes physical servers running database software, networks that link them, and the environment in which these components function. US Centers for Disease Control and Prevention (CDC) and American Thrombosis and Hemostasis Network (ATHN) coordinate and fund data collection at HTCs on the health and well-being of thousands of PWIBD into a registry used in research studies.National Hemophilia Foundation (NHF) and ATHN asked our group of health care professionals, technology experts, and lived experience experts (LEE) to identify the infrastructure, workforce, and resources needed to do the research most important to PWIBD. We identified the types of CDC/ATHN studies all HTCs should be able to perform, and the physical and human infrastructure this requires. We prioritized finding the best clinical trial designs to study inherited bleeding disorders, identifying ways to share personnel and tools between HTCs, and innovating how research is governed and funded. Involving LEEs in designing, managing, and carrying out research will be key in conducting research to improve the lives of PWIBD.