Improving the assessment of axonal injury in early multiple sclerosis.

RATIONALE AND OBJECTIVES: Several quantitative magnetic resonance imaging (MRI) methods are available to measure tissue injury in multiple sclerosis (MS), but their pathological specificity remains limited. The multi-compartment diffusion imaging using the spherical mean technique (SMT) overcomes several technical limitations of the diffusion-weighted image signal, thus delivering metrics with increased pathological specificity. Given these premises, here we assess whether the SMT-derived apparent axonal volume (Vax) provides a better tissue classifier than the diffusion tensor imaging (DTI)-derived axial diffusivity (AD) in the white matter (WM) of MS brains. METHODS: Forty-three treatment-naïve people with newly diagnosed MS, clinically isolated syndrome, or radiologically isolated syndrome and 18 healthy controls (HCs) underwent a 3.0 Tesla MRI inclusive of T1-weighted (T1-w) and T2-w fluid-attenuated inversion recovery (FLAIR) sequences, and multi-b shell diffusion-weighted imaging. In patients only, pre- and post-gadolinium diethylenetriamine penta-acetic acid T1-w sequences were obtained for the evaluation of contrast-active lesions (CELs). Vax and AD were calculated in T2-lesions, chronic black holes (cBHs), and normal appearing (NAWM) in patients and normal WM (NWM) in HCs. Vax and AD values were compared across all the possible combinations of these regions. CELs were excluded from the analyses. RESULTS: Vax differed in all comparisons (p ≤ 0.047 by paired t-test); AD differed in most comparisons (p < 0.001) except between NAWM and NWM, and between cBHs and T2-lesions. Vax had higher accuracy (p ≤ 0.029 by DeLong test) and larger effect size (p ≤ 0.038 by paired t-test) than AD in differentiating areas with even minimal tissue injury. CONCLUSIONS: Vax provides a better radiological quantitative discriminator of different degrees of axonal-mediated tissue injury even between areas with expected minimal pathology. Our data support further studies to assess the readiness of Vax as a measure of outcome for clinical trials on neuroprotection in MS.

Studies of the Energy Dependence of Diboson Polarization Fractions and the Radiation-Amplitude-Zero Effect in WZ Production with the ATLAS Detector.

This Letter presents the first study of the energy dependence of diboson polarization fractions in WZ→ℓνℓ^{'}ℓ^{'}(ℓ,ℓ^{'}=e,µ) production. The dataset used corresponds to an integrated luminosity of 140 fb^{-1} of proton-proton collisions at a center-of-mass energy of 13 TeV recorded by the ATLAS detector. Two fiducial regions with an enhanced presence of events featuring two longitudinally polarized bosons are defined. A nonzero fraction of events with two longitudinally polarized bosons is measured with an observed significance of 5.3 standard deviations in the region with 100200 GeV, where p_{T}^{Z} is the transverse momentum of the Z boson. This Letter also reports the first study of the radiation-amplitude-zero effect. Events with two transversely polarized bosons are analyzed for the ΔY(ℓ_{W}Z) and ΔY(WZ) distributions defined respectively as the rapidity difference between the lepton from the W boson decay and the Z boson and the rapidity difference between the W boson and the Z boson. Significant suppression of events near zero is observed in both distributions. Unfolded ΔY(ℓ_{W}Z) and ΔY(WZ) distributions are also measured and compared to theoretical predictions.

A simple transconjunctival technique for the management of intraconal orbital hydatid cyst.

PURPOSE: To present a simple transconjunctival technique for the excision of intraconal orbital hydatid cysts. METHODS: This retrospective, non-comparative, clinical intervention case study was conducted between April 2018 and October 2023. The study included five patients presented to the Orbital unit of Assiut University Hospital with an intraconal orbital cyst, which histologically proved to be a hydatid cyst. In all cases, a conjunctival incision near the fornix was made depending on the cyst location as revealed by computed tomography (CT) or magnetic resonance imaging (MRI). A traction suture was applied to the two relevant recti muscles to guide the globe toward the desired direction. Blunt orbital dissection was made toward the cyst until exposing its anterior surface. A 20-gauge needle was introduced into the cyst and followed by aspiration of its content. The collapsed cyst was then removed by non-toothed forceps and followed by copious irrigation of the field. The follow-up period ranged from 11 to 58 months. RESULTS: The age of patients ranged from 11 to 44 years. Three were males and two were females. The cyst was iso-dense to the vitreous on CT and iso-intense to the vitreous on MRI. In all cases after aspiration of the content, the collapsed cyst was easily removed. None of the five patients developed recurrence during the follow-up period. CONCLUSION: The removal of the collapsed orbital hydatid cyst in the intraconal space after the aspiration of its content via transconjunctival anterior orbitotomy is a simple, fast technique with early recovery and maximum cosmesis.

Key Disease-Related Genes and Immune Cell Infiltration Landscape in Inflammatory Bowel Disease: A Bioinformatics Investigation.

Inflammatory Bowel Diseases (IBD), which encompass ulcerative colitis (UC) and Crohn's disease (CD), are characterized by chronic inflammation and tissue damage of the gastrointestinal tract. This study aimed to uncover novel disease-gene signatures, dysregulated pathways, and the immune cell infiltration landscape of inflamed tissues. Eight publicly available transcriptomic datasets, including inflamed and non-inflamed tissues from CD and UC patients were analyzed. Common differentially expressed genes (DEGs) were identified through meta-analysis, revealing 180 DEGs. DEGs were implicated in leukocyte transendothelial migration, PI3K-Akt, chemokine, NOD-like receptors, TNF signaling pathways, and pathways in cancer. Protein-protein interaction network and cluster analysis identified 14 central IBD players, which were validated using eight external datasets. Disease module construction using the NeDRex platform identified nine out of 14 disease-associated genes (CYBB, RAC2, GNAI2, ITGA4, CYBA, NCF4, CPT1A, NCF2, and PCK1). Immune infiltration profile assessment revealed a significantly higher degree of infiltration of neutrophils, activated dendritic cells, plasma cells, mast cells (resting/activated), B cells (memory/naïve), regulatory T cells, and M0 and M1 macrophages in inflamed IBD tissue. Collectively, this study identified the immune infiltration profile and nine disease-associated genes as potential modulators of IBD pathogenesis, offering insights into disease molecular mechanisms, and highlighting potential disease modulators and immune cell dynamics.

Health professionals' views and experiences of breaking bad news in the Eastern Mediterranean Region: a scoping review.

Introduction: Breaking bad news is a critical yet challenging aspect of healthcare that requires effective communication skills, empathy, and cultural sensitivity. Health professionals in the World Health Organization's (WHO) Eastern Mediterranean Region face unique cultural and social factors distinct from other parts of the world. This scoping review aims to comprehensively explore the peer-reviewed literature on the health professionals' experiences in delivering bad news within the WHO's Eastern Mediterranean Region. Methods: This scoping review was conducted according to the Joanna Brigg Institute's scoping review methodology and reported utilizing the Preferred Reporting Items for Systematic Reviews extension for scoping review (PRISMA-ScR) guidelines. A search using a combination of keywords and MeSH terms related to "breaking bad news" and "health professionals" was performed in PubMed, Scopus, CINAHL, EBSCO, ERIC via Embase, and Dar Almandumah (Arabic) databases. Common themes were synthesized from studies conducted in the WHO's Eastern Mediterranean Region. Results: Out of 4,883 studies initially identified in the databases, 24 studies met the inclusion criteria, involving a total of 4,710 participants, including physicians, nurses, and residents. The studies were published between 2006 and 2022, predominantly from Iran (n = 12). The majority employed a cross-sectional design (n = 21) or mixed methods (n = 3), with a notable absence of qualitative studies. No studies used theoretical frameworks. More than half of the studies (n = 14) reported that participants had positive attitudes toward breaking bad news. This positivity was evident in their willingness to share bad news, perceived possession of adequate knowledge, positive attitudes, having received training, awareness of accepted approaches, and adherence to protocols. The lack of training and limited awareness of established protocols like SPIKES, ABCDE, and BREAKS for breaking bad news were major concerns among participants. Conclusion: The scoping review reveals both positive and negative experiences of breaking bad news by health professionals in the WHO's Eastern Mediterranean Region. Most studies highlight the need for culturally sensitive targeted education and training programs on breaking bad news. Further research, particularly using qualitative methodologies and theoretical frameworks is warranted.

Bridging the generational gap between nurses and nurse managers: a qualitative study from Qatar.

BACKGROUND: The nursing workforce comprises multiple generations, each with unique values, beliefs, and expectations that can influence communication, work ethic, and professional relationships. In Qatar, the generational gap between nurses and nurse managers poses challenges to effective communication and teamwork, impacting job satisfaction and patient outcomes. AIM: This study investigates the generational gap between nurses and nurse managers in Qatar, aiming to identify strategies to enhance collaboration and create a positive work environment. METHODS: A qualitative research design was used, involving semi-structured interviews with 20 participants, including frontline nurses and senior nurse managers. Participants were purposively sampled to represent different generations. Data were collected through face-to-face and virtual interviews, then transcribed and thematically analyzed. FINDINGS: Four key themes emerged: Optimizing the Work Environment: Older generations preferred transformational and situational leadership, while younger nurses valued respect, teamwork, accountability, and professionalism. Strengthening Work Atmosphere through Communication and values: Older nurses favored face-to-face communication, while younger nurses preferred digital tools. Cultivating Respect and Empathy: Younger nurses emphasized fairness in assignments and promotions, while older nurses focused on empathy and understanding. Dynamic Enhancement of Healthcare Systems: Younger nurses were more adaptable to technology and professional development, while older nurses prioritized clinical care and patient outcomes. CONCLUSION: The study reveals significant generational differences in leadership preferences, communication styles, and adaptability to technology. Addressing these gaps through effective leadership, ongoing education, and open communication can improve job satisfaction and patient care.

The Imaging-Neuropathological Gap in Acute Large Vessel Occlusive Stroke.

While imaging has traditionally played a fundamental role in the selection of patients undergoing endovascular thrombectomy, recent thrombectomy trials involving patients with large ischemic strokes demonstrated a consistent benefit of endovascular thrombectomy across all imaging strata, suggesting that reperfusion benefit may exist independent of current imaging constructs. Although these findings attest to the uniformly beneficial effects of reperfusion, they also shed doubt on the accuracy and utility of our imaging modalities in defining reversible versus irreversible ischemia and challenge the premise of imaging-based selection. We aimed to review the histopathologic studies and clinical trials that have shaped our understanding of current imaging constructs aiming to outline the existing imaging-neuropathological gap that may be far wider than previously perceived.

Assessment of Public Awareness on Alarming Signs of Posttraumatic Intracranial Hemorrhage in the Jeddah Population: A Cross-Sectional Study.

Background Post-traumatic intracranial hemorrhage is a life-threatening condition, and early detection and response can significantly reduce morbidity and mortality rates. The aim of this study was to assess public awareness of the alarming signs of intracranial bleeding after trauma in adults in Jeddah, Saudi Arabia. Methodology From August 2023 to April 2024, a descriptive cross-sectional study was conducted using a five-scale structured questionnaire: demographics, risk factors for road traffic accidents, alarm signs and symptoms, ability to recognize these signs, and participants who experienced head trauma. The study focused on people aged 18 and over living in Jeddah. Results A total of 584 participants were included, with 34.2% males and 65.8% females. Findings revealed that 57% recognized the critical need for medical help after head trauma. Furthermore, only 45% of the population were unaware that low levels of awareness or wakefulness indicate bleeding, reflecting a low awareness level in the adult population. Among children, only 34% recognized changes in eating and lactation habits, and 54% identified continuous crying and irritability. Furthermore, 66% of participants identified loss or change in consciousness as a symptom that required hospital admission, while 60% recognized chronic headaches as a critical sign. Conclusion This study revealed the critical need for public health campaigns to improve awareness and understanding of signs of intracranial post-traumatic bleeding. The results highlighted the importance of early medical interventions to reduce the morbidity and mortality associated with this condition.

Durability of response to immune checkpoint blockade following treatment discontinuation and efficacy of rechallenge in advanced Merkel cell carcinoma.

BACKGROUND: Advanced Merkel cell carcinoma (MCC) has a high response rate to immune checkpoint blockade (ICB) therapy, but the durability of responses once treatment is discontinued remains unclear. We therefore reviewed the long-term outcomes of advanced patients with MCC who discontinued ICB treatment after achieving favorable initial response. METHODS: We performed a retrospective review of advanced patients with MCC treated at a single high-volume referral center, including all patients who received at least one dose of anti-programmed death receptor 1 (ligand) monotherapy for unresectable or metastatic disease, achieved stable disease (SD) or better, and discontinued treatment for a reason other than disease progression. RESULTS: Of 195 advanced patients with MCC treated with ICB, we identified 45 who met the study criteria. Of these, 21 (46.6%) had a complete response (CR) to initial ICB treatment, 23 (51.1%) a partial response and 1 (2.2%) SD. 25 (55.6%) patients discontinued ICB electively and 20 (44.4%) discontinued due to toxicity. In total, 21 of the 45 patients (46.6%) experienced disease progression at a median of 11.3 months (range 2.1-22.7 months) from ICB cessation. There was a lower rate of progression in patients who achieved CR versus non-CR (23.8% vs 66.7%, p=0.006) and a trend towards a lower rate in those who discontinued electively versus due to toxicity (36.0% vs 60.0%, p=0.14). There was a higher risk for progression in patients with viral positive MCC compared with viral negative MCC (75.0 vs 30.8%, p=0.02). 16 of the 21 patients who experienced progression were retreated subsequently with ICB therapy, including both single-agent rechallenge (12) and escalation to combination ICB (4). 11 of 15 evaluable ICB-retreated patients (73.3%) achieved an objective response. CONCLUSIONS: Patients with advanced MCC have a substantial risk of disease progression following treatment discontinuation despite initial favorable ICB response, particularly in those that achieve less than a CR. Most of these patients maintain sensitivity to retreatment with the same drug class. Virus-positive MCC may be a risk factor for post-discontinuation relapse, which should be validated in future studies.

Developing and Validating Entrustable Professional Activities (EPAs) for Rheumatology Fellowship Training Programs in Saudi Arabia: A Delphi Study.

Background: Entrustable professional activities (EPAs) define the core tasks that a graduating rheumatologist needs to perform independently in practice. The objective of this study was to develop and validate EPAs for rheumatology fellowship training programs in Saudi Arabia. Methods: Experts met to develop an initial set of potential end-of-training EPAs by conducting a comprehensive literature review of EPAs and studying the Saudi rheumatology fellowship curriculum. Then, to validate the EPAs, we conducted two rounds of the modified Delphi technique among rheumatology experts in Saudi Arabia. A response rate of 80% was considered and the minimum number of experts needed to be 25 to 30. Descriptive statistics were utilized to describe participants' demographic characteristics and group responses to each statement in all rounds. The experts were asked to rate the relevancy of each EPA using a 5-point Likert scale in both Delphi rounds. Results: In the preliminary phase, four rheumatologists developed an initial set of 36 core EPAs for rheumatology training program in Saudi Arabia. For the two-rounds Delphi techniques, 32 experts were invited to complete the study. The response rate of the first and second round were, 78.12% (25) and 93.75% (30), respectively. The first-round Delphi resulted in a robust consensus on 31 EPAs for rheumatology training. Five EPAs were excluded, and one new EPA was proposed. In the subsequent round, all 32 EPAs achieved strong consensus. The eliminated EPAs likely fell short in one or more of the following areas: relevance to rheumatology practice in Saudi Arabia, overlapping with other EPAs, or practical challenges in the implementation. Conclusion: We have developed and validated a core set of EPAs for rheumatology fellowship training programs in Saudi Arabia. Mapping and identifying milestones for these EPAs are essential steps to follow to enhance workplace curriculum development.

Ethical framework for artificial intelligence in healthcare research: A path to integrity.

The integration of Artificial Intelligence (AI) into healthcare research promises unprecedented advancements in medical diagnostics, treatment personalization, and patient care management. However, these innovations also bring forth significant ethical challenges that must be addressed to maintain public trust, ensure patient safety, and uphold data integrity. This article sets out to introduce a detailed framework designed to steer governance and offer a systematic method for assuring that AI applications in healthcare research are developed and executed with integrity and adherence to medical research ethics.

Tinnitus Prevalence, Associated Characteristics, and Treatment Patterns among Adults in Saudi Arabia.

Tinnitus affects millions of people around the world and causes significant negative impacts on their quality of life (QoL). Tinnitus is rarely examined in Saudi Arabia. This study aimed to estimate the prevalence of tinnitus among adults, explore their experience with tinnitus, investigate the impact of tinnitus on their QoL, and discover their tinnitus management methods. A descriptive cross-sectional study design was performed utilizing a non-probability purposive sampling technique and a face-to-face in-person administered questionnaire. Descriptive statistics and a chi-square test were used to assess the data and find any correlation between the variables. Out of 4860 adults, 320 (males: n = 172; females: n = 148; age range = 18-90 years) had tinnitus, mainly described as a daily, gradual, continuous, whistling, and ringing tinnitus in both ears. Tinnitus prevalence was estimated at 6.54% with a slight predominance in males (6.9%) compared with females (6.2%). Most of the participants were unaware of the cause of their tinnitus. The modal value of the severity of tinnitus signals was severe for both genders. The modal value of the impact of tinnitus on the QoL was moderate for males and severe for females. Sleep, social activities, quiet settings, and concentration were largely affected by tinnitus. Significant associations (p < 0.05) between the impact of tinnitus on the QoL and risk factors, such as gender, age, hearing loss, and hyperacusis were determined. Also, the impact of tinnitus on the QoL was significantly associated (p < 0.05) with the duration of complaints and the severity of tinnitus signals. Approximately, 61% of the participants did not use any tinnitus treatment, while the remaining participants usually used hearing aids, medications, and counseling to manage their tinnitus. By increasing awareness, establishing standard practice, developing guidelines for managing tinnitus, expanding access to suitable interventions, and carrying out additional research, adults living with tinnitus in Saudi Arabia will have better support and, ultimately, an enhancement of their overall well-being.

Enhanced multimodal medical image fusion via modified DWT with arithmetic optimization algorithm.

Medical image fusion (MIF) techniques are proficient in combining medical images in distinct morphologies to obtain a reliable medical analysis. A single modality image could not offer adequate data for an accurate analysis. Therefore, a novel multimodal MIF-based artificial intelligence (AI) method has been presented. MIF approaches fuse multimodal medical images for exact and reliable medical recognition. Multimodal MIF improves diagnostic accuracy and clinical decision-making by combining complementary data in different imaging modalities. This article presents a new multimodal medical image fusion model utilizing Modified DWT with an Arithmetic Optimization Algorithm (MMIF-MDWTAOA) approach. The MMIF-MDWTAOA approach aims to generate a fused image with the significant details and features from each modality, leading to an elaborated depiction for precise interpretation by medical experts. The bilateral filtering (BF) approach is primarily employed for noise elimination. Next, the image decomposition process uses a modified discrete wavelet transform (MDWT) approach. However, the approximation coefficient of modality_1 and the detailed coefficient of modality_2 can be fused interchangeably. Furthermore, a fusion rule is derived from combining the multimodality data, and the AOA model is enforced to ensure the optimum selection of the fusion rule parameters. A sequence of simulations is accomplished to validate the enhanced output of the MMIF-MDWTAOA technique. The investigational validation of the MMIF-MDWTAOA technique showed the highest entropy values of 7.568 and 7.741 bits/pixel over other approaches.

Impact of Climate Change on Cellulitis: A Literature Review.

Climate change is a phenomenon that has had, and will continue to have, wide-ranging effects on the world in both the near and distant future. With regards to human health, research has demonstrated the impact of climate change on heat-related illness, mental health, and vector-borne infectious diseases. Through a review of the literature, this paper aims to elucidate both current and future consequences of climate change on cellulitis, a type of skin infection that is associated with significant morbidity, mortality, and cost. Factors such as elevated temperature, pollution, rising sea levels, and the increased frequency of natural disasters pose an alarming risk for the increased proliferation of infections such as cellulitis. Lastly, in light of these trends, this paper will address potential strategies individuals can implement to reduce the effects of climate change on cellulitis.

Investigation of potential ascorbate peroxidase inhibitors for anti-leishmaniasis therapy.

L eishmaniasis is a prevalent disease that impacts 98 countries and territories, mainly in Africa, Asia, and South America. It can cause substantial illness and death, particularly in its visceral manifestation that can be specifically targeted in the development of medications to combat leishmaniasis. This study has found natural compounds with possible inhibitory activity against APX using a reliable and accurate QSAR model. Despite the severe side effects of current treatments and the absence of an effective vaccination, these compounds show promise as a potential treatment for the disease. Nine hit compounds were found, and subsequent molecular docking was performed. Estradiol cypionate showed the lowest binding energy (- 10.5 kcal/mol), thus showing the strongest binding, and also had the strongest binding affinity, with a ΔGTotal of - 26.31 ± 3.01 kcal/mol, second only to the control molecule. Additionally, three hits viz. cloxacillin-sodium (- 16.57 ± 2.89 kcal/mol), cinchonidine (- 16.04 ± 3.27 kcal/mol), and quinine hydrochloride dihydrate (13.38 ± 1.06 kcal/mol) also showed significant binding affinity. Multiple interactions between drugs and active site residues demonstrated a substantial binding affinity with the target protein. The identified compounds exhibited drug-like effects and were orally bioavailable based on their ADME-toxicology features. Overall, estradiol cypionate, cloxacillin sodium, cinchonidine, and quinine hydrochloride dihydrate all exhibited inhibitory effects on the APX enzyme of Leishmania donovani. These results suggest that further investigation is needed to explore the potential of developing novel anti-leishmaniasis drugs using these compounds.

Corrigendum: Predicting 90-day prognosis for patients with stroke: a machine learning approach.

[This corrects the article DOI: 10.3389/fneur.2023.1270767.].

Role of Inflammation and the NF-κB Signaling Pathway in Hirschsprung's Disease.

Hirschsprung's disease (HSCR, incidence 1/5000 live births) is caused by the failure of neural crest-derived precursors to migrate, survive, proliferate, or differentiate during the embryonic development of the Enteric Nervous System (ENS), which could be disrupted by many factors, including inflammatory processes. The NF-κB family controls several biological processes, including inflammation, neurogenesis, and cell migration. With the aim of studying the potential role of NF-κB in HSCR, we have analyzed the expression of the NF-κB main subunits and other NF-κB-related genes by RT-qPCR in HSCR tissue samples (sub-divided into ganglionic and aganglionic segments). We found decreased gene expression of the NF-κB main subunit RELA but also of NFKBIA, TNFA, TFGBR2, and ERBB3 in the pathologic distal aganglionic segments compared to the proximal ganglionic segments. Moreover, we could also confirm the lower protein expression of RelA/p65 in the aganglionic distal segments by immunofluorescence staining. Further, we show that the expression of RelA/p65 protein in the proximal segments concurs with lymphocyte infiltration in the bowel tissue, indicating a pro-inflammatory activation of p65 in the proximal ganglionic HSCR tissue in the patients analyzed. All in all, our findings suggest that the modulation of NF-κB signaling in the neuro-enteric system does obviously contribute to the pathological effects of HSCR.

Exploring Community Perspectives on Functional Paediatric Habitual Constipation.

(1) Background: Functional habitual constipation (FC) in children is a common gastrointestinal problem. This study aimed to explore the local community's view on this problem, emphasising the challenges that parents face in managing the condition and its impact on the child's quality of life. (2) Methods: A prospective, cross-sectional, community-based study was conducted between March and July 2023. The survey received 933 responses. The target population was adults over 18 years of age living in the Eastern Province of Saudi Arabia. An electronically distributed questionnaire was designed in the Arabic language. (3) Results: The mean knowledge scores were significantly higher in females than males, with t (931) = -2.701 and p = 0.007. The Bonferroni post hoc test results indicated that participants between 20 and 29 years exhibited significantly higher levels of knowledge scores compared to those between 30 and 39 years. Furthermore, the results revealed that those with three or more children had significantly higher knowledge scores compared to those with only one child. (4) Conclusions: This study demonstrated that parents in the local community have a good perceived knowledge of FC, but it needs to be linked with practice. They tended to report high levels of perception and demonstrated better practices. These results emphasise the importance of exploring the local community's view on constipation among children.

A standard set of outcome measures for the comprehensive assessment of oral health and occlusion in individuals with osteogenesis imperfecta.

BACKGROUND: Osteogenesis imperfecta (OI) is a group of inherited connective tissue disorders of varying severity characterized by bone fragility. The primary objective of this international multidisciplinary collaboration initiative was to reach a consensus for a standardized set of clinician and patient-reported outcome measures, as well as associated measuring instruments for dental care of individuals with OI, based on the aspects considered important by both experts and patients. This project is a subsequent to the Key4OI project initiated by the Care4BrittleBones foundation which aims to develop a standard set of outcome measures covering a large domain of factors affecting quality of life for people with OI. An international team of experts comprising orthodontists, pediatric dentists, oral and maxillofacial surgeons, and prosthetic dentists used a modified Delphi consensus process to select clinician-reported outcome measures (CROMs) and patient-reported outcome measures (PROMs) to evaluate oral health in individuals with OI. Important domains were identified through a literature review and by professional expertise (both CROMs and PROMs). In three focus groups of individuals with OI, important and relevant issues regarding dental health were identified. The input from the focus groups was used as the basis for the final set of outcome measures: the selected issues were attributed to relevant CROMs and, when appropriate, matched with validated questionnaires to establish the final PROMs which represented best the specific oral health-related concerns of individuals with OI. RESULTS: Consensus was reached on selected CROMs and PROMs for a standard set of outcome measures and measuring instruments of oral health in individuals with OI. CONCLUSIONS: Our project resulted in consensus statements for standardization oral health PROMs and CROMs in individuals with OI. This outcome set can improve the standard of care by incorporating recommendations of professionals involved in dental care of individuals with OI. Further, it can facilitate research and international research co-operation. In addition, the significant contribution of the focus groups highlights the relevance of dental and oral health-related problems of individuals with OI.