Diagnostic delay of sarcoidosis: an integrated systematic review.

BACKGROUND: Sarcoidosis is a chronic inflammatory granulomatous disease of unknown cause. Delays in diagnosis can result in disease progression and poorer outcomes for patients. Our aim was to review the current literature to determine the overall diagnostic delay of sarcoidosis, factors associated with diagnostic delay, and the experiences of people with sarcoidosis of diagnostic delay. METHODS: Three databases (PubMed/Medline, Scopus, and ProQuest) and grey literature sources were searched. Random effects inverse variance meta-analysis was used to pool mean diagnostic delay in all types of sarcoidosis subgroup analysis. Diagnostic delay was defined as the time from reported onset of symptoms to diagnosis of sarcoidosis. RESULTS: We identified 374 titles, of which 29 studies were included in the review, with an overall sample of 1531 (694 females, 837 males). The overall mean diagnostic delay in all types of sarcoidosis was 7.93 months (95% CI 1.21 to 14.64 months). Meta-aggregation of factors related to diagnostic delay in the included studies identified three categories: (1) the complex and rare features of sarcoidosis, (2) healthcare factors and (3) patient-centred factors. Meta-aggregation of outcomes reported in case studies revealed that the three most frequent outcomes associated with diagnostic delay were: (1) incorrect diagnosis, (2) incorrect treatment and (3) development of complications/disease progression. There was no significant difference in diagnostic delay between countries with gatekeeper health systems (where consumers are referred from a primary care clinician to specialist care) and countries with non-gatekeeper systems. No qualitative studies examining people's experiences of diagnostic delay were identified. CONCLUSION: The mean diagnostic delay for sarcoidosis is almost 8 months, which has objective consequences for patient management. On the other hand, there is a paucity of evidence about the experience of diagnostic delay in sarcoidosis and factors related to this. Gaining an understanding of people's experiences while seeking a diagnosis of sarcoidosis is vital to gain insight into factors that may contribute to delays, and subsequently inform strategies, tools and training activities aimed at increasing clinician and public awareness about this rare condition. TRIAL REGISTRATION: PROSPERO Registration number: CRD42022307236.

Diagnostic delay of sarcoidosis: Protocol for an integrated systematic review.

INTRODUCTION: Sarcoidosis is a rare systemic inflammatory granulomatous disease of unknown cause. It can manifest in any organ. The incidence of sarcoidosis varies across countries, and by ethnicity and gender. Delays in the diagnosis of sarcoidosis can lead to extension of the disease and organ impairment. Diagnosis delay is attributed in part to the lack of a single diagnostic test or unified commonly used diagnostic criteria, and to the diversity of disease manifestations and symptom load. There is a paucity of evidence examining the determinants of diagnostic delay in sarcoidosis and the experiences of people with sarcoidosis related to delayed diagnosis. We aim to systematically review available evidence about diagnostic delay in sarcoidosis to elucidate the factors associated with diagnostic delay for this disease in different contexts and settings, and the consequences for people with sarcoidosis. METHODS AND ANALYSIS: A systematic search of the literature will be conducted using PubMed/Medline, Scopus, and ProQuest databases, and sources of grey literature, up to 25th of May 2022, with no limitations on publication date. We will include all study types (qualitative, quantitative, and mixed methods) except review articles, examining diagnostic delay, incorrect diagnosis, missed diagnosis or slow diagnosis of all types of sarcoidosis across all age groups. We will also examine evidence of patients' experiences associated with diagnostic delay. Only studies in English, German and Indonesian will be included. The outcomes we examine will be diagnostic delay time, patients' experiences, and factors associated with diagnostic delay in sarcoidosis. Two people will independently screen the titles and abstracts of search results, and then the remaining full-text documents against the inclusion criteria. Disagreements will be resolved with a third reviewer until consensus is reached. Selected studies will be appraised using the Mixed Methods Appraisal Tool (MMAT). A meta-analysis and subgroup analyses of quantitative data will be conducted. Meta-aggregation methods will be used to analyse qualitative data. If there is insufficient data for these analyses, a narrative synthesis will be conducted. DISCUSSION: This review will provide systematic and integrated evidence on the diagnostic delay, associated factors, and experiences of diagnosis delay among people with all types of sarcoidosis. This knowledge may shed light on ways to improve diagnosis delays in diagnosis across different subpopulations, and with different disease presentations. ETHICS AND DISSEMINATION: Ethical approval will not be required as no human recruitment or participation will be involved. Findings of the study will be disseminated through publications in peer-reviewed journals, conferences, and symposia. TRIAL REGISTRATION: PROSPERO Registration number: CRD42022307236. URL of the PROSPERO registration: https://www.crd.york.ac.uk/PROSPEROFILES/307236_PROTOCOL_20220127.pdf.

Relação radiologista-paciente no exame ultrassonográfico obstétrico sob o enfoque bioético / Radiologist-patient relationship in the obstetrics ultrasound exam under bioethics? view

Este trabalho objetivou conhecer a relação radiologista-paciente durante os exames de ultrassonografia obstétrica sob os pontos de vista materno e do radiologista, com enfoque do principialismo. Com abordagem qualitativa, exploratória e transversal foram entrevistadas 10 gestantes e 10 radiologistas, mediante entrevista semi-estruturada, gravada e transcrita literalmente. As diretrizes metodológicas do Discurso do Sujeito Coletivo (DSC) foram utilizadas para a seleção das ideias centrais e das expressães-chave. Foram identificadas as seguintes representaçães sociais por parte das gestantes:-informaçães sobre o feto-, -dia do parto-,-orientação sobre o exame-, -expectativa negativa superada-, -bondade-,-tratar bem-,-pouca expectativa- e -atenção-. E por parte dos radiologistas: -tranquilizar a paciente-,-administrar situaçães-, -tratar bem- e -atenção, respeito, confiança e carinho- para com a paciente. Constatou-se que na relação radiologista-paciente os conhecimentos técnicos específicos, o relacionamento humano e os cuidados com a gestante são imprescindíveis para a valorização da vida humana e dos preceitos bioéticos.

Arterial switch with full-flow cardiopulmonary bypass and limited circulatory arrest: neurodevelopmental outcome.

OBJECTIVES: Neonatal cardiac surgery has been associated with unfavorable neurodevelopmental events. We investigated a patient cohort operated on predominantly with full-flow cardiopulmonary bypass (150 mL x kg(-1) x min(-1), alpha-stat, alpha-blockade, median arrest = 6 minutes, temperature of 22 degrees C) as the major support strategy for neonatal arterial switch operations (transposition of the great arteries and intact ventricular septum). METHODS: Seventy-four patients and "best-friend" control subjects were assessed 109 months (range, 48-166 months) postoperatively with general medical and neurologic evaluation, IQ testing, formal movement scores, and detailed parent-teacher behavioral-social reports. Fetal, neonatal, and perioperative data were collated. RESULTS: The prevalence of perioperative seizures was 6.8% (4/5 cases occurring preoperatively). The incidence of all perioperative neurologic abnormalities was 20%. Patients who had a neurologic event were (as a group) older at the time of operation and had a lower arterial blood pH before the operation. Selected perioperative factors (not related directly to cardiopulmonary bypass variables) predicted early (before discharge) neurologic outcome in a multivariate model. At late assessment, patients were more likely than control subjects to have a mild neurologic abnormality (P = 0.002). Full-scale IQ scores (Wechsler Preschool and Primary Scale of Intelligence and Wechsler Intelligence Scale for Children-Third Edition) were higher in control subjects (101.9 [SD = 13] vs 108.6 [SD = 12], P =.0007), with both groups having scores greater than the population-based test means. Full-scale IQ scores related most significantly to years of paternal education (beta = 1.51, P =.0078) but were also influenced by perioperative neurologic abnormalities, birth weight, and circulatory arrest time. Patients had higher motor impairment scores (Movement Assessment Battery) than control subjects (P =.0004). Parents (Achenbach Child Development Checklist) assigned higher total social-behavioral competence scores to control subjects (P =.05). Teachers (Achenbach Teacher Report Form) suggested that patients were more likely to be perceived as having various speech and expressive language problems, as well as minor behavioral problems. CONCLUSION: With the perioperative strategies used, not all survivors can be considered (neurodevelopmentally) normal at late follow-up, although the risk of important impairment is low. Perioperative events might have long-term prognostic value. On the basis of this study and published data regarding other strategies, continued application of full-flow cardiopulmonary bypass is justified, with the proviso that further investigation is required.