关键词: Diagnostic delay Meta-aggregation Meta-analysis Misdiagnosis Rare disease Sarcoidosis Systematic review

Mesh : Female Humans Delayed Diagnosis Disease Progression Qualitative Research Sarcoidosis / diagnosis Male

来  源:   DOI:10.1186/s13023-024-03152-7   PDF(Pubmed)

Abstract:
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.
CONCLUSIONS: 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.
BACKGROUND: PROSPERO Registration number: CRD42022307236.
摘要:
背景:结节病是一种原因不明的慢性炎症性肉芽肿性疾病。诊断延迟会导致疾病进展和患者预后较差。我们的目的是回顾目前的文献,以确定结节病的整体诊断延迟,与诊断延迟相关的因素,以及结节病患者诊断延迟的经验。
方法:三个数据库(PubMed/Medline,Scopus,和ProQuest)和灰色文献来源进行了搜索。随机效应反方差荟萃分析用于汇集所有类型结节病亚组分析中的平均诊断延迟。诊断延迟定义为从报告的症状发作到诊断结节病的时间。
结果:我们确定了374个标题,其中29项研究被纳入审查,总体样本为1531名(694名女性,837名男性)。所有类型结节病的总体平均诊断延迟为7.93个月(95%CI1.21至14.64个月)。在纳入的研究中,与诊断延迟相关的因素的荟萃汇总确定了三类:(1)结节病的复杂和罕见特征,(2)医疗保健因素和(3)以患者为中心的因素。病例研究中报告的结果的荟萃汇总显示,与诊断延迟相关的三个最常见的结果是:(1)不正确的诊断,(2)不正确的治疗和(3)并发症/疾病进展的发展。具有看门人卫生系统的国家(将消费者从初级保健临床医生转到专科护理)和具有非看门人系统的国家之间的诊断延迟没有显着差异。没有定性研究检查人们的诊断延迟的经验。
结论:结节病的平均诊断延迟近8个月,这对患者管理有客观影响。另一方面,关于结节病诊断延迟的经验以及与此相关的因素的证据很少。在寻求结节病诊断的同时了解人们的经历对于深入了解可能导致延迟的因素至关重要。随后通知策略,旨在提高临床医生和公众对这种罕见疾病的认识的工具和培训活动。
背景:PROSPERO注册号:CRD42022307236。
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