Capturing what matters: Patient-reported LGI1-ANTibody encephalitis outcome RatiNg scale (LANTERN)

IF 4.4 2区医学 Q1 CLINICAL NEUROLOGY

Annals of Clinical and Translational Neurology Pub Date : 2025-02-25 DOI:10.1002/acn3.70006

Mark J Kelly, Barbara Wagner, Bryan Ceronie, Christine Strippel, Ann Yee Lin, Adam Handel, John Soltys, Sophie Binks, Philip A Powell, Sarosh R Irani

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引用次数: 0

Abstract

Background

LGI1-antibody encephalitis (LGI1-Ab-E) is a common form of autoimmune encephalitis where most patients demonstrate ‘good’ clinician-rated outcomes. However, more targeted questionnaires reveal numerous debilitating symptoms for many years. To better quantify these persistent features, we designed the LGI1-Antibody Encephalitis Rating (LANTERN) scale, a quantified, disease-specific patient-reported outcome measure (PROM), adhering to FDA guidelines.

Methods

A participant-driven mixed-methods approach to develop a clinically valid questionnaire over three stages: (1) Item generation through semi-structured interviews; (2) Repeated cognitive debriefing rounds to advance comprehensibility, relevance and comprehensiveness; (3) Psychometric survey to condense the most sensitive and valid questions. Analyses incorporated sensitivity testing with multiple internal and external validations.

Results

From 73 items across six domains (Stage 1; n = 18), a questionnaire assessing the frequency and severity of 43 symptoms (80 questions), plus nine activities of daily living (ADL), was developed through cognitive debriefing (Stage 2; n = 15). This 89-question survey was completed (Stage 3; n = 66 patients and 32 relatives) and distilled, using exploratory factor analyses, to a three-factor symptom-burden questionnaire comprising 41 questions (19 symptoms and 6 ADL), separated into physical, cognitive/behavioural and ADL domains. These factors demonstrated strong internal reliability (Cronbach alpha: 0.85–0.91), correlations with relative-completed questionnaires (R = 0.73–0.85; p < 0.001), good-to-excellent intraclass re-testing correlations (0.81–0.98; n = 19) and strong associations with numerous predefined external measures.

Discussion

LANTERN represents a PROM for LGI1-Ab-E, with initial content, structural and construct validity and test–retest reliability. It can be used as a reliable, tailored, efficient and sensitive method to establish symptom burden in people with LGI1-Ab-E, both in clinical practice and trials.

Abstract Image

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捕捉重要信息：患者报告的 LGI1-ANTibody 脑炎结果比值表（LANTERN）。

背景：lgi1抗体脑炎（LGI1-Ab-E）是一种常见的自身免疫性脑炎，大多数患者表现出“良好”的临床评价结果。然而，更有针对性的调查问卷揭示了许多年来的衰弱症状。为了更好地量化这些持续性特征，我们设计了lgi1抗体脑炎分级（LANTERN）量表，这是一种量化的疾病特异性患者报告的结果测量（PROM），遵循FDA指南。方法：采用参与者驱动的混合方法，分三个阶段编制临床有效的问卷：(1)通过半结构化访谈生成项目；(2)反复进行认知汇报，提高可理解性、相关性和全面性；(3)心理测量调查，浓缩最敏感、最有效的问题。分析结合了多个内部和外部验证的灵敏度测试。结果：从6个领域的73个项目(阶段1；n = 18)，通过认知述评(第二阶段；n = 15)。这个89个问题的调查已经完成(第三阶段；n = 66名患者和32名亲属)，并通过探索性因素分析，提炼出一份包括41个问题（19个症状和6个ADL）的三因素症状负担问卷，分为身体、认知/行为和ADL领域。这些因素具有较强的内部信度（Cronbach alpha: 0.85-0.91），与相对完成问卷的相关性(R = 0.73-0.85；p讨论：LANTERN代表LGI1-Ab-E的PROM，具有初始内容、结构和构造效度以及重测信度。无论是在临床实践还是试验中，它都可以作为一种可靠、量身定制、高效和敏感的方法来确定LGI1-Ab-E患者的症状负担。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Annals of Clinical and Translational Neurology Medicine-Neurology (clinical)

CiteScore

9.10

自引率

1.90%

发文量

218

审稿时长

8 weeks

期刊介绍： Annals of Clinical and Translational Neurology is a peer-reviewed journal for rapid dissemination of high-quality research related to all areas of neurology. The journal publishes original research and scholarly reviews focused on the mechanisms and treatments of diseases of the nervous system; high-impact topics in neurologic education; and other topics of interest to the clinical neuroscience community.