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探索 B:对有慢性症状的急性肝性卟啉症患者进行的前瞻性、长期自然病史研究。

EXPLORE B: A prospective, long-term natural history study of patients with acute hepatic porphyria with chronic symptoms.

机构信息

Department of Gastroenterology-Hepatology and Center for Metabolic Diseases, University Hospital Leuven, Leuven, Belgium.

Department of Medicine, University Hospital of Helsinki, Helsinki, Finland.

出版信息

J Inherit Metab Dis. 2022 Nov;45(6):1163-1174. doi: 10.1002/jimd.12551. Epub 2022 Sep 18.

DOI:10.1002/jimd.12551
PMID:36069414
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9825970/
Abstract

One-year data from EXPLORE Part A showed high disease burden and impaired quality of life (QOL) in patients with acute hepatic porphyria (AHP) with recurrent attacks. We report baseline data of patients who enrolled in EXPLORE Part B for up to an additional 3 years of follow-up. EXPLORE B is a long-term, prospective study evaluating disease activity, pain intensity, and QOL in patients with AHP with ≥1 attack in the 12 months before enrollment or receiving hemin or gonadotropin-releasing hormone prophylaxis. Data were evaluated in patients with more (≥3 attacks or on prophylaxis treatment) or fewer (<3 attacks and no prophylaxis treatment) attacks. Patients in the total population (N = 136), and more (n = 110) and fewer (n = 26) attack subgroups, reported a median (range) of 3 (0-52), 4 (0-52), and 1 (0-2) acute attacks, respectively, in the 12 months prior to the baseline visit. Pain, mood/sleep, digestive/bladder, and nervous system symptoms were each experienced by ≥80% of patients; most received hemin during attacks. Almost three-quarters of patients reported chronic symptoms between attacks, including 85% of patients with fewer attacks. Pain intensity was comparable among both attack subgroups; most patients required pain medication. All groups had diminished QOL on the EuroQol visual analog scale and the European Organisation for Research and Treatment of Cancer Quality-of-life Questionnaire Core 30 versus population norms. Patients with AHP with recurrent attacks, even those having fewer attacks, experience a high disease burden, as evidenced by chronic symptoms between attacks and impaired QOL.

摘要

EXPLORE 研究 A 部分的一年数据显示,反复发作急性肝性卟啉症(AHP)患者疾病负担重,生活质量(QOL)受损。我们报告了参与 EXPLORE 研究 B 的患者的基线数据,这些患者的随访时间最长可达 3 年。EXPLORE B 是一项长期前瞻性研究,评估了 12 个月内有≥1 次发作或接受过血红素或促性腺激素释放激素预防治疗的 AHP 患者的疾病活动度、疼痛强度和 QOL。根据发作次数(≥3 次发作或接受预防治疗)或发作次数(<3 次发作且无预防治疗)对患者进行了评估。总人群(N=136)、发作次数较多(n=110)和较少(n=26)的患者亚组在基线访视前的 12 个月内,分别报告了中位数(范围)为 3(0-52)、4(0-52)和 1(0-2)次急性发作。≥80%的患者经历过疼痛、情绪/睡眠、消化/膀胱和神经系统症状;大多数患者在发作期间接受了血红素治疗。几乎 3/4 的患者在发作之间报告有慢性症状,其中发作次数较少的患者占 85%。发作次数较少和较多的患者疼痛强度相似;大多数患者需要使用止痛药。所有患者的欧洲五维健康量表和欧洲癌症研究与治疗组织生活质量问卷核心 30 评分均低于人群正常值,表明 QOL 受损。反复发作 AHP 的患者,即使发作次数较少,也会经历较高的疾病负担,这表现在发作之间存在慢性症状和 QOL 受损。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/d5a5ad2eb7ff/JIMD-45-1163-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/4dea8cdc9362/JIMD-45-1163-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/4300b7559188/JIMD-45-1163-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/48da3e8cbb4b/JIMD-45-1163-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/d5a5ad2eb7ff/JIMD-45-1163-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/4dea8cdc9362/JIMD-45-1163-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/4300b7559188/JIMD-45-1163-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/48da3e8cbb4b/JIMD-45-1163-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9737/9825970/d5a5ad2eb7ff/JIMD-45-1163-g001.jpg

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