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先天性纯红细胞再生障碍性贫血综合征的诊断、治疗及监测:国际共识声明

Diagnosis, treatment, and surveillance of Diamond-Blackfan anaemia syndrome: international consensus statement.

作者信息

Wlodarski Marcin W, Vlachos Adrianna, Farrar Jason E, Da Costa Lydie M, Kattamis Antonis, Dianzani Irma, Belendez Cristina, Unal Sule, Tamary Hannah, Pasauliene Ramune, Pospisilova Dagmar, de la Fuente Josu, Iskander Deena, Wolfe Lawrence, Liu Johnson M, Shimamura Akiko, Albrecht Katarzyna, Lausen Birgitte, Bechensteen Anne Grete, Tedgard Ulf, Puzik Alexander, Quarello Paola, Ramenghi Ugo, Bartels Marije, Hengartner Heinz, Farah Roula A, Al Saleh Mahasen, Hamidieh Amir Ali, Yang Wan, Ito Etsuro, Kook Hoon, Ovsyannikova Galina, Kager Leo, Gleizes Pierre-Emmanuel, Dalle Jean-Hugues, Strahm Brigitte, Niemeyer Charlotte M, Lipton Jeffrey M, Leblanc Thierry M

机构信息

Department of Hematology, St Jude Children's Research Hospital, Memphis, TN, USA; Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, Medical Center, Faculty of Medicine, University of Freiburg, Freiburg, Germany.

Cohen Children's Medical Center, Hematology/Oncology and Stem Cell Transplantation, Hew Hyde Park, NY, USA; Feinstein Institutes for Medical Research, Manhasset, NY, USA; Zucker School of Medicine at Hofstra/Northwell, Hempstead, NY, USA.

出版信息

Lancet Haematol. 2024 May;11(5):e368-e382. doi: 10.1016/S2352-3026(24)00063-2.

Abstract

Diamond-Blackfan anaemia (DBA), first described over 80 years ago, is a congenital disorder of erythropoiesis with a predilection for birth defects and cancer. Despite scientific advances, this chronic, debilitating, and life-limiting disorder continues to cause a substantial physical, psychological, and financial toll on patients and their families. The highly complex medical needs of affected patients require specialised expertise and multidisciplinary care. However, gaps remain in effectively bridging scientific discoveries to clinical practice and disseminating the latest knowledge and best practices to providers. Following the publication of the first international consensus in 2008, advances in our understanding of the genetics, natural history, and clinical management of DBA have strongly supported the need for new consensus recommendations. In 2014 in Freiburg, Germany, a panel of 53 experts including clinicians, diagnosticians, and researchers from 27 countries convened. With support from patient advocates, the panel met repeatedly over subsequent years, engaging in ongoing discussions. These meetings led to the development of new consensus recommendations in 2024, replacing the previous guidelines. To account for the diverse phenotypes including presentation without anaemia, the panel agreed to adopt the term DBA syndrome. We propose new simplified diagnostic criteria, describe the genetics of DBA syndrome and its phenocopies, and introduce major changes in therapeutic standards. These changes include lowering the prednisone maintenance dose to maximum 0·3 mg/kg per day, raising the pre-transfusion haemoglobin to 9-10 g/dL independent of age, recommending early aggressive chelation, broadening indications for haematopoietic stem-cell transplantation, and recommending systematic clinical surveillance including early colorectal cancer screening. In summary, the current practice guidelines standardise the diagnostics, treatment, and long-term surveillance of patients with DBA syndrome of all ages worldwide.

摘要

钻石黑范贫血(DBA)于80多年前首次被描述,是一种先天性红细胞生成障碍疾病,易引发出生缺陷和癌症。尽管科学不断进步,但这种慢性、使人衰弱且危及生命的疾病仍给患者及其家庭带来巨大的身体、心理和经济负担。受影响患者极其复杂的医疗需求需要专业知识和多学科护理。然而,在将科学发现有效应用于临床实践以及向医疗服务提供者传播最新知识和最佳实践方面仍存在差距。2008年首个国际共识发布后,我们对DBA的遗传学、自然史和临床管理的认识取得了进展,有力地支持了制定新的共识建议的必要性。2014年在德国弗莱堡,来自27个国家的包括临床医生、诊断专家和研究人员在内的53名专家组成了一个小组。在患者权益倡导者的支持下,该小组在随后几年里多次开会,进行持续讨论。这些会议促成了2024年新的共识建议的制定,取代了先前的指南。为了涵盖包括无贫血表现在内的各种表型,该小组同意采用“DBA综合征”这一术语。我们提出了新的简化诊断标准,描述了DBA综合征及其表型模拟的遗传学,并介绍了治疗标准的重大变化。这些变化包括将泼尼松维持剂量降至每天最大0·3 mg/kg,将输血前血红蛋白提高到9 - 10 g/dL(与年龄无关),建议早期积极螯合治疗,扩大造血干细胞移植的适应证,并建议进行系统的临床监测,包括早期结直肠癌筛查。总之,当前的实践指南规范了全球所有年龄段DBA综合征患者的诊断、治疗和长期监测。

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