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《关于成骨不全症管理原则的第一份欧洲共识》。

The first European consensus on principles of management for achondroplasia.

机构信息

Centre of Reference for Constitutional Bone Diseases (MOC), Department of Clinical Genetics, Paris Centre University, INSERM UMR 1163, Imagine Institute, Paris Centre University, Paris, France.

Department of Medical Genetics, King Faisal Specialist Hospital and Research Center, Riyadh, Kingdom of Saudi Arabia.

出版信息

Orphanet J Rare Dis. 2021 Jul 31;16(1):333. doi: 10.1186/s13023-021-01971-6.

Abstract

Achondroplasia is the most common type of skeletal dysplasia, caused by a recurrent pathogenic variant in the fibroblast growth factor receptor 3 (FGFR3). The management of achondroplasia is multifaceted, requiring the involvement of multiple specialties across the life course. There are significant unmet needs associated with achondroplasia and substantial differences in different countries with regard to delivery of care. To address these challenges the European Achondroplasia Forum (EAF), a network of senior clinicians and orthopaedic surgeons from Europe and the Middle East representative of the achondroplasia clinical community, came together with the overall aim of improving patient outcomes. The EAF developed a consensus on guiding principles of management of achondroplasia to provide a basis for developing optimal care in Europe. All members of the EAF were invited to submit suggestions for guiding principles of management, which were consolidated and then discussed during a meeting in December 2020. The group voted anonymously on the inclusion of each principle, with the requirement of a 75% majority at the first vote to pass the principle. A vote on the level of agreement was then held. A total of six guiding principles were developed, which cover management over the lifetime of a person with achondroplasia. The principles centre on the lifelong management of achondroplasia by an experienced multidisciplinary team to anticipate and manage complications, support independence, and improve quality of life. There is focus on timely referral to a physician experienced in the management of achondroplasia on suspicion of the condition, shared decision making, the goals of management, access to adaptive measures to enable those with achondroplasia to access their environment, and the importance of ongoing monitoring throughout adolescence and adulthood. All principles achieved the 75% majority required for acceptance at the first vote (range 91-100%) and a high level of agreement (range 8.5-9.6). The guiding principles of management for achondroplasia provide all healthcare professionals, patient advocacy groups and policy makers involved in the management of achondroplasia with overarching considerations when developing health systems to support the management of achondroplasia.

摘要

软骨发育不全是最常见的骨骼发育不良类型,由成纤维细胞生长因子受体 3(FGFR3)中的重复致病性变异引起。软骨发育不全的管理是多方面的,需要在整个生命周期中涉及多个专业。软骨发育不全存在着显著的未满足需求,并且不同国家在提供护理方面存在着巨大差异。为了解决这些挑战,欧洲软骨发育不全论坛(EAF)汇集了来自欧洲和中东的资深临床医生和矫形外科医生,他们代表了软骨发育不全的临床社区,旨在改善患者的治疗效果。EAF 就软骨发育不全的管理指导原则达成了共识,为在欧洲制定最佳护理方案提供了基础。EAF 的所有成员都被邀请提出管理指导原则的建议,这些建议经过整合,并在 2020 年 12 月的一次会议上进行了讨论。该组织以匿名方式对每项原则的纳入进行投票,第一次投票需要 75%的多数票通过该原则。然后对投票结果的一致性进行了表决。总共制定了六项指导原则,涵盖了软骨发育不全患者一生的管理。这些原则围绕着由经验丰富的多学科团队对软骨发育不全进行终生管理,以预测和管理并发症、支持独立性和提高生活质量。重点是及时向有管理软骨发育不全经验的医生转介,以怀疑该疾病,共同决策,管理目标,获得适应性措施,使患有软骨发育不全的人能够进入环境,并在整个青春期和成年期进行持续监测。所有原则都在第一次投票中获得了 75%的多数票(范围为 91-100%)和高度一致的投票结果(范围为 8.5-9.6)。软骨发育不全的管理指导原则为参与软骨发育不全管理的所有医疗保健专业人员、患者权益团体和政策制定者提供了在制定支持软骨发育不全管理的卫生系统时的总体考虑因素。

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本文引用的文献

1
Achondroplasia is associated with increased occurrence of apparent life-threatening events.
Acta Paediatr. 2021 Jun;110(6):1842-1846. doi: 10.1111/apa.15760. Epub 2021 Feb 12.
2
Quality of life in adults with achondroplasia in the United States.
Am J Med Genet A. 2021 Mar;185(3):695-701. doi: 10.1002/ajmg.a.62018. Epub 2020 Dec 24.
3
Assessing physical symptoms, daily functioning, and well-being in children with achondroplasia.
Am J Med Genet A. 2021 Jan;185(1):33-45. doi: 10.1002/ajmg.a.61903. Epub 2020 Oct 20.
4
Achondroplasia Foramen Magnum Score: screening infants for stenosis.
Arch Dis Child. 2021 Feb;106(2):180-184. doi: 10.1136/archdischild-2020-319625. Epub 2020 Sep 3.
5
Birth prevalence of achondroplasia: A systematic literature review and meta-analysis.
Am J Med Genet A. 2020 Oct;182(10):2297-2316. doi: 10.1002/ajmg.a.61787. Epub 2020 Aug 17.
6
Health Supervision for People With Achondroplasia.
Pediatrics. 2020 Jun;145(6). doi: 10.1542/peds.2020-1010.
7
High prevalence of symptomatic spinal stenosis in Norwegian adults with achondroplasia: a population-based study.
Orphanet J Rare Dis. 2020 May 25;15(1):123. doi: 10.1186/s13023-020-01397-6.
8
New perspectives on the treatment of skeletal dysplasia.
Ther Adv Endocrinol Metab. 2020 Mar 3;11:2042018820904016. doi: 10.1177/2042018820904016. eCollection 2020.
9
Obesity in achondroplasia patients: from evidence to medical monitoring.
Orphanet J Rare Dis. 2019 Nov 14;14(1):253. doi: 10.1186/s13023-019-1247-6.
10
Quality of life of children with achondroplasia and their parents - a German cross-sectional study.
Orphanet J Rare Dis. 2019 Aug 9;14(1):194. doi: 10.1186/s13023-019-1171-9.

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