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BMJ Open. 2023 Nov 22;13(11):e078164. doi: 10.1136/bmjopen-2023-078164.
2
The correlation between transient osteoporosis of the hip and pregnancy: A review.髋关节一过性骨质疏松症与妊娠的相关性:综述。
Medicine (Baltimore). 2023 Oct 13;102(41):e35475. doi: 10.1097/MD.0000000000035475.
3
Mice heterozygous for an osteogenesis imperfecta-linked MBTPS2 variant display a compromised subchondral osteocyte lacunocanalicular network associated with abnormal articular cartilage.杂合子突变型成骨不全相关 MBTPS2 变体的小鼠表现出骨下骨细胞腔隙管网络受损,与关节软骨异常有关。
Bone. 2023 Dec;177:116927. doi: 10.1016/j.bone.2023.116927. Epub 2023 Oct 4.
4
Osteogenesis Imperfecta Is More Than a Pediatric Disorder-There Is a Need for Adult Care Evidence to Guide Clinicians.成骨不全症不止是一种儿科疾病——需要成人护理证据来指导临床医生。
J Clin Endocrinol Metab. 2024 Jan 18;109(2):e866-e867. doi: 10.1210/clinem/dgad423.
5
The International Standard Set of Outcome Measures for the Assessment of Hearing in People with Osteogenesis Imperfecta.《成骨不全症患者听力评估的国际通用结局指标集》。
Otol Neurotol. 2023 Aug 1;44(7):e449-e455. doi: 10.1097/MAO.0000000000003921. Epub 2023 Jun 16.
6
Systematic review of health related-quality of life in adults with osteogenesis imperfecta.成骨不全症成人健康相关生活质量的系统评价。
Orphanet J Rare Dis. 2023 Feb 22;18(1):36. doi: 10.1186/s13023-023-02643-3.
7
The patient clinical journey and socioeconomic impact of osteogenesis imperfecta: a systematic scoping review.成骨不全症患者的临床病程和社会经济影响:系统范围界定综述。
Orphanet J Rare Dis. 2023 Feb 22;18(1):34. doi: 10.1186/s13023-023-02627-3.
8
Key4OI Recommendations for Lung Function Guidance in Osteogenesis Imperfecta: Based on an Internationally Performed Comprehensive International Consortium for Health Outcomes Measurement Procedure.Key4OI 关于成骨不全症肺功能指导的建议:基于一项国际执行的全面国际健康结果测量程序联盟。
Chest. 2023 May;163(5):1201-1213. doi: 10.1016/j.chest.2022.12.047. Epub 2023 Jan 12.
9
Distinct type I collagen alterations cause intrinsic lung and respiratory defects of variable severity in mouse models of osteogenesis imperfecta.不同类型 I 型胶原的改变导致成骨不全症小鼠模型中肺和呼吸系统缺陷的严重程度不同。
J Physiol. 2023 Jan;601(2):355-379. doi: 10.1113/JP283452. Epub 2022 Nov 9.
10
Accelerated Aging in Cyclophilin B-Deficient Mice Downstream of p21-Cip1/Waf1.亲环蛋白B缺陷小鼠中p21-Cip1/Waf1下游的加速衰老
JBMR Plus. 2022 Sep 9;6(10):e10674. doi: 10.1002/jbm4.10674. eCollection 2022 Oct.

成骨不全症:成年期的骨骼和非骨骼挑战。

Osteogenesis Imperfecta: Skeletal and Non-skeletal Challenges in Adulthood.

机构信息

Department of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark.

Centre for Rare Diseases, Pediatric and Adolescent Medicine, Aarhus University Hospital, Aarhus, Denmark.

出版信息

Calcif Tissue Int. 2024 Dec;115(6):863-872. doi: 10.1007/s00223-024-01236-x. Epub 2024 Jun 5.

DOI:10.1007/s00223-024-01236-x
PMID:38836890
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11606788/
Abstract

Osteogenesis imperfecta (OI) is a Mendelian connective tissue disorder associated with increased bone fragility and other clinical manifestations most commonly due to abnormalities in production, structure, or post-translational modification of type I collagen. Until recently, most research in OI has focused on the pediatric population and much less attention has been directed at the effects of OI in the adult population. This is a narrative review of the literature focusing on the skeletal as well as non-skeletal manifestations in adults with OI that may affect the aging individual. We found evidence to suggest that OI is a systemic disease which involves not only the skeleton, but also the cardiopulmonary and gastrointestinal system, soft tissues, tendons, muscle, and joints, hearing, eyesight, dental health, and women's health in OI and potentially adds negative affect to health-related quality of life. We aim to guide clinicians as well as draw attention to obvious knowledge gaps and the need for further research in adult OI.

摘要

成骨不全症(OI)是一种孟德尔结缔组织疾病,与骨脆性增加和其他临床表现有关,这些临床表现通常是由于 I 型胶原的产生、结构或翻译后修饰异常所致。直到最近,OI 的大多数研究都集中在儿科人群,而对成年人群中 OI 的影响关注较少。这是一篇对文献的叙述性综述,重点介绍了可能影响衰老个体的 OI 成年患者的骨骼和非骨骼表现。我们有证据表明,OI 是一种全身性疾病,不仅涉及骨骼,还涉及心肺和胃肠道系统、软组织、肌腱、肌肉和关节、听力、视力、牙齿健康和女性健康,并且可能对健康相关生活质量产生负面影响。我们的目标是为临床医生提供指导,并引起对 OI 成年患者明显知识空白和进一步研究需求的关注。