Department of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark.
Centre for Rare Diseases, Pediatric and Adolescent Medicine, Aarhus University Hospital, Aarhus, Denmark.
Calcif Tissue Int. 2024 Dec;115(6):863-872. doi: 10.1007/s00223-024-01236-x. Epub 2024 Jun 5.
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.
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。