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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.
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http://dx.doi.org/10.1007/s00223-024-01236-x | DOI Listing |
Calcif Tissue Int
September 2025
Department of Endocrinology, Post-Graduate Institute of Medical Education and Research (PGIMER), 001, Nehru Extension Block, Chandigarh, India.
Rare diseases, defined by the 2002 Rare Disease Act, affect fewer than 5 in 10,000 individuals. Rare metabolic bone diseases (MBDs), such as osteogenesis imperfecta, hypophosphatasia, osteopetrosis, and other unclassified disorders, can disrupt bone development and remodeling, posing diagnostic and management challenges. This study analyzed data from the rarembd.
View Article and Find Full Text PDFWorld J Methodol
December 2025
Department of Orthopaedics, All India Institute of Medical Sciences, Rishikesh 249203, Uttarākhand, India.
Skeletal dysplasia includes numerous genetic disorders marked by abnormal bone and cartilage growth, causing various spinal issues. The 2023 nosology identifies 771 distinct dysplasias involving 552 genes, with achondroplasia being the most common and significantly affecting the spine. Other disorders include type II collagenopathies, sulphation defects, Filamin B disorders, and osteogenesis imperfecta, presenting with short stature, limb deformities, joint contractures, and spinal abnormalities.
View Article and Find Full Text PDFPurpose: Osteogenesis imperfecta (OI) is commonly managed with bisphosphonates (BPs) which are associated with significant side effects. This study aimed to investigate intramedullary sclerosis as a potential side effect of prolonged BP use in paediatric patients.
Methods: Thirteen children with OI underwent surgery at our hospital.
Cleft Palate Craniofac J
September 2025
Department of Community and Preventive Dentistry, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil.
To compare the prevalence of malocclusion and the facial profile of children/adolescents with rare genetic diseases-mucopolysaccharidosis (MPS) and osteogenesis imperfecta (OI) with normotypical children/adolescents.Cross-sectional study.Hospital outpatient clinics in 5 Brazilian states (Ceará, Espírito Santo, Minas Gerais, Rio de Janeiro, and São Paulo).
View Article and Find Full Text PDFHum Genomics
August 2025
Department of Endocrinology, Genetics and Metabolism, Affiliated Hospital of Jining Medical University, 89 Guhuai Road, Jining, 272029, Shandong, PR China.
Backgroud: Osteogenesis imperfecta (OI) is a phenotypically and genetically heterogeneous group of inherited connective tissue disorder. This investigation aims to elucidate the molecular etiology underlying a four-generation Chinese family affected by OI.
Methods: Whole-exome sequencing was employed to identify pathogenic variants in the proband, with subsequent Sanger sequencing performed for familial co-segregation analysis.