ReviewAmerican journal of medical genetics. Part C, Seminars in medical genetics2016
Pharmacological and biological therapeutic strategies for osteogenesis imperfecta.
Review in American journal of medical genetics. Part C, Seminars in medical genetics, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 39 papers, 1 of them a synthesis that pooled it.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
39 citing papers in PubMed, 1 synthesis or guideline pooled it, 63 citations in OpenAlex.
- Do Bisphosphonates Alleviate Pain in Children? A Systematic Review.Current osteoporosis reports · 2020Pooled it
- Clinical diagnosis and challenges in management of Osteogenesis Imperfecta in a resource-limited setting - A case report and review of literature.International journal of surgery case reports · 2025Article
- Effect of recombinant human parathyroid hormone and zoledronic acid on osteoblast gene expression using multifaceted approach: An in vitro study.Indian journal of pharmacology · 2025Article
- Schnurri-3 inhibition rescues skeletal fragility and vascular skeletal stem cell niche pathology in the OIM model of osteogenesis imperfecta.Bone research · 2024Article
- Exosome miR-4738-3p-mediated regulation ofBiomolecules & biomedicine · 2024Article
- Revolutionizing bone defect healing: the power of mesenchymal stem cells as seeds.Frontiers in bioengineering and biotechnology · 2024Review
- The vitamin D status in a Chinese osteogenesis imperfecta population and its correlation with bone metabolic markers and bone density.Frontiers in nutrition · 2024Article
- "Osteogenesis Imperfecta Patients Wish Orthopedic Surgeons Had Better Strategies to Help with…"-Results of a Patient and Parent-Oriented Survey.Children (Basel, Switzerland) · 2023Article
- Vitamin D and Bone fragility in Individuals with Osteogenesis Imperfecta: A Scoping Review.International journal of molecular sciences · 2023Article
- Targeting strategies for bone diseases: signaling pathways and clinical studies.Signal transduction and targeted therapy · 2023Review
- Vitamin D and Osteogenesis Imperfecta in Pediatrics.Pharmaceuticals (Basel, Switzerland) · 2023Review
- Article
- Expanding the genetic and clinical spectrum of osteogenesis imperfecta: identification of novel rare pathogenic variants in type I collagen-encoding genes.Frontiers in endocrinology · 2023Article
- A multicenter study to evaluate pain characteristics in osteogenesis imperfecta.American journal of medical genetics. Part A · 2023Observational
- Targeting TGF-β for treatment of osteogenesis imperfecta.The Journal of clinical investigation · 2022Article
- Skeletal outcomes of patients with osteogenesis imperfecta during drug holiday of bisphosphonates: a real-world study.Frontiers in endocrinology · 2022Article
- Article
- COPB2 loss of function causes a coatopathy with osteoporosis and developmental delay.American journal of human genetics · 2021Article
- Current Overview of Osteogenesis Imperfecta.Medicina (Kaunas, Lithuania) · 2021Review
- Impact of Intrinsic Muscle Weakness on Muscle-Bone Crosstalk in Osteogenesis Imperfecta.International journal of molecular sciences · 2021Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
Osteogenesis imperfecta (OI) is a connective tissue disorder characterized by bone fragility, low bone mass, and bone deformities. The majority of cases are caused by autosomal dominant pathogenic variants in the COL1A1 and COL1A2 genes that encode type I collagen, the major component of the bone matrix. The remaining cases are caused by autosomal recessively or dominantly inherited mutations in genes that are involved in the post-translational modification of type I collagen, act as type I collagen chaperones, or are members of the signaling pathways that regulate bone homeostasis. The main goals of treatment in OI are to decrease fracture incidence, relieve bone pain, and promote mobility and growth. This requires a multi-disciplinary approach, utilizing pharmacological interventions, physical therapy, orthopedic surgery, and monitoring nutrition with appropriate calcium and vitamin D supplementation. Bisphosphonate therapy, which has become the mainstay of treatment in OI, has proven beneficial in increasing bone mass, and to some extent reducing fracture risk. However, the response to treatment is not as robust as is seen in osteoporosis, and it seems less effective in certain types of OI, and in adult OI patients as compared to most pediatric cases. New pharmacological treatments are currently being developed, including anti-resorptive agents, anabolic treatment, and gene- and cell-therapy approaches. These therapies are under different stages of investigation from the bench-side, to pre-clinical and clinical trials. In this review, we will summarize the recent findings regarding the pharmacological and biological strategies for the treatment of patients with OI. © 2016 Wiley Periodicals, Inc.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.