ArticleAmerican journal of medical genetics. Part A2020
Hearing loss in individuals with osteogenesis imperfecta in North America: Results from a multicenter study.
Article in American journal of medical genetics. Part A, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 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
21 citing papers in PubMed, 1 synthesis or guideline pooled it, 33 citations in OpenAlex.
- Treatments for hearing loss in osteogenesis imperfecta: a systematic review and meta-analysis on their efficacy.Scientific reports · 2022Pooled it
- The Effect of Bisphosphonate Therapy on Hearing and Middle Ear Dysfunction in Pediatric Osteogenesis Imperfecta.Journal of the American Academy of Audiology · 2026Article
- The natural history of osteogenesis imperfecta: a systematic review.Bone reports · 2026Review
- Rare disease care pathways profile in France: leveraging hospital big data to characterize natural history in osteogenesis imperfecta.JBMR plus · 2026Article
- Type III Osteogenesis Imperfecta andHuman mutation · 2026Article
- Hearing loss in Chinese osteogenesis imperfecta patients.European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery · 2025Article
- Frequency of Audiology Testing Among Individuals with Osteogenesis Imperfecta and Suggestions for Improving Audiogram Participation.Journal of the American Academy of Audiology · 2025Observational
- Hearing impairment amongst people with Osteogenesis Imperfecta in Germany.European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery · 2025Article
- Osteogenesis Imperfecta: Skeletal and Non-skeletal Challenges in Adulthood.Calcified tissue international · 2024Review
- The PATCH study: Prevalence of Hearing Loss During Ageing and Treatment Choices in Osteogenesis Imperfecta: A Danish Nationwide Register-Based Cohort Study.Calcified tissue international · 2024Article
- Genetic Evaluation for Monogenic Disorders of Low Bone Mass and Increased Bone Fragility: What Clinicians Need to Know.Current osteoporosis reports · 2024Review
- Emerging Landscape of Osteogenesis Imperfecta Pathogenesis and Therapeutic Approaches.ACS pharmacology & translational science · 2024Review
- Transition from Pediatric to Adult Health Care in Osteogenesis Imperfecta.Current osteoporosis reports · 2023Review
- The patient clinical journey and socioeconomic impact of osteogenesis imperfecta: a systematic scoping review.Orphanet journal of rare diseases · 2023Article
- Musculoskeletal and Gait Characteristics in Patients with Stickler Syndrome: A Cross-Sectional Study.Children (Basel, Switzerland) · 2022Article
- Alterations of a serum marker of collagen X in growing children with osteogenesis imperfecta.Bone · 2021Article
- Pregnancy in women with osteogenesis imperfecta: pregnancy characteristics, maternal, and neonatal outcomes.American journal of obstetrics & gynecology MFM · 2021Article
- The Anesthetic Management of a Parturient With Osteogenesis Imperfecta Type I Undergoing Cesarean Delivery.Cureus · 2021Article
- Best practice guidelines in managing the craniofacial aspects of skeletal dysplasia.Orphanet journal of rare diseases · 2021Article
- Positive airway pressure therapy for obstructive sleep apnea in patients with Osteogenesis imperfecta: a prospective pilot study.BMC musculoskeletal disorders · 2021Observational
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
23 authors at 9 institutions in 2 countries.
Funding
Abstract
Hearing loss (HL) is an extra-skeletal manifestation of the connective tissue disorder osteogenesis imperfecta (OI). Systematic evaluation of the prevalence and characteristics of HL in COL1A1/COL1A2-related OI will contribute to a better clinical management of individuals with OI. We collected and analyzed pure-tone audiometry data from 312 individuals with OI who were enrolled in the Linked Clinical Research Centers and the Brittle Bone Disorders Consortium. The prevalence, type, and severity of HL in COL1A1/COL1A2-related OI are reported. We show that the prevalence of HL in OI is 28% and increased with age in Type I OI but not in Types III and IV. Individuals with OI Types III and IV are at a higher risk to develop HL in the first decade of life when compared to OI Type I. We also show that the prevalence of SNHL is higher in females with OI compared to males. This study reveals new insights regarding prevalence of HL in OI including a lower general prevalence of HL in COL1A1/COL1A2-related OI than previously reported (28.3 vs. 65%) and high prevalence of SNHL in females. Our data support the need in early routine hearing evaluation in all types of OI that can be adjusted to the severity of the skeletal disease.
Indexed as
Identifiers
What OpenQuestion holds
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.