Evidence map›Paper›PMID 24754836›Full record

ArticleClinical genetics2015

A cross-sectional multicenter study of osteogenesis imperfecta in North America - results from the linked clinical research centers.

R M Patel, S C S Nagamani, D Cuthbertson, P M Campeau, J P Krischer, J R Shapiro, R D Steiner, P A Smith, M B Bober, P H Byers and 7 more

Registry-linked trialOpen access · greenAbstract readMulticenter Study
In one paragraph

Article in Clinical genetics, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT04152551 (Effects of Bisphosphonates on OI-Related Hearing Loss), which is not on this map. Cited by 52 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
52citing papers in PubMed, 2 pooled it
3.3field-weighted citation impact, top 8% of its field
1 · What the graph read from 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.

2 · The registry

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.

NCT04152551 phase4recruitingnot on this mapstarted 2019, after this paper: background citation

Effects of Bisphosphonates on OI-Related Hearing Loss: A Pilot Study

TypeinterventionalSponsorHospital for Special Surgery, New YorkRan2019 to 2027Enrolled100ConditionsOsteogenesis ImperfectaArmsRisedronate Oral Tablet
3 · Its place in the literature

Who cites it

52 citing papers in PubMed, 2 syntheses or guidelines pooled it, 80 citations in OpenAlex.

  1. Pooled it
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  11. 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 · 2025
    Article
  12. Observational
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

17 authors at 10 institutions in 2 countries.

R M PatelDepartment of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
S C S Nagamani
D Cuthbertson
P M Campeau
J P Krischer
J R Shapiro
R D Steiner
P A Smith
M B Bober
P H Byers
M Pepin
M Durigova
F H Glorieux
F Rauch
B H Lee
T Hart
V R Sutton
Baylor College of Medicine · USMcGill University · CAUniversity of South Florida · USUniversity of Washington Medical Center · USAlfred I. duPont Hospital for Children · USHoward Hughes Medical Institute · USKennedy Krieger Institute · USMarshfield Clinic · USOsteogenesis Imperfecta Foundation · USShriners Hospitals for Children - Chicago · US

Funding

UNDERSTANDING FXTAS AMONG MALES WITH THE FMR1 PREMUTATIONP30HD024064 · NICHD · BAYLOR COLLEGE OF MEDICINE · PI TOLIAS, KIMBERLY R · 1988 to 2013
$29.4M
Project 3: Collagen post-translational modification and cross-linking in OIP01HD070394 · NICHD · BAYLOR COLLEGE OF MEDICINE · PI LEE, BRENDAN · 2011 to 2020
$13.4M
Doris Duke Charitable Foundation 2013095NICHD NIH HHS HD024064NICHD NIH HHS HD070394NICHD NIH HHS P01 HD070394NICHD NIH HHS P30 HD024064
6 · The paper itself

Abstract

Osteogenesis imperfecta (OI) is the most common skeletal dysplasia that predisposes to recurrent fractures and bone deformities. In spite of significant advances in understanding the genetic basis of OI, there have been no large-scale natural history studies. To better understand the natural history and improve the care of patients, a network of Linked Clinical Research Centers (LCRC) was established. Subjects with OI were enrolled in a longitudinal study, and in this report, we present cross-sectional data on the largest cohort of OI subjects (n = 544). OI type III subjects had higher prevalence of dentinogenesis imperfecta, severe scoliosis, and long bone deformities as compared to those with OI types I and IV. Whereas the mean lumbar spine area bone mineral density (LS aBMD) was low across all OI subtypes, those with more severe forms had lower bone mass. Molecular testing may help predict the subtype in type I collagen-related OI. Analysis of such well-collected and unbiased data in OI can not only help answering questions that are relevant to patient care but also foster hypothesis-driven research, especially in the context of 'phenotypic expansion' driven by next-generation sequencing.

Indexed as

Bone DensityAdolescentAdultAgedChildChild, PreschoolCollagen Type ICollagen Type I, alpha 1 ChainCross-Sectional StudiesFemaleHumansInfantInfant, NewbornMaleMiddle AgedMutationCollagen Type ICollagen Type I, alpha 1 Chainbone mineral density in osteogenesis imperfectahistory studylongitudinal studynatural history studyosteogenesis imperfecta

Identifiers

PMID24754836
PMCPMC5529599
OpenAlexW1900428395

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

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.