Evidence map›Paper›PMID 41206390›Full record

Trial reportCalcified tissue international2025

Baseline Characteristics of the TOPaZ Study: Randomised Trial of Teriparatide and Zoledronic Acid Compared with Standard Care in Adults with Osteogenesis Imperfecta.

Jannie Dahl Hald, Christopher Weir, Catriona Keerie, Lorna Dewar, Morag MacLean, Lynsey Milne, Richard Keen, Jennifer Walsh, Kenneth Poole, Bente Langdahl and 33 more

Abstract readRandomized Controlled TrialMulticenter Study
PubMed Full text at PMC Publisher
In one paragraph

Trial report in Calcified tissue international, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

43 authors.

Jannie Dahl HaldDepartment of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark.
Christopher WeirEdinburgh Clinical Trials Unit, Usher Institute, University of Edinburgh, Edinburgh, UK.
Catriona KeerieEdinburgh Clinical Trials Unit, Usher Institute, University of Edinburgh, Edinburgh, UK.
Lorna DewarEdinburgh Clinical Trials Unit, Usher Institute, University of Edinburgh, Edinburgh, UK.
Morag MacLeanEdinburgh Clinical Trials Unit, Usher Institute, University of Edinburgh, Edinburgh, UK.
Lynsey MilneEdinburgh Clinical Trials Unit, Usher Institute, University of Edinburgh, Edinburgh, UK.
Richard KeenMetabolic Bone Clinic, Royal National Orthopaedic Hospital NHS Trust, London, UK.
Jennifer WalshMetabolic Bone Centre, University of Sheffield, Sheffield, UK.
Kenneth PooleCambridge University Hospitals NHS Trust, Addenbrookes Hospital, Cambridge, UK.
Bente LangdahlDepartment of Endocrinology and Internal Medicine, Aarhus University Hospital, Aarhus, Denmark.
John R LindsayBelfast Health and Social Care Trust, Royal Victoria Hospital, and Musgrave Park Hospital, Belfast, UK.
Nazim GhouriEndocrinology, Queen Elizabeth University Hospital, University of Glasgow, Glasgow, UK.
Rosemary J HollickAberdeen Centre for Arthritis and Musculoskeletal Health (Epidemiology), University of Aberdeen, Aberdeen, UK.
Terry AsprayBone Clinic- Freeman Hospital, University of Newcastle, Newcastle, UK.
Rachel K CrowleyDepartment of Endocrinology, Saint Vincent's University Hospital, Dublin, Ireland.
Martine Cohen-SolalDepartment of Rheumatology, Inserm U1132 Bioscar, F-75010, Paris, France.
Zaki Hassan-SmithAston Medical School, Aston University, Birmingham, UK.
Stephen TuckSouth Tees Hospitals NHS Foundation Trust, Rheumatology, Middlesborough, UK.
Elizabeth CurtisMRC Lifecourse Epidemiology Centre, University Hospital Southampton, Southampton, UK.
Nick HarveyMRC Lifecourse Epidemiology Centre, University Hospital Southampton, Southampton, UK.
E Marelise W EekhoffCenter for Rare Bone Diseases, Department of Endocrinology, Amsterdam Reproduction and Development Research Institute, Amsterdam, Netherlands.
Johannes FeenstraIsala Hospital Zwolle, Zwolle, Netherlands.
Geeta HampsonClinical Chemistry and Metabolic Medicine, Guy's and St Thomas NHS Trust, London, UK.
Mike StoneMetabolic Bone Clinic, University Hospital Llandough, Cardiff, UK.
Jane TurtonMetabolic Bone Clinic, University Hospital Llandough, Cardiff, UK.
Prashanth PatelChemical Pathology and Metabolic Disease, University Hospitals of Leicester NHS Trust, Leicester, UK.
Mashood SiddiqiMetabolic Bone Diseases, University Hospitals of Liverpool Group, Liverpool, UK.
Robin MunroUniversity Hospital Wishaw, Wishaw, UK.
Matthew RoyUniversity Hospitals Bristol NHS Trust, Bristol Royal Infirmary, Bristol, UK.
Zoe PaskinsUniversity of Keele, Rheumatology, Stoke-on-Trent, UK.
Deepa NarayananThe Centre for Metabolic Bone Disease, Hull University Teaching Hospitals NHS Trust, Hull, UK.
Ellen MalcolmDepartment of Medicine, Ninewells Hospital and Medical School, Dundee, UK.
Muhammad Kassim JavaidNuffield Orthopaedic Centre, University of Oxford, Oxford, UK.
Patricia OsborneBrittle Bone Society, Dundee, UK.
Jonathan C Y TangBioanalytical Facility, Norwich Medical School, Norwich, UK.
Wayne LamSouth East Scotland NHS Genetics Service, Edinburgh, UK.
David MooreSouth East Scotland NHS Genetics Service, Edinburgh, UK.
Holly A BlackSouth East Scotland NHS Genetics Service, Edinburgh, UK.
Andrew D DuckworthDepartment of Orthopaedic Surgery, Royal Infirmary of Edinburgh, NHS Lothian, Edinburgh, UK.
Navnit MakaramDepartment of Orthopaedic Surgery, Royal Infirmary of Edinburgh, NHS Lothian, Edinburgh, UK.
Tianyu GuoDepartment of Radiology, Royal Infirmary of Edinburgh, NHS Lothian, Edinburgh, UK.
Gregor StenhouseDepartment of Radiology, Royal Infirmary of Edinburgh, NHS Lothian, Edinburgh, UK.
Stuart H RalstonDepartment of Rheumatology, Western General Hospital, NHS Lothian, Edinburgh, UK. stuart.ralston@ed.ac.uk.ORCID http://orcid.org/0000-0002-2804-7586

Funding

Efficacy and Mechanism Evaluation Programme 14/200/18
6 · The paper itself

Abstract

introductionOsteogenesis imperfecta (OI) is a rare disorder causing multiple fractures throughout life. No treatment has been shown to reduce the risk of fractures in OI. Here, we present the baseline characteristics of participants in the Treatment of Osteogenesis Imperfecta with Parathyroid Hormone and Zoledronic Acid (TOPaZ) trial. The aim of the trial is to determine whether teriparatide and zoledronic acid are superior to standard care in reducing the risk of clinical fractures.

methodsWe summarised data on the baseline characteristics of TOPaZ participants, including demographics, genetic diagnosis, clinical features, bone density measurements, previous treatments, and fracture history.

resultsWe recruited 350 adults with a clinical diagnosis of OI in 27 European referral centres between June 2017 and October 2022. Overall, 266 (76.2%) had type I OI, 55 (15.8%) had type IV, and 19 (5.4%) had type III. The type was unknown in 9 (2.6%). Blue sclera were noted in 80.8%, and 35.8% had dentinogenesis imperfecta. Bisphosphonates had been administered to 28.1% in the 2 years prior to enrolment. Pathogenic variants in COL1A1 or COL1A2 were found in 87.6%. Fractures occurring in the 2 years prior to enrolment were not associated with bone density.

conclusionsThe TOPaZ population represents a unique cohort with which to study the genetic epidemiology and outcome of OI in relation to bone density and biochemical markers of bone turnover. When the trial reports, it will also provide new insights into the effect of an anabolic therapy, followed by antiresorptive treatment in the management of OI.

Indexed as

Bone Density Conservation AgentsOsteogenesis ImperfectaTeriparatideZoledronic AcidAdultAgedBone DensityFemaleFractures, BoneHumansMaleMiddle AgedBone Density Conservation AgentsTeriparatideZoledronic AcidBisphosphonateClinical trialGeneticOsteogenesis imperfectaTeriparatide

Identifiers

PMID41206390
PMCPMC12596313

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

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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 Socratic graph.