Evidence map›Paper›PMID 30918362›Full record

Trial reportEuropean journal of human genetics : EJHG2019

Stakeholder views and attitudes towards prenatal and postnatal transplantation of fetal mesenchymal stem cells to treat Osteogenesis Imperfecta.

Melissa Hill, Celine Lewis, Megan Riddington, Belinda Crowe, Catherine DeVile, Anna L David, Oliver Semler, Magnus Westgren, Cecilia Götherström, Lyn S Chitty

Open access · hybridAbstract readClinical Trial
In one paragraph

Trial report in European journal of human genetics : EJHG, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed
2.1field-weighted citation impact, top 12% 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.

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

12 citing papers in PubMed, 21 citations in OpenAlex.

  1. Article
  2. Review
  3. Article
  4. Article
  5. Review
  6. Improving Development of Drug Treatments for Pregnant Women and the Fetus.Therapeutic innovation & regulatory science · 2022
    Review
  7. Regenerative medicine: prenatal approaches.The Lancet. Child & adolescent health · 2022
    Review
  8. Current Overview of Osteogenesis Imperfecta.Medicina (Kaunas, Lithuania) · 2021
    Review
  9. Review
  10. Fetal and Maternal Safety Considerations for In Utero Therapy Clinical Trials: iFeTiS Consensus Statement.Molecular therapy : the journal of the American Society of Gene Therapy · 2020
    Review
  11. Review
  12. Review
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

10 authors at 5 institutions in 3 countries.

Melissa HillNorth East Thames Regional Genetics Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK. Melissa.hill@ucl.ac.uk.
Celine LewisNorth East Thames Regional Genetics Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.ORCID 0000-0001-7169-1521
Megan RiddingtonOsteogenesis Imperfecta Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Belinda CroweOsteogenesis Imperfecta Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Catherine DeVileOsteogenesis Imperfecta Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Anna L DavidInstitute for Women's Health, University College London, London, UK.ORCID 0000-0002-0199-6140
Oliver SemlerFaculty of Medicine and University Hospital Cologne, Department of Pediatrics, University of Cologne, Cologne, Germany.ORCID 0000-0003-0029-7556
Magnus WestgrenDepartment of Clinical Science, Intervention & Technology, Karolinska Institutet, Stockholm, Sweden.
Cecilia GötherströmDepartment of Clinical Science, Intervention & Technology, Karolinska Institutet, Stockholm, Sweden.
Lyn S ChittyNorth East Thames Regional Genetics Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Great Ormond Street Hospital · GBGreat Ormond Street Hospital for Children NHS Foundation Trust · GBKarolinska Institutet · SELondon Women's Clinic · GBUniversity of Cologne · DE

Funding

EC | Horizon 2020 Framework Programme (EU Framework Programme for Research and Innovation H2020) 681045
6 · The paper itself

Abstract

The Boost Brittle Bones Before Birth (BOOSTB4) clinical trial is investigating the safety and efficacy of transplanting fetal derived mesenchymal stromal cells (MSCs) prenatally and/or in early postnatal life to treat severe Osteogenesis Imperfecta (OI). This study aimed to explore stakeholder views to understand perceived benefits or concerns, identify ethical issues and establish protocols for support and counselling. Semi-structured qualitative interviews were conducted with three groups; 1. Adults affected with OI, with and without children, and parents of children affected with OI; 2. Health professionals who work with patients with OI; 3. Patient advocates from relevant patient support groups. Interviews were digitally recorded, transcribed verbatim and analysed using thematic analysis. Interviews with 56 participants revealed generally positive views towards using fetal MSC transplantation to treat OI. Early treatment was considered advantageous for preventing fractures and reducing severity and could bring psychological benefits for parents. Common concerns were procedure safety, short/long-term side effects and whether transplantation would be effective. Difficulties inherent in decision-making were frequently discussed, as treatment efficacy is unknown and, by necessity, parents will make decisions at a time when they are vulnerable. Support needs may differ where there is a family history of OI compared to an unexpected diagnosis of OI. Explaining fetal MSC transplantation in a way that all parents can understand, clear expectation setting, psychological support and time for reflection during the decision-making process will be crucial to allow parents to make informed decisions about participation in the BOOSTB4 clinical trial.

Indexed as

AdultAttitudeChildFemaleFetal Stem CellsHumansMaleMesenchymal Stem CellsMesenchymal Stem Cell TransplantationMiddle AgedOsteogenesis ImperfectaOutcome Assessment, Health CareParentsStem Cell TransplantationYoung Adult

Identifiers

PMID30918362
PMCPMC6777523
OpenAlexW2923783964

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.