Evidence map›Paper›PMID 31249378›Full record

ArticleScientific reports2019

Validation of the Fetal Lamb Model of Spina Bifida.

Luc Joyeux, Alexander C Engels, Johannes Van Der Merwe, Michael Aertsen, Premal A Patel, Marjolijn Deprez, Ahmad Khatoun, Savitree Pranpanus, Marina Gabriela Monteiro Carvalho Mori da Cunha, Stephanie De Vleeschauwer and 6 more

Abstract read
In one paragraph

Article in Scientific reports, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
15citing papers in PubMed, 1 pooled it
–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

15 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
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  4. Review
  5. Article
  6. Article
  7. Article
  8. Review
  9. Fetal Surgery for Myelomeningocele: Neurosurgical Perspectives.Advances and technical standards in neurosurgery · 2023
    Article
  10. Review
  11. Article
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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

16 authors.

Luc JoyeuxDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium.ORCID http://orcid.org/0000-0001-7331-6354
Alexander C EngelsDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium.
Johannes Van Der MerweDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium.
Michael AertsenDepartment of Radiology, University Hospital Gasthuisberg, Leuven, Belgium.
Premal A PatelRadiology Department, Great Ormond Street Hospital for Children, NHS Foundation Trust, London, United Kingdom.
Marjolijn DeprezResearch group Experimental Neurosurgery and Neuroanatomy, Department of Neurosciences, KU Leuven, Leuven, Belgium.
Ahmad KhatounExperimental Otorhinolaryngology, Department of Neurosciences, KU Leuven, Leuven, Belgium.
Savitree PranpanusDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium.
Marina Gabriela Monteiro Carvalho Mori da CunhaDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium.ORCID http://orcid.org/0000-0001-6658-7797
Stephanie De VleeschauwerAnimal Research Center, Biomedical Sciences, KU Leuven, Leuven, Belgium.
Johanna ParraBarcelona Center for Maternal Fetal and Neonatal Medicine, University of Barcelona, Barcelona, Spain.
Katerina ApeltDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium.
Myles Mc LaughlinExperimental Otorhinolaryngology, Department of Neurosciences, KU Leuven, Leuven, Belgium.
Frank Van CalenberghDepartment of Neurosurgery, University Hospital Gasthuisberg, Leuven, Belgium.
Enrico RadaelliDepartment of Pathobiology, Ryan Veterinary Hospital, University of Pennsylvania School of Veterinary Medicine, Philadelphia, PA, United States.
Jan DeprestDepartment of Development and Regeneration, Cluster Urogenital, Abdominal and Plastic surgery, Biomedical Sciences, Katholieke Universiteit (KU) Leuven, Leuven, Belgium. jan.deprest@uzleuven.be.

Funding

Wellcome TrustWellcome Trust (Wellcome) WT101957
6 · The paper itself

Abstract

A randomized trial demonstrated that fetal spina bifida (SB) repair is safe and effective yet invasive. New less invasive techniques are proposed but are not supported by adequate experimental studies. A validated animal model is needed to bridge the translational gap to the clinic and should mimic the human condition. Introducing a standardized method, we comprehensively and reliably characterize the SB phenotype in two lamb surgical models with and without myelotomy as compared to normal lambs. Hindbrain herniation measured on brain magnetic resonance imaging (MRI) was the primary outcome. Secondary outcomes included gross examination with cerebrospinal fluid (CSF) leakage test, neurological examination with locomotor assessment, whole-body MRI, motor and somatosensory evoked potentials; brain, spinal cord, hindlimb muscles, bladder and rectum histology and/or immunohistochemistry. We show that the myelotomy model best phenocopies the anatomy, etiopathophysiology and symptomatology of non-cystic SB. This encompasses hindbrain herniation, ventriculomegaly, posterior fossa anomalies, loss of brain neurons; lumbar CSF leakage, hindlimb somatosensory-motor deficit with absence of motor and somatosensory evoked potentials due to loss of spinal cord neurons, astroglial cells and myelin; urinary incontinence. This model obtains the highest validity score for SB animal models and is adequate to assess the efficacy of novel fetal therapies.

Indexed as

Disease Models, AnimalFetusSpinal DysraphismAnimalsFemaleMagnetic Resonance ImagingMotor ActivityPhenotypePregnancyReproducibility of ResultsSheep

Identifiers

PMID31249378
PMCPMC6597719

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.