Evidence map›Paper›PMID 24990152›Full record

ArticleHuman molecular genetics2014

Lack of CCM1 induces hypersprouting and impairs response to flow.

Tara M Mleynek, Aubrey C Chan, Michael Redd, Christopher C Gibson, Chadwick T Davis, Dallas S Shi, Tiehua Chen, Kandis L Carter, Jing Ling, Raquel Blanco and 3 more

Open access · hybridAbstract read
In one paragraph

Article in Human molecular genetics, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.

0numbers the graph read from it
0cells of the map it votes in
25citing papers in PubMed
2.4field-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

25 citing papers in PubMed, 34 citations in OpenAlex.

  1. Review
  2. Article
  3. KRIT1 in vascular biology and beyond.Bioscience reports · 2024
    Review
  4. Article
  5. Article
  6. Article
  7. Review
  8. Article
  9. Article
  10. Article
  11. Review
  12. VEGF signalling enhances lesion burden in KRIT1 deficient mice.Journal of cellular and molecular medicine · 2020
    Article
  13. Article
  14. Phenotypic characterization of murine models of cerebral cavernous malformations.Laboratory investigation; a journal of technical methods and pathology · 2019
    Article
  15. Vascular heterogeneity and specialization in development and disease.Nature reviews. Molecular cell biology · 2017
    Review
  16. Article
  17. Review
  18. Article
  19. Article
  20. Article
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

13 authors at 4 institutions in 6 countries.

Tara M MleynekDepartment of Molecular Medicine, Department of Oncological Sciences.
Aubrey C ChanDepartment of Molecular Medicine, Department of Oncological Sciences.
Michael ReddFlourescence Imaging Core.
Christopher C GibsonDepartment of Molecular Medicine, Department of Bioengineering.
Chadwick T DavisDepartment of Molecular Medicine, Department of Human Genetics.
Dallas S ShiDepartment of Molecular Medicine, Department of Human Genetics.
Tiehua ChenDepartment of Molecular Medicine, Small Animal Ultrasound Core, University of Utah, Salt Lake City 84112, USA.
Kandis L CarterDepartment of Molecular Medicine, Small Animal Ultrasound Core, University of Utah, Salt Lake City 84112, USA.
Jing LingDepartment of Molecular Medicine.
Raquel BlancoVascular Biology Laboratory, London Research Institute, Cancer Research UK, London WC2A 3LY, UK.
Holger GerhardtVascular Patterning Laboratory, VIB3-Vesalius Research Center and CMVB, Department of Oncology, KU Leuven Campus Gasthuisberg O&N4, Herestraat 49 box 912, Leuven B-3000, Belgium.
Kevin WhiteheadDepartment of Molecular Medicine, Small Animal Ultrasound Core, University of Utah, Salt Lake City 84112, USA, Division of Cardiovascular Medicine, Salt Lake City 84132, USA and.
Dean Y LiDepartment of Molecular Medicine, Department of Oncological Sciences, Division of Cardiovascular Medicine, Salt Lake City 84132, USA and The Key Laboratory for Human Disease Gene Study of Sichuan Province, Institute of Laboratory Medicine, Sichuan Academy of Medical Sciences & Sichuan Provincial People's Hospital, Chengdu, Sichuan 610072, China dean.li@u2m2.utah.edu kevin.whitehead@u2m2.utah.edu.
Institute of Molecular Medicine · INBioengineering Center · RUCancer Research UK · GBKU Leuven · BE

Funding

Vascular Malformations of the CNSR01HL065648 · NHLBI · UNIVERSITY OF UTAH · PI LI, DEAN YAW · 2000 to 2013
$2.9M
KRIT1 in Vascular Development and DysfunctionR01NS075168 · NINDS · UNIVERSITY OF UTAH · PI WHITEHEAD, KEVIN J · 2011 to 2015
$1.6M
High Content Sceening for Hereditary Stroke SyndromeR01NS080893 · NINDS · UNIVERSITY OF UTAH · PI LI, DEAN YAW · 2013 to 2015
$975k
NHLBI NIH HHS R01HL065648NINDS NIH HHS R01 NS075168NINDS NIH HHS R01 NS080893
6 · The paper itself

Abstract

Cerebral cavernous malformation (CCM) is a disease of vascular malformations known to be caused by mutations in one of three genes: CCM1, CCM2 or CCM3. Despite several studies, the mechanism of CCM lesion onset remains unclear. Using a Ccm1 knockout mouse model, we studied the morphogenesis of early lesion formation in the retina in order to provide insight into potential mechanisms. We demonstrate that lesions develop in a stereotypic location and pattern, preceded by endothelial hypersprouting as confirmed in a zebrafish model of disease. The vascular defects seen with loss of Ccm1 suggest a defect in endothelial flow response. Taken together, these results suggest new mechanisms of early CCM disease pathogenesis and provide a framework for further study.

Indexed as

AnimalsAnimals, Genetically ModifiedHemangioma, Cavernous, Central Nervous SystemHumansKRIT1 ProteinMice, KnockoutMicrotubule-Associated ProteinsProto-Oncogene ProteinsRetinaZebrafishKRIT1 ProteinKrit1 protein, mouseMicrotubule-Associated ProteinsProto-Oncogene Proteins

Identifiers

PMID24990152
PMCPMC4222362
OpenAlexW2130856505

What OpenQuestion holds

Textmetadata
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.