Evidence map›Paper›PMID 39569520›Full record

ArticleArteriosclerosis, thrombosis, and vascular biology2025

Intravital Imaging of Disease Mechanisms in a Mouse Model of CCM Skin Lesions-Brief Report.

Michael M Orlich, Amanda Norrén, Malavika Desai, Annegret Holm, Harry Kozakewich, Hans Schoofs, Taija Mäkinen, Joyce Bischoff, Konstantin Gaengel

Abstract read
In one paragraph

Article in Arteriosclerosis, thrombosis, and vascular biology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0cells of the map it votes in
0citing papers in PubMed
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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

9 authors.

Michael M OrlichDepartment of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, Sweden (M.M.O., A.N., M.D., H.S., T.M., K.G.).ORCID 0000-0001-8632-2593
Amanda NorrénDepartment of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, Sweden (M.M.O., A.N., M.D., H.S., T.M., K.G.).ORCID 0009-0006-7783-0338
Malavika DesaiDepartment of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, Sweden (M.M.O., A.N., M.D., H.S., T.M., K.G.).ORCID 0009-0005-0055-8148
Annegret HolmVascular Biology Program, Department of Surgery (A.H., J.B.), Boston Children's Hospital, Harvard Medical School, MA.ORCID 0000-0002-8691-4131
Harry KozakewichDepartment of Pathology (H.K.), Boston Children's Hospital, Harvard Medical School, MA.
Hans SchoofsDepartment of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, Sweden (M.M.O., A.N., M.D., H.S., T.M., K.G.).ORCID 0000-0003-3429-912X
Taija MäkinenDepartment of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, Sweden (M.M.O., A.N., M.D., H.S., T.M., K.G.).ORCID 0000-0002-9338-1257
Joyce BischoffVascular Biology Program, Department of Surgery (A.H., J.B.), Boston Children's Hospital, Harvard Medical School, MA.ORCID 0000-0002-6367-1974
Konstantin GaengelDepartment of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, Sweden (M.M.O., A.N., M.D., H.S., T.M., K.G.).ORCID 0000-0002-2682-2833

Funding

Capillary malformation: From somatic GNAQ mutations to disrupted endothelial biologyR01HL127030 · NHLBI · BOSTON CHILDREN'S HOSPITAL · PI BISCHOFF, JOYCE E., GREENE, ARIN K. · 2016 to 2023
$5.2M
NHLBI NIH HHS R01 HL127030
6 · The paper itself

Abstract

backgroundCerebral cavernous malformation (CCM) is a disease characterized by vascular malformations that primarily develop in the brain. These malformations are prone to leak, and their rupture or thrombotic closure can cause life-threatening hemorrhages and strokes. Mouse models have been instrumental to study the disease, but most cause premature lethality, precluding the investigation of disease mechanisms through intravital microscopy. Current mouse models also do not recapitulate human CCM skin lesions.

methodsEndothelial-specific deletion of

resultsWe report that human CCM skin lesions can be recapitulated in a mouse model and that skin lesions share hallmarks of CCM brain lesions. Intravital imaging reveals that CCM skin lesions are slow-flow malformations prone to thrombus formation.

conclusionsIntravital imaging of CCM skin lesions expands the toolkit of CCM research and allows longitudinal studies of lesion growth.

Indexed as

Disease Models, AnimalHemangioma, Cavernous, Central Nervous SystemIntravital MicroscopySkinAnimalsApoptosis Regulatory ProteinsHumansMembrane ProteinsMiceMice, Inbred C57BLMice, KnockoutTamoxifenThrombomodulinThrombosisApoptosis Regulatory ProteinsMembrane ProteinsPDCD10 protein, mouseTamoxifenTHBD protein, humanThrombomodulinbraincerebral cavernous malformations 3intravital microscopyskin abnormalitiesthrombosis

Identifiers

PMID39569520
PMCPMC11668616

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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 OpenQuestion graph.