Vascular Organoids Derived from Capillary malformation-induced Pluripotent Stem Cells Exhibit Disease-Relevant Phenotypes

Capillary malformation (CM) is a congenital vascular anomaly that affects the skin, mucosa, eye, and brain. A major obstacle to mechanistic and drug screening studies for CM has been the lack of preclinical models. In this study, we established vascular organoids (VOs) generated through the self-assembly of vascular lineages of endothelial cells and smooth muscle cells differentiated from CM-induced pluripotent stem cells (iPSC). Within these VOs induced endothelial cells and smooth muscle cells organized into juxtapositions to form vascular branches. CM patient iPSC-derived VOs showed a higher density of endothelial and smooth muscle cell populations and greater vascular branch lengths as compared with VOs derived from iPSCs generated from healthy skin biopsies. Overall, this study represents the first disease-relevant VO model of CM, providing a valuable platform for future mechanistic studies and drug screening.

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Publication Details

Journal
Stem Cell Reviews and Reports
Published
2025-09-25
DOI
https://doi.org/10.1007/s12015-025-10984-8
Primary Topic
Vascular Malformations and Hemangiomas
Type
article
Field-Weighted Citation Impact
0.00

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article

Vascular Organoids Derived from Capillary malformation-induced Pluripotent Stem Cells Exhibit Disease-Relevant Phenotypes

Anna Harper, Wenbin Tan, Camilla Ferreira Wenceslau, Siyi He et al.
Stem Cell Reviews and Reports
Vascular Malformations and Hemangiomas
article

Vascular Organoids Derived from Capillary malformation-induced Pluripotent Stem Cells Exhibit Disease-Relevant Phenotypes

Anna Harper, Wenbin Tan, Camilla Ferreira Wenceslau, Siyi He, Dong‐bao Chen, Vi Nguyen, Anil Goud Jegga, J. Stuart Nelson, Yunguan Wang, Ivonne Astrid Arias Castellanos, Daping Fan, Marcelo Hochman, Mackenzie Azuero
article en

Abstract

Capillary malformation (CM) is a congenital vascular anomaly that affects the skin, mucosa, eye, and brain. A major obstacle to mechanistic and drug screening studies for CM has been the lack of preclinical models. In this study, we established vascular organoids (VOs) generated through the self-assembly of vascular lineages of endothelial cells and smooth muscle cells differentiated from CM-induced pluripotent stem cells (iPSC). Within these VOs induced endothelial cells and smooth muscle cells organized into juxtapositions to form vascular branches. CM patient iPSC-derived VOs showed a higher density of endothelial and smooth muscle cell populations and greater vascular branch lengths as compared with VOs derived from iPSCs generated from healthy skin biopsies. Overall, this study represents the first disease-relevant VO model of CM, providing a valuable platform for future mechanistic studies and drug screening.

Stem Cell Reviews and ReportsVol. 22(1)
Cincinnati Children's Hospital Medical Center (US), Medical University of South Carolina (US), University of South Carolina (US), University of California, Irvine (US), College of Charleston (US), Beckman Laser Institute and Medical Clinic (US), University of Cincinnati Medical Center (US)
University of South Carolina, School of Medicine, University of South Carolina
Openalex Percentile: Top 58%
Vascular Malformations and Hemangiomas
DOIOpen Access PDFOpenAlex
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