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Erythematous capillary-lymphatic malformations mimicking blood vascular anomalies.

Authors :
Hägerling R
Van Zanten M
Behncke RY
Ulferts S
Hansmeier NR
Märkl B
Witzel C
Ho B
Keeley V
Riches K
Mansour S
Gordon K
Ostergaard P
Mortimer PS
Source :
JCI insight [JCI Insight] 2023 Oct 23; Vol. 8 (20). Date of Electronic Publication: 2023 Oct 23.
Publication Year :
2023

Abstract

Superficial erythematous cutaneous vascular malformations are assumed to be blood vascular in origin, but cutaneous lymphatic malformations can contain blood and appear red. Management may be different and so an accurate diagnosis is important. Cutaneous malformations were investigated through 2D histology and 3D whole-mount histology. Two lesions were clinically considered as port-wine birthmarks and another 3 lesions as erythematous telangiectasias. The aims were (i) to demonstrate that cutaneous erythematous malformations including telangiectasia can represent a lymphatic phenotype, (ii) to determine if lesions represent expanded but otherwise normal or malformed lymphatics, and (iii) to determine if the presence of erythrocytes explained the red color. Microscopy revealed all lesions as lymphatic structures. Port-wine birthmarks proved to be cystic lesions, with nonuniform lymphatic marker expression and a disconnected lymphatic network suggesting a lymphatic malformation. Erythematous telangiectasias represented expanded but nonmalformed lymphatics. Blood within lymphatics appeared to explain the color. Blood-lymphatic shunts could be detected in the erythematous telangiectasia. In conclusion, erythematous cutaneous capillary lesions may be lymphatic in origin but clinically indistinguishable from blood vascular malformations. Biopsy is advised for correct phenotyping and management. Erythrocytes are the likely explanation for color accessing lymphatics through lympho-venous shunts.

Details

Language :
English
ISSN :
2379-3708
Volume :
8
Issue :
20
Database :
MEDLINE
Journal :
JCI insight
Publication Type :
Academic Journal
Accession number :
37698920
Full Text :
https://doi.org/10.1172/jci.insight.172179