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Angiolymphoid hyperplasia with eosinophilia developing within a port wine stain
Robert N Manton1, Tinte Itinteang2, Sophie de Jong2
1Centre for the Study & Treatment of Vascular Birthmarks, Wellington Regional Plastic, Maxillofacial & Burns Unit, Hutt Hospital, Wellington, New Zealand.
Journal of Cutaneous Pathology
|May 27, 2015
Summary
A rare case of angiolymphoid hyperplasia with eosinophilia (ALHE) developed in a port wine stain. The condition was linked to elevated renin levels, suggesting a role for the renin-angiotensin system.
Area of Science:
- Dermatology
- Pathology
- Endocrinology
Background:
- Port wine stains are congenital vascular malformations.
- Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular proliferation.
Observation:
- A 19-year-old male presented with skin thickening and bleeding from a port wine stain.
- Histopathology confirmed ALHE arising from the port wine stain, a previously unreported occurrence.
- Elevated serum renin levels were noted, which normalized post-excision.
Findings:
- Demonstrated expression of angiotensin-converting enzyme (ACE) and angiotensin II receptors (AT1, AT2) in the ALHE lesion.
- The renin-angiotensin system (RAS) components were present within the affected tissue.
Implications:
- This case suggests a potential role for the renin-angiotensin system in the pathogenesis of ALHE associated with port wine stains.
- Further research into the RAS may reveal novel therapeutic targets for this rare condition.

