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Angiolymphoid hyperplasia with eosinophilia occurring in bilateral eyelids.

Ueda S, Goto H, Usui Y, Nagai T, Nagao T - BMC Ophthalmol (2013)

Bottom Line: Histopathological examination of the excised masses demonstrated proliferated blood vessels lined by plump endothelial cells together with a lymphoid and eosinopilic infiltrate, compatible with a diagnosis of ALHE.Flow cytometry studies showed that the mass consisted of mostly CD3-positive cells.The cause of ALHE remains uncertain, but our results of flow cytemetry suggest that T cells are related to the pathogenesis of this disease.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Ophthalmology, Tokyo Medical University Hospital, Tokyo, Japan. shunueda@tg8.so-net.ne.jp

ABSTRACT

Background: Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon benign lesion, primarily occurring in the head and neck. ALHE arising from the ocular adnexa is rare, and the bilateral presentation is especially rare in the eyelids.

Case presentation: A 64-year-old Japanese man presented with tearing. Multiple nodules, approximately 5 mm in size, were observed in bilateral upper and lower eyelids. Surgical excisions of the both eyelids masses were performed. Histopathological examination of the excised masses demonstrated proliferated blood vessels lined by plump endothelial cells together with a lymphoid and eosinopilic infiltrate, compatible with a diagnosis of ALHE. Flow cytometry studies showed that the mass consisted of mostly CD3-positive cells. During two-year follow-up, no recurrence of the mass was observed and the patient had no subjective symptom of tearing

Conclusion: ALHE may occur in the bilateral eyelids. The cause of ALHE remains uncertain, but our results of flow cytemetry suggest that T cells are related to the pathogenesis of this disease.

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Related in: MedlinePlus

Histopathological examinations of the biopsy. A: Hematoxylin-eosin staining shows plump endothelial cells surrounded by collagenous stroma containing a lymphoid and eosinopilic infiltration; ×600. B: CD31 immunostaining highlights the prominent vascularity; ×600.
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Figure 2: Histopathological examinations of the biopsy. A: Hematoxylin-eosin staining shows plump endothelial cells surrounded by collagenous stroma containing a lymphoid and eosinopilic infiltration; ×600. B: CD31 immunostaining highlights the prominent vascularity; ×600.

Mentions: For diagnosis purpose, biopsy of the right upper eyelid mass was performed via the skin and conjunctiva. Since a pathological examination during surgery showed eosinophilic infiltration and an allergic disease was suspected, local injection of triamcinolone acetonide was performed at the end of procedures. Histopathological examination showed proliferated blood vessels lined by plump endothelial cells with an epithelioid appearance, surrounded by a collagenous stroma containing a lymphoid and eosinophilic infiltration, and no evidence of lymphoid follicles (Figure 2). These histopathological findings were compatible with a diagnosis of ALHE. In immunohistochemical studies, CD31 immunostaining highlighted the prominent vascularity and the plump endothelial cells. CD3 and CD20 immunostaining was diffusely positive in the infiltrate, indicating that most of the infiltrating cells were T cells. Flow cytometry studies showed that the mass was consisted of mainly CD3-positive cells, confirming the immunohistochemical findings. No evidence of IgH or T cell receptor (TCR) rearrangement was detected.


Angiolymphoid hyperplasia with eosinophilia occurring in bilateral eyelids.

Ueda S, Goto H, Usui Y, Nagai T, Nagao T - BMC Ophthalmol (2013)

Histopathological examinations of the biopsy. A: Hematoxylin-eosin staining shows plump endothelial cells surrounded by collagenous stroma containing a lymphoid and eosinopilic infiltration; ×600. B: CD31 immunostaining highlights the prominent vascularity; ×600.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3750691&req=5

Figure 2: Histopathological examinations of the biopsy. A: Hematoxylin-eosin staining shows plump endothelial cells surrounded by collagenous stroma containing a lymphoid and eosinopilic infiltration; ×600. B: CD31 immunostaining highlights the prominent vascularity; ×600.
Mentions: For diagnosis purpose, biopsy of the right upper eyelid mass was performed via the skin and conjunctiva. Since a pathological examination during surgery showed eosinophilic infiltration and an allergic disease was suspected, local injection of triamcinolone acetonide was performed at the end of procedures. Histopathological examination showed proliferated blood vessels lined by plump endothelial cells with an epithelioid appearance, surrounded by a collagenous stroma containing a lymphoid and eosinophilic infiltration, and no evidence of lymphoid follicles (Figure 2). These histopathological findings were compatible with a diagnosis of ALHE. In immunohistochemical studies, CD31 immunostaining highlighted the prominent vascularity and the plump endothelial cells. CD3 and CD20 immunostaining was diffusely positive in the infiltrate, indicating that most of the infiltrating cells were T cells. Flow cytometry studies showed that the mass was consisted of mainly CD3-positive cells, confirming the immunohistochemical findings. No evidence of IgH or T cell receptor (TCR) rearrangement was detected.

Bottom Line: Histopathological examination of the excised masses demonstrated proliferated blood vessels lined by plump endothelial cells together with a lymphoid and eosinopilic infiltrate, compatible with a diagnosis of ALHE.Flow cytometry studies showed that the mass consisted of mostly CD3-positive cells.The cause of ALHE remains uncertain, but our results of flow cytemetry suggest that T cells are related to the pathogenesis of this disease.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Ophthalmology, Tokyo Medical University Hospital, Tokyo, Japan. shunueda@tg8.so-net.ne.jp

ABSTRACT

Background: Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon benign lesion, primarily occurring in the head and neck. ALHE arising from the ocular adnexa is rare, and the bilateral presentation is especially rare in the eyelids.

Case presentation: A 64-year-old Japanese man presented with tearing. Multiple nodules, approximately 5 mm in size, were observed in bilateral upper and lower eyelids. Surgical excisions of the both eyelids masses were performed. Histopathological examination of the excised masses demonstrated proliferated blood vessels lined by plump endothelial cells together with a lymphoid and eosinopilic infiltrate, compatible with a diagnosis of ALHE. Flow cytometry studies showed that the mass consisted of mostly CD3-positive cells. During two-year follow-up, no recurrence of the mass was observed and the patient had no subjective symptom of tearing

Conclusion: ALHE may occur in the bilateral eyelids. The cause of ALHE remains uncertain, but our results of flow cytemetry suggest that T cells are related to the pathogenesis of this disease.

Show MeSH
Related in: MedlinePlus