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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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Appearance of the eyelids at presentation. A, B: right eyelid. C, D: left eyelid.
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Figure 1: Appearance of the eyelids at presentation. A, B: right eyelid. C, D: left eyelid.

Mentions: A 64-year-old Japanese man consulted a local clinic because of tearing. Multiple nodules each approximately 5 mm in size were observed in bilateral upper and lower eyelids. He was referred to our hospital for further examination and treatment. He had an unremarkable medical history and no history of trauma. The corrected visual acuity was 0.8 in each eye. Well defined hard lesions in both upper and lower eyelids were present, without inflammatory signs such as redness (FigureĀ 1). Ophthalmoscopic findings were within normal limits except bilateral mild cataract changes. Laboratory investigations showed a white blood cell count of 10,400 per cm3 with 0.2% eosinophils. Serum concentration of immunoglobulin E was slightly elevated at 278.7 mg/dL, and immunoglobulin G4 was 59.3 mg/dL (normal range less than 135 mg/dL). Magnetic resonance imaging demonstrated bilateral multiple masses localized in the eyelids. The lesions were isointense to cerebral parenchyma on T1-weighted images, and hyperintense on T2-weighted images. After gadolinium infusion, the masses demonstrated moderate enhancement.


Angiolymphoid hyperplasia with eosinophilia occurring in bilateral eyelids.

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

Appearance of the eyelids at presentation. A, B: right eyelid. C, D: left eyelid.
© Copyright Policy - open-access
Related In: Results  -  Collection

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

Figure 1: Appearance of the eyelids at presentation. A, B: right eyelid. C, D: left eyelid.
Mentions: A 64-year-old Japanese man consulted a local clinic because of tearing. Multiple nodules each approximately 5 mm in size were observed in bilateral upper and lower eyelids. He was referred to our hospital for further examination and treatment. He had an unremarkable medical history and no history of trauma. The corrected visual acuity was 0.8 in each eye. Well defined hard lesions in both upper and lower eyelids were present, without inflammatory signs such as redness (FigureĀ 1). Ophthalmoscopic findings were within normal limits except bilateral mild cataract changes. Laboratory investigations showed a white blood cell count of 10,400 per cm3 with 0.2% eosinophils. Serum concentration of immunoglobulin E was slightly elevated at 278.7 mg/dL, and immunoglobulin G4 was 59.3 mg/dL (normal range less than 135 mg/dL). Magnetic resonance imaging demonstrated bilateral multiple masses localized in the eyelids. The lesions were isointense to cerebral parenchyma on T1-weighted images, and hyperintense on T2-weighted images. After gadolinium infusion, the masses demonstrated moderate enhancement.

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