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Angiolymphoid Hyperplasia with

Eosinophilia

Spencer Rusin M4, CUMC

Deba P Sarma, MDOmaha

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Patient Presentation

F 55, presents with a 10-month history of :

Non-ulcerated, painless nodule (0.5 cm)on her lower lip

No history of trauma or ear-nose-throatdisease.

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Differential Diagnosis

Mucocele

Lymphocytoma cutis

Granuloma faciale Benign and malignant tumors of skin and

adnexae

Kimura disease

Others

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A well circumscribed

dermal nodule

composed of central

angiomatous vascular

proliferation with

stromal and peripheral

infiltrates of

lymphocytes andeosinophils.

H&E: low power

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Proliferation of small blood vessels, lined by enlargedendothelial cells (epitheliod in appearance) with uniform ovoid

nuclei and intracytoplasmic vacuoles.

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Prominent eosinophilic and lymphocytic infiltration

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Lymphoid aggregate with follicle formation amongst the

vascular proliferative cells.

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Immunostains

CD 3Peripheral lymphocytes: Positive

CD 20 - Peripheral lymphocytes: Positive

CD31Vascular epitheliod endothelial cells:Positive

CK AE1/3 - Negative

S-100 - Negative

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CD 31 stain highlights the endothelial cells demonstrating a

strong angiogenesis component to the nodule.

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Diagnosis

Angiolymphoid hyperplasia with eosinophilia

(ALHE)

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Differential Diagnosis

ALHE

Primarily a localizedhyperplasia

Infrequentlymphadenopathy

20%

Rare blood eosinophlia

Histology:

Vascularproliferation>inflammatory cells

Epitheliod endothelialcells lining bloodvessels

Eosinophils present

Kimura Disease

Systemic involvement: Lymphadenopathy

Blood eosinophlia Nephrotic syndrome as

a result of glomerularIgE deposition.

Histological presentation ofKimura disease differs fromALHE in two factors.

Vascular proliferation

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ALHE

General presentation:

Range from asymptomatic to itchy orpainful erythematous nodules, 2-3cm indiameter.

The nodules may pulsate or bleed.

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Pre-auricular lesions of ALHE.

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ALHE Hypotheses regarding ALHEs origin:

Reactive process to insect bites

Hyperestrogen states

Immunologic mechanisms

Reactive vascular proliferation subsequent toinflammation associated with traumatized blood

vessels One study reported a history of trauma in only 9%

of 116 patients with ALHE

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ALHE

Age: 20-50 years, M = F

Locations affected by ALHE:

Head and neck:

Specifically the forehead, scalp, and skin aroundears.

Trunk and genitalia

Three documented cases of ALHE affecting the lip.

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ALHE

Progression of ALHE:

Most common course: ALHE remains stable

Infrequent outcome: ALHE spontaneously

regresses Chronic nature of ALHE necessitates treatment.

Recurrence rate ranges from 33-50%

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Treatment

Medical:

Isoretinoin

Corticosteroids

interferon alfa-2b Benefits:

Improved cosmetic outcomes

Limitations:

Relies on patient compliance Not a permanent cure

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Treatment

Surgical:

Laser therapy

Carbon dioxide laser

Ultralong pulsed dye laser Nd:YAG laser

Benefits:

Improved cosmetic outcome

Limitations: Multiple treatments

Adversely affected by the depth of invasion orsize of vessels

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Treatment

Surgical:

Excisional

Simple surgical excision

Mohs surgery Benefits:

Excision of the arterial and venous segments atthe base decrease recurrence

Limitations:

Scarring

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References

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G.C. Wells, I.W. Whimster, "Subcutaneous angiolymphoid hyperplasia with eosinophilia,British Journal of Dermatology, vol. 81, pp 1-15, 1969.

R.L. Moy, D.B. Luftman, Q.H. Nguyen, J.S. Amenta,"Estrogen receptors and the response to

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f i d

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J.I. Lopez, S.B. Battaglino, Angiolymphoid hyperplasia with eosinophilia of the lower lip,International Journal of Dermatology, vol. 32, issue 5, pp. 361-362, 1993.

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B.V. Diaz, M.C. Lenoir, A. Ladoux, C. Frelin, M. Demarchez, S. Michel, Regulation ofvascular endothelial growth factor expression in human keratinocytes by retinoids,Journal of Biological Chemistry, vol. 275, no. 1, pps. 642-650, 2000.

F. El Sayed, R. Dhaybi, A. Ammoury, M. Chababi, Angiolymphoid hyperplasia witheosinophilia: efficacy of isotretinoin?, Head & Face Medicine, vol. 2, p. 32-36, 2006. T. Kaur, K. Sandhu, S. Gupta, A.J. Kanwar, B. Kumar, Treatment of angiolymphoid

hyperplasia with eosinophilia with the carbon dioxide laser, Journal of DermatologicalTreatment, vol. 15, issue 5, pps. 328-330, 2004.

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C.J. Miller, M.D. Ioffreda, C.T. Ammirati, Mohs micrographic surgery for angiolymphoidhyperplasia with eosinophilia, Dermatological Surgery, vol 30,issue 8, pps. 1169-1173,2004.

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