Angiolymphoid Hyperplasia with Eosinophilia. Spencer Rusin M4, CUMC Deba P Sarma, MD Omaha

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1 Angiolymphoid Hyperplasia with Eosinophilia Spencer Rusin M4, CUMC Deba P Sarma, MD Omaha

2 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-throat disease.

3 Differential Diagnosis Mucocele Lymphocytoma cutis Granuloma faciale Benign and malignant tumors of skin and adnexae Kimura disease Others

4 H&E: low power A well circumscribed dermal nodule composed of central angiomatous vascular proliferation with stromal and peripheral infiltrates of lymphocytes and eosinophils.

5 Proliferation of small blood vessels, lined by enlarged endothelial cells (epitheliod( in appearance) with uniform ovoid nuclei and intracytoplasmic vacuoles.

6 Prominent eosinophilic and lymphocytic infiltration

7 Lymphoid aggregate with follicle formation amongst the vascular proliferative cells.

8 Immunostains CD 3 Peripheral lymphocytes: Positive CD 20 - Peripheral lymphocytes: Positive CD31 Vascular epitheliod endothelial cells: Positive CK AE1/3 - Negative S Negative

9 CD 31 stain highlights the endothelial cells demonstrating a strong angiogenesis component to the nodule.

10 Diagnosis Angiolymphoid hyperplasia with eosinophilia (ALHE)

11 Differential Diagnosis ALHE Kimura Disease Primarily a localized hyperplasia Infrequent lymphadenopathy 20% Rare blood eosinophlia Histology: Vascular proliferation>inflamma tory cells Epitheliod endothelial cells lining blood vessels Eosinophils present Systemic involvement: Lymphadenopathy Blood eosinophlia Nephrotic syndrome as a result of glomerular IgE deposition. Histological presentation of Kimura disease differs from ALHE in two factors. Vascular proliferation <inflammatory cells Blood vessels are lined by attenuated endothelial cells, not epitheliod endothelial cells

12 ALHE General presentation: Range from asymptomatic to itchy or painful erythematous nodules, 2-3cm in diameter. The nodules may pulsate or bleed.

13 Pre-auricular lesions of ALHE.

14 ALHE Hypotheses regarding ALHE s origin: Reactive process to insect bites Hyperestrogen states Immunologic mechanisms Reactive vascular proliferation subsequent to inflammation associated with traumatized blood vessels One study reported a history of trauma in only 9% of 116 patients with ALHE

15 ALHE Age: years, M = F Locations affected by ALHE: Head and neck: Specifically the forehead, scalp, and skin around ears. Trunk and genitalia Three documented cases of ALHE affecting the lip.

16 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%

17 Treatment Medical: Isoretinoin Corticosteroids interferon alfa-2b Benefits: Improved cosmetic outcomes Limitations: Relies on patient compliance Not a permanent cure

18 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 or size of vessels

19 Treatment Surgical: Excisional Simple surgical excision Moh s surgery Benefits: Excision of the arterial and venous segments at the base decrease recurrence Limitations: Scarring

20 References S. Seregard, Angiolymphoid hyperplasia with eosinophilia should not be confused with Kimura's disease, Acta Ophthalmologica Scandinavica,, vol. 79, issue 1, pps , 93, S.W. Weiss, J.R. Goldblum,, "Enzinger" and Weiss's Soft Tissue Tumors, 4th edition," St. Louis: Mosby,, G.C. Wells, I.W. Whimster,, "Subcutaneous angiolymphoid hyperplasia with eosinophilia, British Journal of Dermatology,, vol. 81, pp 1-15, R.L. Moy,, D.B. Luftman,, Q.H. Nguyen, J.S. Amenta,"Estrogen receptors and the response to sex hormones in angiolymphoid hyperplasia with eosinophilia," Archives of Dermatology, vol 128, pp , R. Grimwood,, J.M. Swinehart,, J.L Aeling,, "Angiolymphoid hyperplasia with eosinophilia," Archives of Dermatology,, vol. 115, pp , P. Von den Driesch,, M. Gruschwitz,, H. Schell, W. Sterry, Distribution of adhesion molecules, IgE,, and CD23 in a case of angiolymphoid hyperplasia with eosinophilia, Journal of the American Academy of Dermatology,, vol. 26, issue 5, part 2, pp , T.G. Olsen, E.B. Helwig,, "Angiolymphoid hyperplasia with eosinophilia," Journal of American Academy of Dermatology, vol 12, pp , P.G. Henry, J.W. Burnett, Angiolymphoid hyperplasia with eosinophilia, Archives of Dermatology,, vol. 114, no. 8, pp , J.F. Fetsch,, S.W. Weiss, Observations concerning the pathogenesis of epithelioid hemangioma (angiolymphoid hyperplasia), Modern Pathology,, vol. 4, issue 4, pp , T.G. Olsen, E.B. Helwig, Angiolymphoid hyperplasia with eosinophilia. A clinicopathologic study of 118 patients, Journal of the American Academy of Dermatology,, vol. 12, issue 5, pp , J. Scurry, G. Dennerstein,, J. Brenan, Angiolymphoid hyperplasia with eosinophilia of the vulva, Australian and New Zealand Journal of Obstetrics and Gynaecology,, vol. 35, issue 3, pp , 1995.

21 References continued J.R. Srigley,, A.G. Ayala, N.G. Ordonez, A.W. van Nostrand, Epithelioid hemangioma of the penis. A rare and distinctive vascular lesion, Archives of Pathology and Laboratory Medicine,, vol. 109, pp , J.I. Lopez, S.B. Battaglino, Angiolymphoid hyperplasia with eosinophilia of the lower lip, International Journal of Dermatology,, vol. 32, issue 5, pp , H. Suzuki, A. Hatamochi,, M. Horie,, T. Suzuki, S. Yamazaki, A A case of angiolymphoid hyperplasia with eosinophilia (ALHE) of the upper lip, Journal of Dermatology,, vol. 32, no. 12, pp , O.F. Salinas, Y.S. Corredoira, G.A. Rojas, Angiolymphoid hyperplasia of the lip with eosinophilia. Report of one case, Revista Medica de Chile,, vol. 135, no.5, pp , (in Spanish) A. Satpathy,, C. Moss, F. Raafat,, R. Slator, Spontaneous regression of a rare tumour in a child: angiolymphoid hyperplasia with eosinophilia of the hand: case report and review of the literature, British Journal of Plastic Surgery,, vol. 58, issue 6, pps , B.V. Diaz, M.C. Lenoir, A. Ladoux,, C. Frelin,, M. Demarchez,, S. Michel, Regulation of vascular endothelial growth factor expression in human keratinocytes by retinoids, Journal of Biological Chemistry,, vol. 275, no. 1, pps , F. El Sayed,, R. Dhaybi,, A. Ammoury,, M. Chababi, Angiolymphoid hyperplasia with eosinophilia: efficacy of isotretinoin?, Head & Face Medicine,, vol. 2, p , T. Kaur,, K. Sandhu,, S. Gupta, A.J. Kanwar,, B. Kumar, Treatment of angiolymphoid hyperplasia with eosinophilia with the carbon dioxide laser, Journal of Dermatological Treatment,, vol. 15, issue 5, pps , C. Angel, A. Lewis, T. Griffin, E. Levy, A. Benedetto, Angiolymphoid hyperplasia successfully treated with an ultralong pulsed dye laser, Dermatologic Surgery,, vol. 31, pps , C.J. Miller, M.D. Ioffreda,, C.T. Ammirati, Mohs micrographic surgery for angiolymphoid hyperplasia with eosinophilia, Dermatological Surgery, vol 30,issue 8, pps , T. Rohrer, A.Allan, ANgiolymphoid hyperplasia with eosinophilia successfully treated with a long-pulsed tunable dye laser, Dermatologic Surgery,, vol. 26, issue 3, pps , 2000.

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