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| CASE REPORT |
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| Year : 2015 | Volume
: 3
| Issue : 1 | Page : 40-42 |
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Angiolymphoid hyperplasia - A case report
M Patel1, R Mahajan2, K Ninama3, FE Bilimoria4
1 Resident; Department of Dermatology, SBKS MI & RC, Piparia, Waghodia, Vadodara, Gujarat, India
2 Associate Professor; Department of Dermatology, SBKS MI & RC, Piparia, Waghodia, Vadodara, Gujarat, India
3 Assistant Professor; Department of Dermatology, SBKS MI & RC, Piparia, Waghodia, Vadodara, Gujarat, India
4 Professor & HOD; Department of Dermatology, SBKS MI & RC, Piparia, Waghodia, Vadodara, Gujarat, India
| Date of Web Publication | 3-Aug-2018 |
Correspondence Address:
M Patel
Resident; Department of Dermatology, SBKS MI & RC, Piparia, Waghodia, Vadodara, Gujarat
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/2347-6486.238520
Angiolymphoid hyperplasia with eosinophilia consists of erythematous papules and nodules involving the head and neck, though the other sites may also be involved. Here we present a case of 50 year old female patient who presented with pruritic papules on the pinna since 6-7 months, which on histopathology revealed features of angiolymphoid hyperplasia. Her lesions were ablated with Carbon dioxide laser in 2 sittings, with no recurrence of lesions over a 1 year follow up period.
How to cite this article:
Patel M, Mahajan R, Ninama K, Bilimoria F E. Angiolymphoid hyperplasia - A case report. J Integr Health Sci 2015;3:40-2 |
How to cite this URL:
Patel M, Mahajan R, Ninama K, Bilimoria F E. Angiolymphoid hyperplasia - A case report. J Integr Health Sci [serial online] 2015 [cited 2023 Mar 22];3:40-2. Available from: https://www.jihs.in/text.asp?2015/3/1/40/238520 |
| Introduction | |  |
Angiolymphoid hyperplasia is a benign lesion composed of vascular channels lined by endothelial cells with pink cytoplasm and vesicular nuclei. It usually affects young adults with lesions predominantly occurring around the head and neck. However they may occur over the trunk and extremities. Spontaneous regression may be seen in smaller lesions, nevertheless surgery, radiotherapy and lasers are useful adjuncts in the treatment. Here we report a case of 50 year old female with skin lesions over her ear, diagnosed as angiolymphoid hyperplasia due to the characteristic histopathology treated with carbon dioxide laser.
Case report:
A 50 year old female patient presented in our OPD with the history of multiple skin lesions over the right pinna since 6 to 7 months. The lesions were spontaneous in onset with gradual increase in the number of lesions. The patient had severe itching over the site of lesions. There was no complaint of pain or constitutional symptoms such as fever, joint pain or weight loss. Examination revealed multiple shiny, coalescent skin coloured and erythematous nodules over the crux of helix, cymba, conchae,and triangular fossa and antihelix of the pinna [Figure 1]a. Scattered papules were also present over the posterior aspect of the right ear [Figure 1]b. There was no local or generalized lymphadenopathy. The lesions were non tender and did not bleed to touch. | Figure 1: (a) skin coloured and erythematous nodules over crux of helix, cymba conchae, triangular fossa and antihelix of pinna (b)Scattered papules over posterior aspect of right ear.
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A complete hemogram revealed normal Hb (12.4 gm.%), total count (9800 cell/cumm), differential count (polymorphs 66%, lymphocytes 20% eosinophils 3% and monocytes 1%). Other indices including liver function tests and renal function tests were also within normal limits. HIV test was negative.
The clinical differential diagnosis considered were Benign cutaneous lymphoid hyperplasia, Kimura’s disease, Angiosarcoma, Lymphocytoma cutis and Angiolymphoid hyperplasia with eosinophilia.
An excision biopsy from a single nodule revealed numerous vascular channels lined by prominent large epithelioid endothelial cells, surrounded by inflammatory cells.[Figure 2]a, [Figure 2]b, [Figure 2]c;thereby confirming the diagnosis of angiolymphoid hyperplasia with eosinophilia. | Figure 2: (a) numerous vascular channels with lobular architecture and prominent inflammatory cell infiltrate,4x (b) numerous vascular channels lined by prominent large pink endothelial cells, 10x (c) prominent endothelial cells with abundant eosinophil
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We used carbon dioxide laser in a superpulse mode (9 watts) to ablate the nodules followed by application of potent topical steroid (clobetasol propionate0.05%cream) once daily for a period of 3 to 4 weeks. The patient was symptom free for 6 months following which she developed a tiny nodule of 2mm in size which was ablated once again[Figure 3]a, [Figure 3]b. Thereafter there has been no recurrence over a 1 year follow up period. | Figure 3: (a) Immediate post-op (b) After 3 months–post inflammatory hypopigmentation
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| Discussion | | |
First described in 1969 by Wells and Whimster[1], angiolymphoid hyperplasia with eosinophilia (AHLE) presents with pink to red brown papules or nodules over the head or neck especially over the ears and scalp. Nodules may merge to form plaques and grape like clusters. Recent reports of this condition are abound with cases occurring over variant sites such as mouth, trunk, arms and genitalia.[2],[3] This condition has a female preponderance with an average age of onset of 32 years. The lesions are often associated with pruritus; they may be painful or even pulsatile.[4] Occasionally, the patients may have regional lymph node enlargement. Blood eosinophilia has been reported in less than 10% of the patients.[5]
The pathogenesis of AHLE has been attributed to trauma and consequent arteriovenous shunting due to the frequent observation of mural damage in intralesional large vessels of AHLE.[6] Histopathology reveals a well circumscribed lesion with vascular hyperplasia involving capillary sized vessels around larger central vessels with a variable perivascular inflammatory infiltrate comprising lymphocytes and eosinophils[7] Enlarged endothelial cells (responsible for the synonym ‘epithelioid hemangioma’) may be seen protruding into lumen of larger vessels producing a scalloped or cobble stone appearance. Differential diagnosis includes pyogenic granuloma, epitheloid hemangioendothelioma, kimura’s disease and Kaposi’s sarcoma. Kimura’s disease sometimes poses diagnostic difficulties; however differentiating points between AHLE and kimura’s disease include a wider age span, male preponderance, extensive lesions with involvement of salivary glands and lymph nodes; histologically lesser vascular hyperplasia, presence of occasional eosinophilic abscesses, lack of prominent epithelial cells and marked fibrosis in the latter.[8]
While spontaneous regression has been reported, surgical excision is usually required. Topical and intralesional steroids, imiquimod and flash lamp pulsed dye laser are reportedly useful in this disorder.[9],[10]
In conclusion the message we would like to convey is that several dermatological conditions involve the ear. Angiolymphoid hyperplasia with eosinophilia is a rare condition; clinical judgement and confirmation through histopathology are mandatory towards the final diagnosis.
| References | | |
| 1. | Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. British Journal of Dermatology. 1969 Jan 1;81(1):1-5.  |
| 2. | William D James, Timothy G Berger, Dirk M Elston. Dermal and Subcutaneous tumors; In Andrews .Dermatology 10 th ed. Saunders; 2006.p. 591 |
| 3. | Bartralot R, Garcia-Patos V, Hueto J, Huguet P, Raspall G, Castblls A. Angiolymphoid hyperplasia with eosinophilia affecting the oral mucosa: report of a case and a review of the literature. British Journal of Dermatology. 1996 Apr 1;134(4):744-8. |
| 4. | Olsen TG, Helwig EB. Angiolymphoid hyperplasia with eosinophilia: a clinicopathologic study of 116 patients. Journal of the American Academy of Dermatology. 1985 May 31;12(5):781-96. |
| 5. | E Calonje. Soft tissue tumors and tumor-like conditions. In: Rooks Tony B, Stephen B , Neil Cox, Christopher G, editors. Dermatology 8 th ed: Blackwell; 2010. p. 56.27 |
| 6. | Eduardo Calonje. Vascular tumors and tumor-like conditions of blood vessels and lymphatics. In: Lever’s David, E Elder, E Rosalie, editors. Dermatology 10 th ed: Wolters Kluwer; 2009 p. 1023. |
| 7. | Christine M. Cole. Cutis. 2013 September; 92(3):110, 117-118. |
| 8. | Gary S Wood. Inflammatory Disease that simulate lymphomas: cutaneous pseudolymphomas. In: Fitzpatrick’s Klaus W, Lowell G, Stephen K, Barbara G, Amy P, David L, editors. Dermatology 7 th ed: McGraw-Hill; 2008.p.1408-1409 |
| 9. | Isohisa T, Masuda K, Nakai N, Takenaka H, Katoh N. Angiolymphoid hyperplasia with eosinophilia treated successfully with imiquimod. International journal of dermatology. 2014 Jan 1;53(1):e43-4. |
| 10. | North PE, Kincannon J. Vascular neoplasm and neoplastic-like proliferations. In: Bolognia JL Jorizzo JL Rapini RP, editors. Dermatology 2 nd ed. St Louis: Mosby; 2008. p. 1774-6 |
[Figure 1], [Figure 2], [Figure 3]
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