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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 1  |  Page : 16-18

Angiolymphoid hyperplasia with eosinophilia: Simultaneous presence of lesions of two different morphologies


1 Department of Dermatology, Venereology and Leprosy, IGMC, Shimla, Himachal Pradesh, India
2 Department of Patholog, IGMC, Shimla, Himachal Pradesh, India

Date of Submission04-Apr-2019
Date of Decision28-Aug-2019
Date of Acceptance28-Oct-2019
Date of Web Publication02-Jun-2020

Correspondence Address:
Mudita Gupta
Department of Dermatology, Venereology and Leprosy, IGMC, Shimla, Himachal Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdpdd.ijdpdd_18_19

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  Abstract 


Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare vascular tumor which presents as a dermal papule or a nodule over the head-and-neck region. Histopathologically, there is vascular proliferation in the upper dermis and diffuse lymphocytic infiltrate with eosinophilia. Kimura's disease is the closest differential of ALHE, which presents as subcutaneous nodules. We report a case of ALHE presenting with a violaceous papule overlying a subcutaneous nodule, histopathologically; both lesions were diagnosed as ALHE.

Keywords: Angiolymphoid hyperplasia with eosinophilia, simultaneous, two different morphologic types


How to cite this article:
Sharma RK, Gupta M, Gulati A, Sood S. Angiolymphoid hyperplasia with eosinophilia: Simultaneous presence of lesions of two different morphologies. Indian J Dermatopathol Diagn Dermatol 2020;7:16-8

How to cite this URL:
Sharma RK, Gupta M, Gulati A, Sood S. Angiolymphoid hyperplasia with eosinophilia: Simultaneous presence of lesions of two different morphologies. Indian J Dermatopathol Diagn Dermatol [serial online] 2020 [cited 2020 Aug 14];7:16-8. Available from: http://www.ijdpdd.com/text.asp?2020/7/1/16/285793




  Introduction Top


Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular tumor of unknown etiology, which occurs more commonly in middle-aged females. It was first described by Wells and Whimster in 1969.[1] ALHE usually occurs over the head-and-neck region, especially auricular; however, other sites such as trunk and limbs may be involved. Kimura's disease presents as subcutaneous nodules with lymphoid hyperplasia. The association of Kimura's disease with ALHE is a subject of debate; few authors consider them to be different presentation of same disease, whereas others have differentiated them on the basis of various clinical and histopathological features.[2] We report a case of ALHE with a violaceous papule overlying a subcutaneous nodule over the left lateral aspect of the forehead in the same patient.


  Case Report Top


A 21-year-old female presented with a swelling over the left side of the forehead for 3 months. The swelling increased in size very slowly; since 1 month, she noticed a reddish brown growth on one side of the swelling. She denied any history of trauma or any other medical illness. There was no history of pain, pruritus, or bleeding on touch. On examination, there were two lesions: a violaceous papular lesion of size about 0.7 cm × 0.8 cm with smooth surface and a subcutaneous nodular lesion of size about 1.5 cm × 1 cm. Both the lesions were nontender and soft in consistency [Figure 1]. Routine investigations including blood counts (eosinophil) and biochemistry were normal. Excision of the papule was done which on histopathology showed unremarkable epidermis, proliferating capillaries in the papillary dermis with epitheloid endothelial cells, and intense perivascular infiltrate of lymphocytes and eosinophils [Figure 2]a. The subcutaneous lesion was excised after obtaining histopathology report of the papule as it appeared clinically different from the violaceous papular lesion. No margins beyond the lesion were excised. Histopathology of the nodular lesion showed a normal epidermis and a dermis; there was proliferation of capillaries in the septa of subcutaneous tissue with perivascular infiltrate of few lymphocytes and plenty of eosinophils [Figure 2]b. The perivascular infiltrate in both the lesions consisted of lymphocytes and many eosinophils [Figure 2]c. Both the biopsies were consistent with ALHE. After 6 months of follow-up, there was no recurrence.
Figure 1: Simultaneous presence of an erythematous to violaceous papule and a subcutaneous nodule on the left side of the forehead

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Figure 2: (a) (H and E, ×40) Histopathological examination of the papular lesion showing subepithelial proliferation of blood vessels surrounded by inflammatory infiltrate. (b) (H and E, ×40) Histopathological examination of the subcutaneous nodule showing proliferation of blood vessels in deeper dermis surrounded by inflammatory infiltrate. (c) (H and E, ×100) Proliferating vascular channels lined by prominent endothelial cells surrounded by lymphocytes and many eosinophils

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  Discussion Top


ALHE is also known as epitheloid hemangioma. It is a benign vascular proliferation of unknown etiology. It can present in a wide age range, however more commonly seen in females aged 20–50 years.[2],[3] Clinically, it is characterized by papules or nodules of angiomatoid aspect varying in color from violaceous to red brown. Depending on the vessels involved, the presentation may vary; the involvement of vessels in subcutaneous layer may lead to a nodular lesion with no surface changes. The sites predominantly involved are head and neck, especially around the ears or around the hairline. Oral mucosal involvement has also been reported.[4]

Initially, Kimura's disease was considered an eosinophilic disorder under the same spectrum of ALHE; however, now it is considered a distinct entity clinically and histopathologically.[2],[5] Kimura's disease presents as subcutaneous nodules with no overlying cutaneous lesions. Extracutaneous manifestations in the form of lymphadenopathy and renal involvement may be present, and blood eosinophilia is more common as compared to ALHE. Histopathologically, Kimura's disease shows a marked follicular hyperplasia surrounded by eosinophils and less neoangiogenesis.[5] On the contrary, there are reports of coexistence of Kimura's disease and ALHE.[6] In our case, there was a cutaneous violaceous lesion suggestive of ALHE overlying a subcutaneous nodular lesion, clinically simulating Kimura's disease; however, histopathologically, both were suggestive of ALHE.

The pathogenesis of ALHE is not clear; however, female predominance suggests some hormonal causes. Some authors have claimed vascular trauma, infections (human herpesvirus -8 and human T-cell lymphotropic virus), and vascular malformations; however, reactive vascular hyperplasia to various stimuli is the most accepted hypothesis.[2]

The diagnosis of ALHE is based on histopathology. There is proliferation of small-to-medium-sized blood vessels, often showing a lobular architecture, many of which are lined by prominent endothelial cells with an epithelioid appearance. There is diffuse infiltration by lymphocytes and eosinophils. The endothelial cells stain positive for CD34, von Willebrand factor, and CD31.[5]

Differential diagnoses include angiosarcoma and bacillary angiomatosis.[7] Angiosarcoma is ruled out by the absence of cellular atypia, and the absence of bacilli and neutrophilic inflammation rules out bacillary angiomatosis.

The treatment of choice for ALHE is surgical excision, but recurrence is common. Cryotherapy, local radiotherapy, topical or intralesional corticosteroids, topical tacrolimus, imiquimod, acitretin, and laser (dye laser and CO2 laser) are also reported as possible treatments with varying success levels.[8],[9] There are reported cases of spontaneous regression.[8],[9]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Das A, Ghosh A, Gharami RC. Angiolymphoid hyperplasia with eosinophilia: Report of two cases in children. Indian J Paediatr Dermatol 2016;17:287-9.  Back to cited text no. 1
  [Full text]  
2.
Bastos JT, Rocha CR, Silva PM, Freitas BM, Cassia FF, Avelleira JC. Angiolymphoid hyperplasia with eosinophilia versus Kimura's disease: A case report and a clinical and histopathological comparison. An Bras Dermatol 2017;92:392-4.  Back to cited text no. 2
    
3.
Mahajan R, Kanwar AJ, Kumar M, Saikia UN, Rajwanshi A, Dogra S. Multiple nodules on the scalp in segmental distribution. Int J Dermatol 2016;55:387-9.  Back to cited text no. 3
    
4.
Tenório JR, Gonzaga AK, Gonçalves PG, Pereira de Oliveira DH, Queiroz LM. Angiolymphoid hyperplasia with eosinophilia: A rare case in the oral cavity. J Vasc Bras 2016;15:317-21.  Back to cited text no. 4
    
5.
Calonje E. Soft-tissue tumours and tumour-like conditions. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8th ed. West Sussex: Wiley Blackwell Publishers; 2010. p. 56, 27-8.  Back to cited text no. 5
    
6.
Chong WS, Thomas A, Goh CL. Kimura's disease and angiolymphoid hyperplasia with eosinophilia: Two disease entities in the same patient: Case report and review of the literature. Int J Dermatol 2006;45:139-45.  Back to cited text no. 6
    
7.
Kurihara Y, Inoue H, Kiryu H, Furue M. Epithelioid hemangioma (angiolymphoid hyperplasia with eosinophilia) in zosteriform distribution. Indian J Dermatol 2012;57:401-3.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Alaidarous A, Bouissou X, Mazereeuw-Hautier J, Bulai-Livideanu C, Lamant L, Paul C. Angiolymphoid hyperplasia with eosinophilia treated with low-dose methotrexate. JAAD Case Rep 2015;1:342-4.  Back to cited text no. 8
    
9.
Wang S, Li W. Angiolymphoid hyperplasia with eosinophilia successfully treated with tacrolimus ointment. J Eur Acad Dermatol Venereol 2010;24:237.  Back to cited text no. 9
    


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