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CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 4  |  Page : 450-451
A nodular GLUT1 positive verrucous haemangioma arising over an acral pseudolymphomatous angiokeratoma of children (APACHE): A diagnosis of rarity


From the Department of Dermatology, Venereology and Leprosy, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India

Date of Web Publication2-Nov-2022

Correspondence Address:
Manoharan Krishnaswamy
From the Department of Dermatology, Venereology and Leprosy, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_1127_20

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How to cite this article:
Kulkarni OD, Sangaiah S, Ravi VN, Krishnaswamy M. A nodular GLUT1 positive verrucous haemangioma arising over an acral pseudolymphomatous angiokeratoma of children (APACHE): A diagnosis of rarity. Indian J Dermatol 2022;67:450-1

How to cite this URL:
Kulkarni OD, Sangaiah S, Ravi VN, Krishnaswamy M. A nodular GLUT1 positive verrucous haemangioma arising over an acral pseudolymphomatous angiokeratoma of children (APACHE): A diagnosis of rarity. Indian J Dermatol [serial online] 2022 [cited 2022 Dec 2];67:450-1. Available from: https://www.e-ijd.org/text.asp?2022/67/4/450/360302




Sir,

Verrucous haemangiomas (VHs) are capillary or venous proliferations principally on the extremities. Arising by two years of age, they may involute spontaneously or persist. These GLUT1 positive[1] lesions are commonly thought to be a reactive phenomenon to several insults thereby explaining the adult onset in a few cases.

Acral pseudolymphomatous angiokeratoma of children (APACHE) first described by Ramsay et al.,[2] is a rare entity of unknown aetiology which presents as erythematous or mildly pigmented, single or multiple papules or keratotic plaques on distal extremities. Despite what the terminology implies it can occur in extra-acral sites and also in adults, thereby supporting a proposed terminology 'papular angiolymphoid hyperplasia'.

A 20-year-old female presented with a pigmented and eroded verrucous nodule on the left leg above the medial malleolus for six months [Figure 1]a. She did not complain of lesions elsewhere. There were no known systemic associations. It began as a pigmented flat patch at birth, over which few keratotic papules gradually developed in the following years. After a trivial trauma to the site six months ago, the lesion rapidly evolved into a pigmented, eroded verrucous nodule that bleeds on touch. On examination the pigmented lesion measured 2 × 3 cm with a verrucous surface and superficial erosions. The perilesional area showed intense pigmentation [Figure 1]b. Spontaneous discharge was absent. On manipulation, the lesion bled mildly. Absence of warmth, tenderness, pulsatility, and localized lymphadenopathy was noted. Clinical differential diagnoses were pyogenic granuloma, angiokeratoma, VH, nodular melanoma, Kaposi's sarcoma, and cornu cutaneum over a keratoacanthoma. On excision biopsy the lesion bled profusely indicating a vascular aetiology. Histopathology revealed pseudo-epitheliomatous hyperplasia, marked vascular proliferation in the entire dermis, and a massive aggregate of lymphocytes and histiocytes in a band like fashion with moderate numbers of interspersed plasma cells and eosinophils present peri-vascularly [Figure 2]a, [Figure 2]b and [Figure 3]a, [Figure 3]b. A positive GLUT1 on immunohistochemistry coupled with histopathological features favoured a diagnosis of VH [Figure 4]a, [Figure 4]b; however, the type and arrangement of the inflammatory cell infiltrate (which included a confluent band like dermal infiltrate composed of lymphocytes, histiocytes, plasma cells, and eosinophils, aggregates of lympho-histiocytes in a pseudolymphomatous pattern, and proliferation of blood vessels[3]) pointed towards the coexistence of a rarer entity named 'APACHE)', which characteristically presents with coinciding histopathology and is considered as a pseudolymphoma per se as per published reports.[4]
Figure 1: (a) Clinical photograph of the verrucous nodule seen on the left lower limb, 2 cm above the medial malleolus. (b) Enlarged photograph of the nodule with the pigmented, eroded, and verrucous appearance

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Figure 2: (a) (40×; H and E stained) Histopathology showing prominent hyperkeratosis, irregular acanthosis, and papillomatosis (pseudo-epitheliomatous hyperplasia), presence of vascular proliferations in the dermis along with an intense inflammatory infiltrate. (b) (40×; H and E stained) Presence of dense inflammatory infiltrate in a pseudolymphomatous lichenoid pattern with vascular proliferations

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Figure 3: (a) (400×; H and E stained) Presence of numerous plasma cells amidst a dense inflammatory infiltrate of lympho-histiocytes. (marked with white arrows). (b) (400x; H and E stained) Presence of numerous eosinophils within the vascular proliferations (marked with blue arrows)

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Figure 4: (a and b) (40 × and 400 × respectively; IHC staining) Positive GLUT1 staining

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We hence conclude by stating this is a singular case of a reactive GLUT1 positive VH developing over a pre-existing APACHE.

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.
Laun K, Laun J, Smith D. Verrucous hemangioma. Eplasty 2019;19:ic1.  Back to cited text no. 1
    
2.
Ramsay B, Dahl MC, Malcolm AJ, Wilson-Jones E. Acral pseudolymphomatous angiokeratoma of children. Arch. Dermatol 1990;126:1524-5.  Back to cited text no. 2
    
3.
Kaddu S, Cerroni L, Pilatti A, Soyer HP, Kerl H. Acral pseudolymphomatous angiokeratoma: a variant of the cutaneous pseudolymphomas. Am J Dermatopathol 1994;16:130-3.  Back to cited text no. 3
    
4.
Kiyohara T, Kumakiri M, Kawasaki T, Takeuchi A, Kuwahara H, Ueda T. Linear acral pseudolymphomatous angiokeratoma of children (APACHE): further evidence that APACHE is a cutaneous pseudolymphoma. J Am Acad Dermatol 2003;48:S15-7.  Back to cited text no. 4
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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