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ARTICLE IN PRESS
doi:
10.25259/IJDVL_970_2022

Acantholytic dyskeratotic acanthoma: A rare and underappreciated entity

Department of Dermatology, College of Medicine, Seoul St. Mary’s Hospital, The Catholic University of Korea, Seoul, South Korea
Corresponding author: Prof. Chul Hwan Bang, Department of Dermatology, College of Medicine, Seoul St. Mary’s Hospital, The Catholic University of Korea, Seoul, South Korea, mrbangga@gmail.com
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This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: Doh JY, Lee JH, Bang CH. Acantholytic dyskeratotic acanthoma: A rare and underappreciated entity. Indian J Dermatol Venereol Leprol doi: 10.25259/IJDVL_970_2022

A 64-year-old man presented with a slightly painful plaque on the neck for 6–7 months [Figure 1a]. He had no other underlying disease except osteoarthritis and denied any family history of skin disease. Clinical examination revealed a 1.4 cm-sized erythematous-to-brownish crusted plaque on the neck. Dermoscopy findings showed a central mass of keratin surrounded by linear-irregular vessels [Figure 1b].

Figure 1a:: A solitary, 1.4 cm-sized, erythematous to brownish plaque with crusts on the neck
Figure 1b:: Dermoscopy showed a central mass of keratin surrounded by linear-irregular vessels

An excisional biopsy was performed for treatment and diagnosis under suspicion of inflamed seborrheic keratosis, squamous cell carcinoma and basal cell carcinoma. Histopathological findings showed acanthosis, parakeratosis, dyskeratosis and acantholysis involving a full epidermal layer. Superficial perivascular lymphocytic infiltration in the dermis was present without follicular involvement [Figures 2a and 2b]. After excision, the lesion healed without a sign of recurrence.

Figure 2a:: Histopathological findings showed acanthosis and dyskeratosis with full epidermal acantholysis in the epidermis. Superficial perivascular lymphocytic infiltration in the dermis is also observed (H & E, original magnification x40)
Figure 2b:: Rounded eosinophilic dyskeratotic cells (corps ronds) and hyperkeratotic and flattened parakeratotic cells (grains) in the cornified layer are also observed (H & E, x200)

Question

What is your Diagnosis?

Answer

Diagnosis: Acantholytic dyskeratotic acanthoma

Discussion

Acanthoma is a benign tumor of epidermal keratinocytes, showing a broad range of histological patterns.1 Acantholytic dyskeratotic acanthoma is a relatively uncommon variant of acanthoma, classified within the past decade. The term ‘focal acantholytic dyskeratosis’ was first used by Ackerman to describe incidental lesions showing acantholysis and dyskeratosis.2 In contrast, genital lesions with similar characteristics had been described as ‘papular acantholytic dyskeratoma’ or ‘papular acantholytic dyskeratosis’.3,4 However, non-genital lesions showing acantholysis and dyskeratosis were not adequately identified until Omulecki et al. defined acantholytic dyskeratotic acanthoma for the first time in 2007.1 Acantholytic dyskeratotic acanthoma refers to a solitary, non-genital lesion with prominent acantholysis and dyskeratosis without cup-shaped architecture or follicular involvement.4,6,7 However, whether it is an actual distinct diagnostic entity is debatable. There is no statement from the World Health Organization, and the description of acantholytic dyskeratotic acanthoma differs even within textbooks. Lever’s histopathology and McKee’s pathology textbooks describe acantholytic dyskeratotic acanthoma as a phenomenon rather than a separate entity,8,9 while Weedon’s textbook considers acantholytic dyskeratotic acanthoma a separate entity.10

About 43 cases of acantholytic dyskeratotic acanthoma have been reported in the English literature to date [Table 1]. Patient ages ranged from 12 to 97 years, with 38 (88.4%) older than 40 years. The patients were often clinically suspected of having basal cell carcinoma, squamous cell carcinoma, and/or actinic keratosis. Most lesions were located on the trunk and often presented as a small papule of <1 cm. Among the 43 reported cases, three were in subungual areas,1315 three occurred in a transplant setting5,12,13 and one after vemurafenib treatment.14 Among the three patients with a history of transplants, two presented with multiple lesions. However, the association between immunosuppression and acantholytic dyskeratotic acanthoma remains inconclusive.

Table 1:: Clinical characteristics of acantholytic dyskeratotic acanthoma cases reported in the English literature
Year Author Age Gender Number/size (mm) Site Clinical diagnosis Duration Medical history
2007 Omulecki et al.1 64 Male 1/20 x 50 Back (a) 7 years DM
2008 Ko et al.4 48 Female 1/7 Ankle BCC (b) (a)
40 Female 1/5 Forearm AK/SCC (b) (a)
45 Female 1/4 Back (a) (b) (a)
64 Female 1/4 Back BCC (b) (a)
48 Female 1/5 Abdomen BCC/SCC (b) (a)
49 Female 1/6 Chest BCC/SCC (b) (a)
43 Female 1/3 Chest BCC (b) (a)
63 Male 1/7 Back BCC/BD (b) (a)
50 Female 1/4 Chest SK (b) (a)
39 Female 1/7 Back Wart (b) (a)
52 Female 1/6 Back SCC (b) (a)
43 Male 1/5 Abdomen BCC (b) (a)
45 Female 1/4 Abdomen SK/AK (b) (a)
49 Female 1/5 Nevus BCC (b) (a)
57 Male 1/2 Chest BCC/AK (b) (a)
63 Male 1/5 Clavicle AK/BCC/SCC (b) (a)
84 Male 1/9 Thigh BD (b) (a)
48 Female 1/4 Flank SK/AK (b) (a)
58 Male 1/7 Sternum BCC (b) (a)
60 Male 1/3 Back BCC (b) (a)
68 Male 1/6 Flank SCC (b) (a)
57 Female 1/6 Chest Papilloma (b) (a)
51 Female 1/5 Shoulder BCC (b) (a)
64 Male 1/3 Chest Nevus (b) (a)
75 Male 1/5 Lower leg BCC (b) (a)
76 Female 1/5 Neck SCC (b) (a)
47 Male 1/7 Back Nevus (b) (a)
56 Female 1/5 Chest BCC (b) (a)
2009 Sass et al.15 53 Male 1/5 Right thumbnail (a) (b) (a)
15 Male 1/3 Right thumbnail Onychopapilloma 6 months (a)
12 Female (a) Right thumbnail Onychopapilloma 9 months (a)
2013 Park et al.16 42 Female Multiple/2-3 Face (a) Several years(b) Associated with rosacea
2013 Pezzolo et al.12 49 Male 1/4 x 4 Lower leg BCC/AK 3 years Kidney allograft patient on immunosuppression
2014 Goldenberg et al.3 72 Female 1/10 x 4 Chest (a) 3 months No other illness
2017 Kim et al.17 38 Female 1/(a) Face (a) 1 month Associated with DLE
2018 Burgler et al.13 60 Male Multiple/(a) Back and lateral chest wall (a) 1 week Heart transplant patient on immunosuppression
2019 Kanitakis et al.5 74 Male Multiple/(a) Back (a) (b) Liver transplant patient on immunosuppression
2020 Nandakumar et al.6 75 Female 2/13 x 15,10 x 12 Thigh Chromoblastomycosis, SCC, tuberculosis verrucosa cutis, viral wart, lupus vulgaris 60 years No other illness
2017 Komori et al.14 62 Female 1/(a) Right shoulder SCC (b) Metastatic melanoma on vemurafenib
2017 Ng et al.18 25 Male 1/(a) Right thumbnail Onychopapilloma, onychomatricoma or a verruca 1 year No other illness
2021 Tanaka et al.19 97 Female 1/10 Right thigh Verruca vulgaris (b) (a)
2022 Current case 64 Male 1/14 Neck Inflamed SK, SCC, BCC 6–7 months No other illness

a): Not available, b): exact duration, not given, AK: actinic keratosis, BCC: basal cell carcinoma, BD: Bowen’s disease, DLE: discoid lupus erythematosus, DM: diabetes mellitus, SCC: squamous cell carcinoma, SK: seborrheic keratosis

Pathogenesis of acantholytic dyskeratotic acanthoma is still elusive. However, genetic and immunological factors along with viral infections, physical stimuli, and sunlight exposure, might play a role.3,10 Histopathologically, acantholytic dyskeratotic acanthoma is characterised by acantholysis and dyskeratosis.3,6 Therefore, it should be differentiated from other acantholytic disorders such as keratosis follicularis (Darier’s disease), transient acantholytic dermatosis (Grover’s disease), Hailey-Hailey disease, and warty dyskeratoma. As Darier’s, Grover’s and Hailey-Hailey’s diseases show distinct clinical features and immunofluorescence findings, they could be easily differentiated from acantholytic dyskeratotic acanthoma.9 On the contrary, it could be challenging to distinguish acantholytic dyskeratotic acanthoma from warty dyskeratoma as they share many histological features. However, unlike acantholytic dyskeratotic acanthoma, warty dyskeratoma is associated with a cup-shaped invagination or follicular involvement.1,3,4,6,11 Last, acantholytic dyskeratotic acanthoma can also be easily differentiated from acantholytic acnathoma by the presence of prominent dyskeratosis.

Clinically, patients with acantholytic dyskeratotic acanthoma typically present with an asymptomatic solitary keratotic papule or plaque showing predilection to the trunk,4,5 and resemble non-melanotic skin cancers such as squamous cell carcinoma, basal cell carcinoma or actinic keratosis.4,5 However, acantholytic dyskeratotic acanthoma is a benign disease that can be easily treated through excision. Therefore, it is necessary to rule out acantholytic dyskeratotic acanthoma from other conditions to avoid unnecessary procedures and treatments. It can be relatively easily differentiated from other non-melanotic skin cancers histopathologically as it lacks cellular atypia and specific morphological features of those conditions.1,3

Our case reinforces the necessity of classifying acantholytic dyskeratotic acanthoma as a distinct clinical entity. Examining and ruling out acantholytic dyskeratotic acanthoma as a clinical entity will assist in avoiding unnecessary or invasive procedures and treatments associated with another malignant disease with similar characteristics.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent.

Financial support and sponsorship

Nil

Conflict of interest

There is no conflict of interest

References

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