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 Table of Contents  
LETTER TO EDITOR
Year : 2020  |  Volume : 7  |  Issue : 1  |  Page : 54-55

A case of papular acantholytic dyskeratosis in a young male


Venkat Centre for Advanced Dermatology and Postgraduate Training Institute, Bengaluru, Karnataka, India

Date of Submission23-Nov-2019
Date of Decision19-Mar-2019
Date of Acceptance11-Apr-2020
Date of Web Publication02-Jun-2020

Correspondence Address:
Venkataram Mysore
Venkat Centre for Advanced Dermatology and Postgraduate Training Institute, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdpdd.ijdpdd_57_19

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How to cite this article:
Jindal A, Mysore V, Noronha M. A case of papular acantholytic dyskeratosis in a young male. Indian J Dermatopathol Diagn Dermatol 2020;7:54-5

How to cite this URL:
Jindal A, Mysore V, Noronha M. A case of papular acantholytic dyskeratosis in a young male. Indian J Dermatopathol Diagn Dermatol [serial online] 2020 [cited 2020 Nov 26];7:54-5. Available from: https://www.ijdpdd.com/text.asp?2020/7/1/54/285803



Sir,

Papular acantholytic dyskeratosis (PAD) is a distinct clinicopathological entity characterized by localized papular lesions over genitocrural or anogenital folds exhibiting acantholysis with dyskeratotic cells and intraepidermal clefts throughout the epidermis. Chorzelskiet al. in 1984 reported the first case of PAD with multiple, dome-shaped papules over the labia majora which on histology showed features of acantholytic dyskeratotic disease.[1]

A 38-year-old male presented to the dermatology outpatient department with complaints of spontaneous appearance of occasionally itchy papules, gradually progressive in number over the perineum for 8 months. Family history was unremarkable. The patient was married since 7 years with no history of similar complaints in the partner and denied of any history suggestive of sexually transmitted diseases. Examination revealed multiple grayish, flat-topped to dome-shaped papules concentrated over the groin folds extending over to the perineum [Figure 1]. A biopsy specimen of 3.5 mm punch was obtained from one of the papular lesions for histopathology with a differential diagnosis of verrucae, lichen planus, or candidiasis. The patient was given a trial of topical antifungals till histopathology report was awaited.
Figure 1: A magnified image at presentation showing multiple grayish white dome-xsshaped to flat-topped papules

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Histopathology showed partial to complete but focal acantholysis mainly concentrated in the stratum spinosum and to a lesser extent to stratum granulosum with the presence of corps ronds and grains in vicinity and stratum corneum showing hyperkeratosis and parakeratosis [Figure 2] and [Figure 3]. Clinical and histopathological features suggested a diagnosis of PAD. The patient denied radiofrequency as lesions were asymptomatic. The patient reported more than 50% improvement with flattening of papules with 14 days of use of topical antifungals [Figure 4].
Figure 2: Hypergranulosis with acantholysis (yellow arrow) limited mainly to stratum spinosum at ×200 (a) and ×400 (b)

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Figure 3: Acantholysis with dyskeratosis in the upper part of the epidermis. Red arrow shows grain (elongated nucleus with eosinophilic cytoplasm) and red circle shows acantholytic cells with spiny (”acanth”) projections (×400)

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Figure 4: At 2-week follow-up after use of topical sertaconazole

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The concept of focal acantholytic dyskeratosis (FAD) was given by Ackerman in 1972, where he divided diverse clinical conditions with unifying histopathological features of focal acantholysis along with dyskeratosis into three subtypes under the heading of FAD.[1]

PAD is not one of the very well-recognized acantholytic disorders due its rare incidence with only around 30 cases reported in literature over the last two decades. It is not limited to any age group; cases have been described from 8 years of age to 82 years as well.

Warts, lichen planus, and candidiasis were considered to be the differential diagnosis in our case, similar to previous studies and reports where the first impression was either warts, Hailey–Hailey disease, lichen planus, or candidiasis (intertrigo).[2] Although there are few reports of atypical clinical presentation of PAD such as lichen sclerosus which was confirmed on histology as PAD.[3]

PAD is most commonly reported to occur on the labia majora or crural folds, which implies that moist areas might be serving as a conducive environment for propagation of these lesions and antifungals might be of help as seen in our patient as well.[4] PAD has also been reported to occur over the lips and chest, with or without coexistence of lesions over the genitocrural area.[5]

Options for the management of PAD include topical steroids; oral retinoids; 0.1% tazarotene; cidofovir; imiquimod; 3% boric acid; and physical modalities such as cryotherapy, ablative laser, and radiofrequency.

To conclude, PAD is a distinct clinicopathological entity characterized by multiple, dome-shaped papules over genitocrural folds and presence of acantholysis with dyskeratoses on histology akin to transient acantholytic disorders. It is important to be familiar with this entity to avoid misdiagnosis and unnecessary distress to patient.

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.
Chorzelski TP, Kudejko J, Jablonska S. Is papular acantholytic dyskeratosis of the vulva a new entity? Am J Dermatopathol. 1984;6:557-60.  Back to cited text no. 1
    
2.
Dowd ML, Ansell LH, Husain S, Grossman ME. Papular acantholytic dyskeratosis of the genitocrural area: A rare unilateral asymptomatic intertrigo. JAAD Case Rep 2016;2:132-4.  Back to cited text no. 2
    
3.
Haddadeen C, Theaker J, Rowen D, Lotery H. Acantholytic dyskeratosis of the vulva presenting with clinical features of vulval lichen sclerosus – A possible rare collision entity. J Cutan Pathol 2020;47:61-4.  Back to cited text no. 3
    
4.
Cooper PH. Acantholytic dermatosis localized to the vulvocrural area. J Cutan Pathol 1989;16:81-4.  Back to cited text no. 4
    
5.
Lee JH, Kim YC, Lew W. A case of focal acantholytic dyskeratosis occurring on both the lip and the anal canal. Yonsei Med J 2003;44:166-8.  Back to cited text no. 5
    


    Figures

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



 

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