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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 1  |  Page : 20-22

Pseudoepitheliomatous keratotic and micaceous balanitis: A distinct entity


1 Dermatologic Surgicenter, Philadelphia, PA, USA
2 Department of Dermatology, Saint George Hospital University Medical Center, Beirut, Lebanon
3 Faculty of Medicine, Lebanese University, Beirut, Lebanon
4 Department of Dermatology, Saint George Hospital University Medical Center, Beirut, Lebanon; Department of Dermatology, Faculty of Medicine, Balamand University, Beirut, Lebanon

Date of Submission01-Sep-2020
Date of Decision13-Feb-2021
Date of Acceptance09-Jun-2021
Date of Web Publication11-Aug-2021

Correspondence Address:
Antoine Salloum
Dermatologic Surgicenter, Philadelphia, PA.
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdpdd.ijdpdd_93_20

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  Abstract 

Pseudoepitheliomatous keratotic and micaceous balanitis (PKMB), an uncommon glans penis skin disorder, affects mainly elderly men and can progress to verrucous carcinoma or invasive squamous cell carcinoma. A 22-year-old male presented with a 5-year history of a slightly pruritic thick scaly plaque on the glans penis that appeared 2 months after undergoing circumcision. Physical examination revealed a well-defined hyperkeratotic plaque with thin mica-like scales. Histological examination of previous biopsies showed acanthosis with elongation of the rete ridges, prominent granular cell layer, and marked orthokeratotic hyperkeratosis. The diagnosis of plaque-stage PKMB was made. The patient had monthly sessions of topical liquid nitrogen and after 20 weeks, the plaque shrank significantly.

Keywords: Enile lesion, penile cancer, PKMB


How to cite this article:
Salloum A, Bachour J, Bazzi N, Megarbane HA. Pseudoepitheliomatous keratotic and micaceous balanitis: A distinct entity. Indian J Dermatopathol Diagn Dermatol 2021;8:20-2

How to cite this URL:
Salloum A, Bachour J, Bazzi N, Megarbane HA. Pseudoepitheliomatous keratotic and micaceous balanitis: A distinct entity. Indian J Dermatopathol Diagn Dermatol [serial online] 2021 [cited 2021 Dec 2];8:20-2. Available from: https://www.ijdpdd.com/text.asp?2021/8/1/20/323706




  Key Messages: Top


PKMB is a disorder of the glans penis affecting mainly elderly males. This is a rare case of PKMB occurring in a young male. The patient was successfully treated with topical liquid nitrogen.


  Introduction Top


Pseudoepitheliomatous keratotic and micaceous balanitis (PKMB) is an uncommon premalignant disorder affecting elderly males and targets the glans penis with a possible progression to verrucous carcinoma or invasive squamous cell carcinoma (SCC). In 1961, Lortat-Jacob and Civatte were the first to report a thick diffuse form of hyperkeratosis on the glans penis,[1] along with mica-like scaly layers.[2]


  Case History Top


A 22-year-old male presented with a 5-year history of a slightly pruritic thick scaly plaque on the glans penis that appeared 2 months after undergoing circumcision [Figure 1]. The patient was otherwise healthy and not on any medical treatment. There was no history of exposure to sexually transmitted infections, history of trauma, or systemic diseases. He was previously biopsied three times with no change in the result, with an inconclusive diagnosis, and managed as hypertrophic lichen sclerosus (LSc) with minimal improvement upon using topical corticosteroids.
Figure 1: Patient glans penis before the treatment

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Physical examination revealed a well-defined hyperkeratotic plaque with thin mica-like scales, measuring 3 × 4 cm over the glans penis. He had a normal penile shaft and scrotum. There was no regional lymphadenopathy.

Differential diagnoses included PKMB, SCC, verrucous carcinoma, hypertrophic LSc, genital warts, and penile intraepithelial neoplasia.

Histological review of previous biopsies showed acanthosis with elongation of the rete ridges, prominent granular cell layer, and marked orthokeratotic hyperkeratosis [Figure 2] and [Figure 3]. The lamina propria showed discrete perivascular lymphocytic cell infiltrate. There was no evidence of koilocytosis, atypia, or dyskeratosis. Periodic acid-Schiff was negative for fungal organisms.
Figure 2: H&E stain with high power magnification

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Figure 3: H&E stain with 10× magnification showing acanthosis with elongation of the rete ridges (epithelial extension into the underlying connective tissue), prominent granular cell layer, and marked orthokeratotic hyperkeratosis. The lamina propria showed lymphocytic cell infiltrate

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The diagnosis of plaque-stage PKMB was made. The patient had monthly sessions of topical liquid nitrogen. After 20 weeks, there was a significant reduction in the size of the plaque [Figure 4], The patient is currently under follow-up surveillance for malignancy.
Figure 4: Mild improvement after 20 weeks of liquid nitrogen

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  Discussion Top


This rare condition first described in 1961 by Lortat-Jacob and Civatte is of unknown etiology. Earlier reports included elderly patients (over 60 years),[2],[3] but the condition has been recently reported in a younger target population.[4],[5] Similar to our patient’s case, most reports involved cases of phimosis or post-circumcision. Moreover, two patients diagnosed with PKMB suffered from progression to verrucous carcinoma, and further advancement into invasive SCC was seen in one of the cases.[2]

Progression of PKMB can be characterized by four manifestations: starting with (i) the initial plaque stage, to the (ii) late tumor stage, followed by a (iii) developing verrucous tumor on the plaque and finally, the appearance (iv) of balano-preputial synechiaes.[6]

Histopathologic findings primarily encompass the presence of acanthotic masses, marked hyperkeratosis, parakeratosis, retes ridges, perivascular lymphocytic infiltration, and the absence of atypia and mitotic figures. However, band-like lymphocytic infiltration is seen in Lichen planus and lichen sclerosus. Rete ridges are uncommon in lichen sclerosus but common in PKMB.[7]

Also, fibro-hyaline epithelial bands with elastic refraction on orcein are present in lichen sclerosus and absent in PKMB.[8]

In the literature search, PKMB was not associated with infections nor inguinal lymphadenopathy.

PKMB is not a tumor-free disease, thus it should be diagnosed and treated early. The following late tumor stage involves an increase in hyperplasia, evolving to deep bulbous squamous outgrowths with marked pleomorphism, increased cell density, and marked basal cell activity.[9]

Upon review of the literature to date, verrucous carcinoma occurred in 6 out of the 20 reported cases, and 4 cases developed SCC, out of which 2 had progressed from verrucous carcinoma to invasive SCC.[9]

Treatment modalities include 5% fluorouracil, radiotherapy, cryotherapy, and local excision with possibility of recurrence. Even though topical therapy is optimal treatment against the initial plaque stage,[10] a more aggressive therapy approach such as wide local excision is required for advanced stages of the penile condition.[6] Long-term surveillance for possible malignant transformation is recommended for all cases of PKMB.

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.
Lortat-Jacob E, Civatte J. [Micaceous and keratotic pseudo-epitheliomatous balanitis]. Bull Soc Fr Dermatol Syphiligr 1961;68:164-7.  Back to cited text no. 1
    
2.
Beljaards RC, van Dijk E, Hausman R. Is pseudoepitheliomatous, micaceous and keratotic balanitis synonymous with verrucous carcinoma? Br J Dermatol 1987;117:641-6.  Back to cited text no. 2
    
3.
Bart RS, Kopf AW. On a dilemma of penile horns: Pseudoepitheliomatous, hyperkeratotic and micaceous balanitis? J Dermatol Surg Oncol 1977;3:580-2.  Back to cited text no. 3
    
4.
Pai VV, Hanumanthayya K, Naveen KN, Rao R, Dinesh U. Pseudoepitheliomatous, keratotic, and micaceous balanitis presenting as cutaneous horn in an adult male. Indian J Dermatol Venereol Leprol 2010;76:547-9.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Das S, Ghoshal L. Pseudoepitheliomatous keratotic and micaceous balanitis of civatte. Indian Dermatol Online J 2014;5:148-50.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Krunic AL, Djerdj K, Starcevic-Bozovic A, Kozomara MM, Martinovic NM, Vesic SA, et al. Pseudoepitheliomatous, keratotic and micaceous balanitis. Case report and review of the literature. Urol Int 1996;56:125-8.  Back to cited text no. 6
    
7.
Fung MA, LeBoit PE. Light microscopic criteria for the diagnosis of early vulvar lichen sclerosus: A comparison with lichen planus. Am J Surg Pathol 1998;22:473-8.  Back to cited text no. 7
    
8.
Dauendorffer JN, Cavelier Balloy B, Bagot M, Renaud-Vilmer C. [Penile squamous hyperplasia]. Ann Dermatol Venereol 2018;145:72-5.  Back to cited text no. 8
    
9.
Perry D, Lynch PJ, Fazel N. Pseudoepitheliomatous, keratotic, and micaceous balanitis: Case report and review of the literature. Dermatol Nurs 2008;20:117-20.  Back to cited text no. 9
    
10.
Zhu H, Jiang Y, Watts M, Kong F. Treatment of pseudoepitheliomatous, keratotic, and micaceous balanitis with topical photodynamic therapy. Int J Dermatol 2015;54:245-7.  Back to cited text no. 10
    


    Figures

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



 

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