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  Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 43  |  Issue : 1  |  Page : 68-69
 

A rare presentation of pseudoepitheliomatous keratotic and micaceous balanitis with malignant transformation


1 Department of Urology, Venereology and Leprosy, Father Muller Medical College, Mangalore, Karnataka, India
2 Department of Dermatology, Venereology and Leprosy, Father Muller Medical College, Mangalore, Karnataka, India
3 Department of Pathology, Father Muller Medical College, Mangalore, Karnataka, India

Date of Submission19-Jan-2021
Date of Decision30-Apr-2021
Date of Acceptance10-Oct-2021
Date of Web Publication07-Jun-2022

Correspondence Address:
Dr. Sonal Fernandes
Department of Dermatology, Venereology and Leprosy, Father Muller Medical College, Kankanady, Mangalore - 575 002, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijstd.ijstd_3_21

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   Abstract 


Pseudoepitheliomatous keratotic and micaceous balanitis (PKMB) is a rare nonvenereal penile condition with a risk of malignant transformation. We report a case of PKMB in a 45-year-old male who presented with multiple keratotic growths over his glans penis after circumcision for long-standing phimosis. Histopathology revealed pseudoepitheliomatous hyperplasia and atypical cells. He was successfully treated with wide local excision.


Keywords: Balanitis, micaceous, pseudoepitheliomatous


How to cite this article:
Nandakishore B, Bhat M R, Fernandes S, Marla NJ. A rare presentation of pseudoepitheliomatous keratotic and micaceous balanitis with malignant transformation. Indian J Sex Transm Dis 2022;43:68-9

How to cite this URL:
Nandakishore B, Bhat M R, Fernandes S, Marla NJ. A rare presentation of pseudoepitheliomatous keratotic and micaceous balanitis with malignant transformation. Indian J Sex Transm Dis [serial online] 2022 [cited 2022 Nov 28];43:68-9. Available from: https://www.ijstd.org/text.asp?2022/43/1/68/346578





   Introduction Top


Lesions on the penis can be a challenge to diagnose often due to late presentation, self-medication, or prior ineffectual treatment causing alarm to the patient. Certain rare conditions of the penis can progress into malignant growths requiring invasive surgery. One such exceedingly rare premalignant lesion is pseudoepitheliomatous keratotic and micaceous balanitis (PKMB) which is outlined by mica-like scales and keratotic horny masses over the glans penis.[1] Although the etiology of PKMB is perplexing, it is generally seen in the elderly male population. It has a potential to evolve into squamous cell carcinoma and verrucous carcinoma rarely.[2] Herein, we report a case of PKMB presenting as multiple cutaneous horny growths in an adult male.


   Case Report Top


A 45-year-old male presented with asymptomatic penile growths of 8-month duration. He first noticed it as a small elevated keratotic growth post circumcision, which was done for preexisting phimosis 10 months back. The lesions had been gradually increasing in size and number. There was no history of preceding trauma or any systemic disease. He gave no history of exposure to the risk of sexually transmitted disease. Prior treatment included the use of topical antibiotic cream without any improvement in symptoms. Clinical examination showed multiple bark-like, hard, yellowish horny masses over the glans penis sparing the urethral meatus [Figure 1]. Lesions were confined to the glans penis, whereas examination of the penile shaft and scrotum was normal. Inguinal lymph nodes were not palpable. The patient's serology was negative for HIV, syphilis, and hepatitis B and C viruses. Routine laboratory investigations which comprised basic blood counts, levels of blood glucose, and tests for renal and liver function showed normal values. Based on these findings, excisional biopsy of the lesion was performed heeding toward a diagnosis of PKMB. Histopathology of the lesion revealed pseudoepitheliomatous hyperplasia with extensive hyperkeratosis, parakeratosis, acanthosis, individual cell keratinization, and atypical cells. Dermis showed dense lymphoplasmacytic inflammatory infiltrate [Figure 2]a and [Figure 2]b. A final diagnosis of PKMB evolving to squamous cell carcinoma of the glans penis was made, and the lesions were surgically excised with wide margins.
Figure 1: Clinical image showing keratotic horny growths circumferentially present over the glans penis

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Figure 2: (a) Histopathological image showing pseudoepitheliomatous hyperplasia, hyperkeratosis, parakeratosis, and acanthosis. H and E, ×4. (b) Histopathological image showing atypical cells. H and E, ×10

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   Discussion Top


In 1961, Lortat-Jacob and Civatte first described a case of PKMB characterized by thick scaly plaques on the glans penis. “Micaceous” literally means mica-like, resembling sheets of white scaly aluminum silicate. It was initially considered an entirely benign condition.[3]

At present, it is reflected upon as a premalignant condition with low-grade malignancy potential. Pseudoepitheliomatous response to chronic inflammation has been proposed as a possible etiopathogenic mechanism.[4] Kang et al. reported a case of recurrent PKMB associated with human papillomavirus and postulated the role of transformation to verrucous carcinoma.[5] Acquired phimosis and adult circumcision have often been linked with this condition.[6] Clinically, PKMB presents as an asymptomatic slow-growing scaly or verrucous plaques on the surface of the glans penis, rarely causing urinary obstruction. The pathogenic evolution of PKMB involves four stages: (a) plaque stage (early), (b) tumor stage (late), (c) verrucous carcinoma, and (d) squamous cell carcinoma with lymphatic and metastatic invasion.[7] Histologically, initial stages show marked hyperkeratosis and parakeratosis, acanthosis, and pseudoepitheliomatous hyperplasia. In later stages, features of cellular atypia, koilocytes, and pleomorphism are seen.[6] Differential diagnoses include lichen sclerosus et atrophicus, penile psoriasis, penile horn, giant condyloma, verrucous or squamous cell carcinoma, and erythroplasia of Queyrat. Treatment with topical 5-fluorouracil, radiotherapy, cryotherapy, and CO2 laser has been used when there is no histological evidence of malignancy. Topical 5-fluorouracil is the most highly recommended treatment for the initial plaque stage.[8],[9] Long-term surveillance with posttreatment biopsies is recommended during the follow-up period. The tumor stage of PKMB showing features of dysplasia and invasion is defiant to topical treatment. Wide local surgical excision, resurfacing and glansectomy with split-thickness skin graft are recommended depending on the progression of the lesion in advanced cases having histological features of cellular atypia.[10]

Our patient presented at a relatively young age with preexisting long-standing phimosis. Adult circumcision could be a possible trigger for PKMB in our patient. PKMB being a rarity should be recognized early due to its association with penile malignancy. Early clinical as well as histopathological identification and prompt treatment is undoubtedly the best course of action for the management of this condition. The basic understanding of this limitative ailment could be better inferred with a lifelong follow-up of the 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.
Hanumaiah B, Mohan NB, Kumaraswamy SK, Vijaya B. Pseudoepitheliomatous keratotic and micaceous balanitis: A rare condition successfully treated with topical 5-Fluorouracil. Indian J Dermatol 2013;58:492.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Kumar P, Mondal AK, Mondal A, Gharami RC, Maiti A. Pseudoepitheliomatous micaceous and keratotic balanitis – A rare case. J Pak Assoc Dermatol 2016;21:74-6.  Back to cited text no. 2
    
3.
Lortat-Jacob E, Civatte J. Micaceous and keratosic pseudo-epitheliomatous balanitis. Bull Soc Fr Dermatol Syphiligr 1961;68:164-7.  Back to cited text no. 3
    
4.
Tonin B, Catenaro R, Girolomoni G, Ghimenton C, Gisondi P. Pseudoepitheliomatous, keratotic, and micaceous balanitis mimicking lichen sclerosus et atrophicus. Dermatol Online J 2020;26:16.  Back to cited text no. 4
    
5.
Kang BS, Lee SD, Park YM, Cho SY, Kang H. Is recurrent pseudoepitheliomatous keratotic balanitis related to human papillomavirus infection? Acta Derm Venereol 2010;90:208-9.  Back to cited text no. 5
    
6.
Choo KJ, Ng SK, Sim CS, Cheng SW. Pseudoepitheliomatous keratotic and micaceous balanitis treated with topical 5-fluorouracil and liquid nitrogen. Clin Exp Dermatol 2017;42:424-6.  Back to cited text no. 6
    
7.
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. 7
    
8.
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. 8
    
9.
Shabbir M, Minhas S, Muneer A. Diagnosis and management of premalignant penile lesions. Ther Adv Urol 2011;3:151-8.  Back to cited text no. 9
    
10.
Kim JY, Kim JY, Park M, Oh CK, Chung JS, Park SH, et al. Surgical managements of pseudoepitheliomatous keratotic and micaceous balanitis: A case report. Int J Surg Case Rep 2019;55:37-40.  Back to cited text no. 10
    


    Figures

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