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E-CASE REPORT
Year : 2013  |  Volume : 58  |  Issue : 6  |  Page : 492
Pseudoepitheliomatous keratotic and micaceous balanitis: A rare condition successfully treated with topical 5-fluorouracil


1 Department of Skin and STD, Mysore Medical College and Research Institute, Mysore, Karnataka, India
2 Department of Plastic Surgery, Mysore Medical College and Research Institute, Mysore, Karnataka, India
3 Department of Pathology, JSS Medical College, Mysore, Karnataka, India

Date of Web Publication17-Oct-2013

Correspondence Address:
Bangaru Hanumaiah
Department of Skin and STD, #14, OPD Block, K.R. Hospital, Mysore Medical College and Research Institute, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.119970

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   Abstract 

A 50-year-old man presented with slow-growing dry, rough, micaceous scaly plaque over glans penis, which was compatible with clinical diagnosis of pseudoepitheliomatous keratotic and micaceous balanitis (PKMB) and histologically suggestive of PKMB without cellular atypia. He was treated successfully with topical 5-fluorouracil with complete clearance of lesion in 3 weeks without recurrence for 10 months. PKMB is an extremely rare and interesting condition; only a handful cases have been reported in world literature.


Keywords: Carcinoma, micaceous scales, pseudoepitheliomatous hyperplasia


How to cite this article:
Hanumaiah B, Mohan, Lingaiah 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

How to cite this URL:
Hanumaiah B, Mohan, Lingaiah NB, Kumaraswamy SK, Vijaya B. Pseudoepitheliomatous keratotic and micaceous balanitis: A rare condition successfully treated with topical 5-fluorouracil. Indian J Dermatol [serial online] 2013 [cited 2019 Jun 26];58:492. Available from: http://www.e-ijd.org/text.asp?2013/58/6/492/119970

What was known?
Pseudoepitheliomatous keratotic and micaceous balanitisis an extremely rare condition, a high index of suspicion is required for early diagnosis and to differentiate it from other common conditions like psoriasis, giant condyloma, and squamous cell carcinoma.



   Introduction Top


Pseudoepitheliomatous keratotic and micaceous balanitis (PKMB) is an extremely rare, distinctive clinical entity characterized by mica-like crusts and keratotic horny masses on glans penis. [1] The exact etiology is not known. Initially, it was considered as a benign condition, it may become locally invasive or verrucous carcinoma. [1],[2] An early clinical diagnosis and biopsy of the lesion is of great value as it can be treated by topical 5-fluorouracil with complete clearance of lesion if there is no cellular atypia as in our case.


   Case Report Top


A 50-year-old circumcised male presented with asymptomatic growth on glans penis of 6 months duration.

Patient had undergone circumcision for phimosis 8 months back. Two months later, patient noticed an asymptomatic scaly lesion over the glans penis, which slowly increased in size and became dry, rough, and elevated.

There was no history of trauma, localized dermatoses, or systemic diseases. There was no history of exposure to risk of sexually transmitted diseases (STD) and no symptoms and signs of STD. There was no significant illness in the partner.

Clinical examination revealed a well-defined dry, rough, hard elevated, mica-like scaly, and verrucous and hyperkeratotic plaques, measuring 4 × 3 cm over glans penis, around urethral meatus [Figure 1] and [Figure 2].
Figure 1: Well-defined, dry, micaceous scaly plaque over the glans penis

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Figure 2: Well-defined, dry, scaly plaque over the glans penis around the urethral orifice

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The shaft of the penis, scrotum, and inguinal area was normal. There was no regional lymphadenopathy.

His blood Venereal Disease Research Laboratory Test and HIV were negative. Hematological, biochemical, and radiological examination did not reveal any abnormality.

Histopathological examination revealed skin with epidermis displaying massive hyperkeratosis, parakeratosis, and pronounced epithelial hyperplasia without cellular atypia. The dermis showed mild lymphocytic infiltrate [Figure 3], [Figure 4], [Figure 5], [Figure 6].
Figure 3: Hyperkeratosis, parakeratosis, acanthosis with elongation of rete ridges (H and E, ×10)

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Figure 4: Extensive parakeratosis and orthokeratosis (H and E, ×40)

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Figure 5: Pronounced acanthosis (H and E, ×40)

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Figure 6: Pronounced acanthosis without cytologic atypia (H and E, ×100)

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The scaly material had dissolved completely in 10% KOH [potassium hydroxide] overnight [Figure 7].
Figure 7: Complete dissolution of keratotic scaly material in 10% KOH after 24 hours

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The patient was treated with topical 5-fluorouracil cream application once daily for 3 weeks and the whole lesion regressed completely without any recurrence for a period of 10 months follow-up [Figure 8] and [Figure 9].
Figure 8: Complete clearance of lesion, 3 weeks after topical 5-fluorouracil application

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Figure 9: Complete clearance of lesion, 3 weeks after topical 5- fluorouracil application

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IJD_457_12

PKMB is an extremely rare and interesting condition in which a coronal balanitis gradually takes on a silvery white appearance with mica-like crusts and keratotic horny masses formed on the glans. It was originally described by Lortat-Jacob and Civatte in 1961. [1]

The keratotic scaling is usually micaceous and resembles psoriasis. It can present as penile horn. [3] A nail-like lesion on the glans has also been described. [4]

The exact etiology and pathomechanism is not known. It has been regarded as a form of pyodermatitis or pseudoepitheliomatous response to infection, possibly a variant of Reiter's syndrome [5] Initially, it was considered as entirely benign condition; [1] recent evidences showed that it is a distinctive entity that represents a histologic spectrum ranging from hypertrophic-hyperplastic penile dystrophy to verrucous carcinoma. [6] The lesion may have locally invasive or aggressive tendencies and should be considered to have low-grade or limited malignant potential. [2] PKMB has been reported arising from a treated case of squamous cell carcinoma. [7] The human papilloma virus may be associated with the development and recurrence of PKMB and a possible role in transformation to verrucous carcinoma. [8] Even though some authors say that there is no relation of balanitis xerotica obliterance with PKMB, some are of the opinion that PKMB sometimes, if not always is a feature of lichen sclerosus. [9] The pathogenesis of PKMB occurs in four stages: (a) Initial plaque stage, (b) Late tumor stage, (c) verrucous carcinoma, and (d) Transformation to squamous cell carcinoma and invasion. [10]

This condition should be differentiated from other diseases on male genitalia-like giant condyloma, psoriasis, squamous cell carcinoma, and erythroplasia of Queyrat.

Treatment includes topical 5-fluorouracil and cryotherapy when there is no histological evidence of malignancy. [10] Extensive surgical excision is required in cases having features of cellular atypia for good cosmetic and functional results. [2],[6] Although not many reports are available, Imiquimod offers another possible approach. [11]

 
   References Top

1.Lortat-Jacob E, Civatte J. Micaceous and keratosic pseudo-epitheliomatous balantis. Bull Soc Fr Dermatol Syphiligr 1961;68:164-7.  Back to cited text no. 1
    
2.Reed SI, Abell E. Pseudoepitheliomatous, keratotic and micaceous balanitis. Arch Dermatol 1981;117:435-7.  Back to cited text no. 2
    
3.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. 3
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4.Subudhi CL, Singh PC. Pseudoepitheliomatous, keratotic and micaceous balanitis producing nail-like lesion on the glans penis. Indian J Dermatol Venereol Leprol 1999;65:75-7.  Back to cited text no. 4
  Medknow Journal  
5.Rook A, Wilkinson DS, Ebling FJ, Champion RH, Burton JL. Textbook of Dermatology. 4 th ed. Boston, Mass: Blackwell Scientific Publications Inc; 1986. p. 2188.  Back to cited text no. 5
    
6.Jenkins D Jr, Jakubovic HR. Pseudoepitheliomatous, keratotic, micaceous balanitis. A clinical lesion with two histologic subsets: Hyperplastic dystrophy and verrucous carcinoma. J Am Acad Dermatol 1988;18:419-22.  Back to cited text no. 6
    
7.Bashir SJ, Grant JW, Burrows NP. Pseudoepitheliomatous, keratotic, micaceous balanitis after penile squamous cell carcinoma. Clin Exp Dermatol 2010;35:749-51.  Back to cited text no. 7
    
8.Kang BS, Lee SD, Park Y, 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. 8
    
9.Bunker CB, Francis N. Pseudoepitheliomatous, keratotic, micaceous balanitis: Comment. Clin Exp Dermatol 2012;37:434-5.  Back to cited text no. 9
    
10.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. 10
    
11.Kohn F-M. Diseases of the male genitalia. In: Burgdorf WHC, Plewig G, Wolff HH, Landthalor M, editors. Braun-Falco's Dermatology. 3 rd ed. Berlin: Springer-Verlag, Heidelberg; 2010. p. 1140-52.  Back to cited text no. 11
    

What is new?
As pseudoepitheliomatous keratotic and micaceous balanitis is an extremely rare condition, a high index of suspicion is required to diagnose and differentiate this interesting condition from other common genital diseases. An early biopsy will help to diagnose and plan the treatment. In the absence of cellular atypia, topical 5.fluorouracil or cryotherapy is effective, and if atypia is present surgical excision is required. Patients should be followed.up regularly as it can recur and malignant transformation can also occur. This case has been reported for its extreme rarity and an excellent response shown to topical 5.fluorouracil therapy.


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]



 

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