Indian Journal of Dermatology
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


Bangaru Hanumaiah1, Mohan2, Nanjundaswamy Bisilavadi Lingaiah1, Surendran Kalale Appaiah Kumaraswamy1, B Vijaya3,  
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

Correspondence Address:
Bangaru Hanumaiah
Department of Skin and STD, #14, OPD Block, K.R. Hospital, Mysore Medical College and Research Institute, Karnataka
India

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.



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;58:492-492


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 2021 Sep 18 ];58:492-492
Available from: https://www.e-ijd.org/text.asp?2013/58/6/492/119970


Full Text

 Introduction



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



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}{Figure 2}

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}{Figure 4}{Figure 5}{Figure 6}

The scaly material had dissolved completely in 10% KOH [potassium hydroxide] overnight [Figure 7].{Figure 7}

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}{Figure 9}

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

1Lortat-Jacob E, Civatte J. Micaceous and keratosic pseudo-epitheliomatous balantis. Bull Soc Fr Dermatol Syphiligr 1961;68:164-7.
2Reed SI, Abell E. Pseudoepitheliomatous, keratotic and micaceous balanitis. Arch Dermatol 1981;117:435-7.
3Pai 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.
4Subudhi 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.
5Rook A, Wilkinson DS, Ebling FJ, Champion RH, Burton JL. Textbook of Dermatology. 4 th ed. Boston, Mass: Blackwell Scientific Publications Inc; 1986. p. 2188.
6Jenkins 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.
7Bashir SJ, Grant JW, Burrows NP. Pseudoepitheliomatous, keratotic, micaceous balanitis after penile squamous cell carcinoma. Clin Exp Dermatol 2010;35:749-51.
8Kang 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.
9Bunker CB, Francis N. Pseudoepitheliomatous, keratotic, micaceous balanitis: Comment. Clin Exp Dermatol 2012;37:434-5.
10Krunic 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
11Kohn 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.