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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 2  |  Issue : 1  |  Page : 25-27

Bowenoid papulosis of genitalia responding to topical 5-Fluorouracil


1 Department of Dermatology, Venereology and Leprosy, ESIC Medical College and PGIMSR, Bengaluru, India
2 Skin Laser Centre, New Delhi, India
3 Department of Pathology, Sri Siddhartha Medical College, Tumkur, Karnataka, India
4 Department of Dermatology, Venereology and Leprosy, Sri Siddhartha Medical College, Tumkur, Karnataka, India

Date of Web Publication5-Jan-2018

Correspondence Address:
Meenakshi Kapoor
B-5/35, Sector-7, Rohini, New Delhi - 110 085
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/CDR.CDR_11_17

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  Abstract 


Bowenoid papulosis (BP), a rare disease with malignant potential, is a distinct clinicopathological entity strongly associated with human papillomavirus (HPV) infection. We described an adult male presenting with well defined, purplish papules of varying size with verrucous to smooth surface distributed discretely over medial side of the left thigh, scrotum, and penile shaft. Histopathology showed focal areas of full thickness epidermal atypia comprising irregularly arranged nuclei which are large, hyperchromatic, and crowded at few places. Pap smear from spouse showed inflammatory infiltrate with moderate-to-severe dysplasia. The patient was treated with topical 5-fluorouracil. Excellent response was noticed at the end of 2 weeks. A patient with BP should be thoroughly educated regarding HPV infection and emphasis should be on prevention. Female patients or sexual partners of male patient with BP are at the risk of developing cervical cancer and hence should be followed with regular cytologic, colposcopic, and histologic examinations.

Keywords: 5-fluorouracil, bowenoid papulosis, genital warts, Pap smear


How to cite this article:
Shivanna R, Kapoor M, Murthy B N, Narendra G. Bowenoid papulosis of genitalia responding to topical 5-Fluorouracil. Clin Dermatol Rev 2018;2:25-7

How to cite this URL:
Shivanna R, Kapoor M, Murthy B N, Narendra G. Bowenoid papulosis of genitalia responding to topical 5-Fluorouracil. Clin Dermatol Rev [serial online] 2018 [cited 2018 Oct 22];2:25-7. Available from: http://www.cdriadvlkn.org/text.asp?2018/2/1/25/222262




  Introduction Top


Bowenoid papulosis (BP), a rare disease with malignant potential, is a distinct clinicopathological entity characterized by multiple, multicentric, multiform, skin colored to reddish brown, or violaceous papules primarily occurring on genitalia of young adults. BP is strongly associated with human papillomavirus (HPV) infection. It is often considered as low grade in situ carcinoma.[1]


  Case Report Top


A 37-year-old married male presented with itchy pigmented skin lesions over the genital area of 6 months duration. Itching was mild in nature, intermittent, and more in daytime. The lesions gradually increased in number and size since then. There was no history suggestive of any immunosuppressed condition or history of long-term drug intake. However, the patient gave history of unprotected sexual intercourse before marriage. On examination, multiple, well defined, purplish papules of varying size with verrucous to smooth surface distributed discretely over medial side of the left thigh, scrotum [Figure 1], and penile shaft was present. Venereal disease research laboratory test and HIV screening test were nonreactive for both husband and wife. Complete blood count and random blood sugar levels were within normal limits.
Figure 1: Multiple, well defined, purplish papules of varying size with verrucous to smooth surface distributed over scrotum and medial side of the left thigh

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Histopathology showed focal areas of full thickness epidermal atypia comprising irregularly arranged nuclei which are large, hyperchromatic, and crowded at few places [Figure 2]. A few dyskeratotic cells and few cells with koilocytic changes were also noted. Basement membrane was intact without evidence of invasion. Per speculum examination of spouse showed thick, white, foul smelling discharge with hypertrophied cervix.  Pap smear More Details from spouse showed inflammatory infiltrate with moderate-to-severe dysplasia.

On the basis of clinical and histopathological features, a diagnosis of BP was made, and patient was treated with topical 5-fluorouracil (5-FU). On follow-up after 1 week, the patient presented with erythema, erosion, and burning sensation at the site of application of 5-FU suggestive of irritant reaction. The patient was advised to stop 5-FU and apply mild corticosteroid two times daily. The patient was asked to restart 5-FU application once the symptoms subsided. After 2 weeks, the majority of the lesions showed excellent response with complete healing [Figure 3]. Both husband and wife were advised regarding the course of the illness and importance of regular follow-up and Pap smear.
Figure 2: Acanthosis of epidermis with focal areas of full thickness epidermal atypia comprising crowded and irregularly arranged large and hyperchromatic nuclei (H and E, ×40)

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Figure 3: Excellent response with flattening of lesions and disappearance of few lesions after 2 weeks of topical 5-fluorouracil therapy

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  Discussion Top


BP was first described by Lloyd as multicentric pigmented Bowen's disease (BD) in 1970. Various authors have given different names to this condition, for example, intraepithelial neoplasia, pseudo-BD, multicentric pigmented BD, and pigmented viral papulosis of genitals.[2]

BP is considered as a virus-induced epithelial dysplasia mainly associated with HPV infection. HPV subtypes, mainly 16, 18, 31, and 33 which have high malignant potential are associated with BP.[2],[3] HPV infection has been implicated in wide range of epithelial or fibroepithelial proliferation of the skin and mucosa. These include common warts, flat pityriasis versicolor suh as lesions in epidermodysplasia verruciformis, oral and laryngeal papillomas, and various genital lesions.[4] Immune response appears to play a role in the development of BP. Spontaneous regression of BP has been reported in AIDS patients after increase in peripheral CD4 T lymphocytes count following antiretroviral therapy.[5] Association of idiopathic CD4 lymphocytopenia with extragenital BP involving dorsal aspect of middle finger caused by HPV-18 has also been reported.[3]

BP usually affects sexually active adults with slight female predominance. Classically, BP presents as multiple skin-colored or pigmented papules of varying size on a circumcised penile shaft, labia majora, labia minora, and vulva, but it may also occur anywhere in genitoanal region.[6] Papules may be discrete, grouped or linear, confined, smooth or verrucous in morphological appearance. Lesions are usually asymptomatic but can itch occasionally. Clinical variants of BP such as papules coalescing to form plaques, lichenoid papules, macules, and leukoplakia-like lesions have been described.[1] In the present case, both genital and adjacent extragenital sites were involved. Extragenital BP is uncommon and usually associated with concomitant genital involvement. It may occur by autoinoculation from preceding genital BP. Most common sites are mainly exposed areas such as chin, neck, fingers, and periungual area.[3]

The natural history of this condition ranges from spontaneous regression to rare progression to invasive squamous cell carcinoma (SCC). The course of the disease is variable; lesions can regress in few months, persist for many years, recur or progress to invasive SCC, especially in immunocompromised individuals.[5]

BP is difficult to differentiate clinically from viral warts and histopathologically from BD and erythroplasia of Queyrat. Other differential diagnosis includes seborrhoeic keratosis, melanocytic nevi, lichen planus, and condyloma lata.[7] BP can be differentiated from BD on the basis of clinical features.[8] BP is characterized by earlier age of onset, multiple small lesions, verrucous appearance, and tendency toward spontaneous regression.[1] Histopathology of BP is characterized by psoriasiform epidermal hyperplasia, hyperkeratosis with focal parakeratosis and focal acanthosis with almost full thickness epidermal atypia (keratinocytes shows loss of cellular polarity, dyskeratosis, multinucleated giant cells, and hyperchromatic nuclei) with spongiosis and exocytosis. Dermis shows dilated and tortuous capillaries with lymphohistiocytic infiltrate. Histopathological features such as extent of epidermal atypia, severity of atypia, and characteristics of koilocytes are important in the diagnosis of BP. As a rule, koilocytes are absent in lesions with severe atypia.[4] Histopathology of BD is characterized by epidermal acanthosis with elongation of rete ridges that reduces the papillae to thin strands giving “windblown” appearance. Epidermal cells are highly atypical with abnormal mitotic figures and dyskeratotic extending up to follicular infundibulum with few cells showing multinucleation and vacuolization. Basement membranes remain intact and upper dermis shows moderate amount of chronic inflammatory infiltrate.[9]

Therapeutic modalities that have been used successfully include topical application of retinoic acid, imiquimod, 5-FU, and cidofovir. Imiquimod has showed promising results and has been used safely in pregnancy. Surgical modalities include cryotherapy, electrodessication, shave biopsy, surgical excision, interferon injection and vaporization by laser.[10] Podophyllin is contraindicated in case of persistent or recurring lesions due to its mutagenic effects.[8] Clinicians are advised to use judiciously an appropriate conservative modality as some cases spontaneously regress, and malignant transformation is rare. Recurrence of BP is frequent. A biopsy should be taken from lesions that are recalcitrant to standard therapy to exclude malignancy. Longer duration of lesions, especially in elderly individual must be regarded as precursor of malignancy and such lesions should be removed surgically or with lasers.[8] In pediatric age group examine the patient for any sexual abuse and to rule out gonorrhea and syphilis. Regular follow-up is required to rule out carcinoma in situ/SCC. Cryotherapy with liquid nitrogen appears to be a safe and effective therapeutic modality in children.[11]

In view of assumed sexual transmission and proven association with cervical neoplasia (HPV-16), emphasis should be on prevention. The patient should be thoroughly educated regarding HPV infection and use of condoms. The patient should be routinely examined for recurrence. Female patients or sexual partners of male patient with BP are at the risk of developing cervical cancer and hence should be followed with regular cytologic, colposcopic, and histologic examinations.[12]

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.
von Krogh G, Horenblas S. Diagnosis and clinical presentation of premalignant lesions of the penis. Scand J Urol Nephrol Suppl 2000;(205):201-14.  Back to cited text no. 1
    
2.
Henquet CJ. Anogenital malignancies and pre-malignancies. J Eur Acad Dermatol Venereol 2011;25:885-95.  Back to cited text no. 2
[PUBMED]    
3.
Purnell D, Ilchyshyn A, Jenkins D, Salim A, Seth R, Snead D. Isolated human papillomavirus 18-positive extragenital bowenoid papulosis and idiopathic CD4+ lymphocytopenia. Br J Dermatol 2001;144:619-21.  Back to cited text no. 3
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4.
Gross G, Ikenberg H, Gissmann L, Hagedorn M. Papillomavirus infection of the anogenital region: Correlation between histology, clinical picture, and virus type. Proposal of a new nomenclature. J Invest Dermatol 1985;85:147-52.  Back to cited text no. 4
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5.
Kawakami A, Saga K, Ono I, Hida T, Jimbow K, Yamashita T. Spontaneous regression of bowenoid papulosis in a patient with acquired immunodeficiency syndrome after an increase in peripheral CD4+ T lymphocytes. Int J Dermatol 2009;48:210-2.  Back to cited text no. 5
    
6.
Heenan PJ. Human papilloma virus induced epidermal proliferations of the anogenital region. Australas J Dermatol 1988;29:17-23.  Back to cited text no. 6
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7.
Bunker CB, Neill SM. The genital, perianal and umbilical regions. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8th ed. Oxford: Wiley-Blackwell; 2010. p. 71.1-102.  Back to cited text no. 7
    
8.
Jablonska S, Majewski S. Bowenoid papulosis transforming into squamous cell carcinoma of the genitalia. Br J Dermatol 1999;141:576-7.  Back to cited text no. 8
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9.
Kirkhum N. Tumors and cysts of epidermis. In: Elder DE, Elenitsas R, Johnson BL, Murphy GF, editors. Lever's Histopathology of the Skin. 9th ed. Philadelphia: Lippincott Williams & Wilkins; 2005. p. 806-66.  Back to cited text no. 9
    
10.
Loo WJ, Holt PJ. Bowenoid papulosis successfully treated with imiquimod. J Eur Acad Dermatol Venereol 2003;17:363-5.  Back to cited text no. 10
[PUBMED]    
11.
Weitzner JM, Fields KW, Robinson MJ. Pediatric bowenoid papulosis: Risks and management. Pediatr Dermatol 1989;6:303-5.  Back to cited text no. 11
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12.
Ricart JM, Cordoba J, Hernandez M, Esplugues I. Extensive genital bowenoid papulosis responding to imiquimod. J Eur Acad Dermatol Venereol 2007;21:113-5.  Back to cited text no. 12
[PUBMED]    


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  [Figure 1], [Figure 2], [Figure 3]



 

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