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Table of Contents 
Year : 2017  |  Volume : 62  |  Issue : 1  |  Page : 97-98
Extragenital human papillomavirus 16-associated bowen's disease

1 Department of Surgical, Microsurgical and Medical Sciences, Dermatology Unit, University of Sassari, Sassari, Italy
2 Department of Surgical, Microsurgical and Medical Sciences, Pathology Unit, University of Sassari, Sassari, Italy
3 Department of Biomedical Sciences, University of Sassari, Sassari, Italy

Date of Web Publication10-Jan-2017

Correspondence Address:
Maria Antonietta Montesu
Department of Surgical, Microsurgical and Medical Sciences, Dermatology Unit, University of Sassari, Sassari
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0019-5154.198035

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How to cite this article:
Montesu MA, Onnis G, Lissia A, Satta R, Are BM, Piana A, Sotgiu G. Extragenital human papillomavirus 16-associated bowen's disease. Indian J Dermatol 2017;62:97-8

How to cite this URL:
Montesu MA, Onnis G, Lissia A, Satta R, Are BM, Piana A, Sotgiu G. Extragenital human papillomavirus 16-associated bowen's disease. Indian J Dermatol [serial online] 2017 [cited 2021 Oct 20];62:97-8. Available from:


Bowen's disease (BD) is an in situ squamous cell carcinoma (SCC) that may occur with multiple lesions in 10%–20% of cases.[1] Furthermore, histological pagetoid cells type has been reported only in 5% of skin BD biopsy.

We describe a case of a 67-year-old male with multiple erythematous, hyperkeratotic, and ulcerated lesions that occurred 10 years before patient's observation, increasing in size and number over the years. Particularly, the widest lesion is hyperkeratotic and exudative plaque on the trunk, focal ulcerated, sized 11 cm × 8 cm [Figure 1]. Other lesions were observed on the trunk and ears. However, human papillomavirus (HPV)-related lesions were never detected on genitalia and the patient denied chronic ultraviolet and arsenic exposure.
Figure 1: Verrucous hyperkeratotic, exudative plaque on the left side of the trunk

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Biopsies from five lesions showed full-thickness epidermis involvement, atypical keratinocytes with large hyperchromatic and pleomorphic nuclei. Several keratinocytes presented vacuolated appearance and perinuclear halo [Figure 2],[Figure 3],[Figure 4]. Immunohistochemical staining showed atypical cells positive for cytokeratins (CKs) 5, 6 and negative for CK 7, carcinoembryonic antigen, epithelial membrane antigen, S100. Cells exhibited intense positivity for p16 (INK4a).
Figure 2: Disorderly maturation of keratinocytes and presence of atypical keratinocytes with perinuclear halos (H and E, ×40)

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Figure 3: At low magnification, verrucous lesion with marked hyperkeratosis and papillomatosis (H and E, ×10)

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Figure 4: Atypical keratinocytes with vacuolated appearance (H and E, ×20)

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Therefore, BD's pagetoid variant diagnosis was made.

DNA was isolated with HPV detection and genotyping; moreover, the L1 gene of HPV and human beta-globin was simultaneously co-amplified [Figure 5]. Furthermore, HPV coinfection 16/18 was detected and confirmed by real-time polymerase chain reaction assay.
Figure 5: Amplification of human papillomavirus L1 gene by real-time polymerase chain reaction: mixed infection of human papillomavirus 16 with human papillomavirus 18

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Routine blood tests were normal, and HIV test was negative.

The largest lesion was treated surgically while the others with liquid nitrogen cryotherapy.

HPV infection is one of the risk factors related to BD. Genitalia involvement is often associated with high-risk oncogenic HPV (16, 18, 31, 33) while extragenital lesions are often associated with other subtypes.

HPV 16 has been studied in genital carcinomas, while his role in nongenital SCC is still debated, but probably β-HPVs (Type 2) may be involved.[2]

In literature, a few cases of HPV-associated extragenital BD have been described. Lu et al. evaluated 91 BD patients HPV-negative.[3] Rubben et al. detected six HPV-16 positive cases of 191 lesions [4] that showed genital infection's history suggesting that patients transported by themselves HPV from genitalia to their own hands.

Our patient had multiple extragenital HPV-16 positive lesions, without hands and genitalia involvement or HPV-infection history. It represents a peculiar histological feature with koilocytes such as keratinocytes and perinuclear halo that could be a BD histological pattern related to viral etiology even in the absence of HPV history. Similarly to our case, Sun and Barr [5] described a “papillated BD” HPV negative, with verrucous appearance and koilocyte-like cells. The author speculated low viral load, believing that more sensitive techniques might be necessary to discover HPV.

Our patient is the first case of multiple extragenital BD with histological pagetoid appearance and HPV-16 DNA detection. The differential diagnosis between BD and SCC is sometimes critical; however, fortunately, our case did not show any malignant transformation.

Although in our case we lost the patient after 3 months, BD patients need close follow-up for the SCC risk and HPV research is recommended.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Gahalaut P, Rastogi MK, Mishra N, Chauhan S. Multiple pigmented Bowen's disease: A diagnostic and therapeutic dilemma. Case Rep Oncol Med 2012;2012:1-5.  Back to cited text no. 1
Asgari MM, Kiviat NB, Critchlow CW, Stern JE, Hawes SE, De Villiers EM: Detection of human papillomavirus DNA in cutaneous squamous cell carcinoma among immunocompetent individuals. J Invest Dermat 2008;128(6):1409-17.  Back to cited text no. 2
Lu S, Syrjänen K, Havu VK, Syrjänen S. Failure to demonstrate human papillomavirus (HPV) involvement in Bowen's disease of the skin. Arch Dermatol Res 1996;289(1):40-5.   Back to cited text no. 3
Rubben A, Baron J, Grussendorfconen E. Prevalence of Human papillomavirus type 16-related DNA in cutaneous Bowen's disease of the skin and squamous cell cancer. Int J Oncol 1996;9(4):609-11.  Back to cited text no. 4
Sun JD, Barr RJ. Papillated Bowen disease, a distinct variant. Am J Dermatopathol 2006;28(5):395-8.  Back to cited text no. 5


  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]


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