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Bowen's disease (BD) is one of the commonest cutaneous malignancies, regardless of race. Dermatologists occasionally observe BD with varying degrees of melanin deposits within the lesion, a condition called pigmented Bowen's disease (pBD). pBD accounts for 1.7–5.5% of all BD cases;[1] however, pBD on the palm or sole is extremely rare. Herein, we report the first case of human papillomavirus (HPV) 16-related pBD on the palm.

An 85-year-old woman presented with an asymptomatic, hyperkeratotic, peripherally pigmented tumour of 3.5 cm long by 2.8 cm wide by 0.7 cm height on her right palm (Fig. 1a,b). The tumour had gradually developed during the 2 years before the patient came to us on referral. The dermoscopic findings included surface scales, linear brown or gray dots and a hypopigmented structureless zone (Fig. 1c,d). The tumour also showed the parallel furrow pattern that is often seen in nevus pigmentosus. There had been no other verruca vulgaris on the palms and the soles. We performed surgical removal of the entire tumour, and the lesion was found to be histopathologically markedly exophytic and papillomatous (Fig. 2a). Throughout the epidermis, cells arranged in complete disorder were observed, and clumping cells and dyskeratotic cells were often also observed (Fig. 2b). In the dermis, patchy aggregations of melanophages were seen (Fig. 2c). On the basis of these findings, we made the final diagnosis of pBD. We investigated 16 HPV genotypes (genotypes 6, 11, 16, 18, 30, 31, 33, 35, 39, 45, 51, 52, 56, 58, 59 and 66) by multiplex PCR, and then only HPV16 was detected from the lesion (Fig. 2d). Upon receiving this result, the patient underwent cervical cytology; however, she was found to be free of cervical cancer.

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Figure 1. Clinical features and dermoscopic patterns of the lesion. (a, b) The tumour on the right palm was found to be asymptomatic, hyperkeratotic, peripherally pigmented and 3.5 cm long by 2.8 cm wide by 0.7 cm height. (c, d) Surface scales (†), brown or gray dots arranged linearly (‡), hypopigmented structureless zone (▶) and parallel furrow pattern(*).

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Figure 2. Histopathology of the lesion, and genotyping of human papillomavirus by multiplex PCR. (a) Histopathologically, hyperkeratosis, acanthosis, exophytic epidermis and papillomatosis are found (haematoxylin and eosin, original magnification ×40). (b) Disorganized atypical keratinocytes, clumping cells (arrow) and dyskeratotic cells (triangle) are observed (haematoxylin and eosin, original magnification ×100) throughout the epidermis. (c) In the epidermis, column of melanin granules was observed. In the dermis, patchy aggregations of melanophages were seen (Fontana-Masson, original magnification ×100). (d) The occurrences of a clear band (marked by red 16) and a faint band (blue circle) show the presence of human papillomavirus type 16 (HPV16).

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HPV16 has been known as a mucosal oncogenic HPV, and the virus is regarded as a high-risk factor in the development of cervical cancer.[2] However, HPV16 has been frequently detected also in BD of the hands.[3, 4] Carcinogenic role of HPV16 in human cutaneous keratinocytes has yet to be elucidated.

In this case, peripheral pigmentation due to melanin deposit was apparent, and some parts of the tumour showed dermoscopic characteristics of acral melanocytic lesions, including the parallel furrow pattern. It is known that HPV4, HPV60 and HPV65 have high amplification rates in pigmented warts.[5] However, a search of the English literature found no reports describing any association between HPV16 infection and pigmented warts or pBD.

In conclusion, this case is the first reported case of HPV16-associated pBD on the palm. The relationship of HPV infection and melanin deposition has not been clarified. Besides, the carcinogenic role of HPV16 in the development of cutaneous malignancies also remains unclear. We expect that more observations of HPV subtypes in pBD of the palm and sole will be accumulated.

References

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    Cameron A, Rosendahl C, Tschandl P, Riedl E, Kittler H. Dermatoscopy of pigmented Bowen's disease. J Am Acad Dermatol 2010; 62: 597604.
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    Ciapponi A, Bardach A, Glujovsky D, Gibbons L, Picconi MA. Type-specific HPV prevalence in cervical cancer and high-grade lesions in Latin America and the Caribbean: systematic review and meta-analysis. PLoS ONE 2011; 6: e25493.
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    Mitsuishi T, Sata T, Matsukura T, Iwasaki T, Kawashima M. The presence of mucosal human papillomavirus in Bowen's disease of the hands. Cancer 1997; 79: 19111917.
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    Clavel CE, Huu VP, Durlach AP, Birembaut PL, Bernard PM, Derancourt CG. Mucosal oncogenic human papillomaviruses and extragenital Bowen disease. Cancer 1999; 86: 282287.
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    Egawa K. New types of human papillomaviruses and intracytoplasmic inclusion bodies: a classification of inclusion warts according to clinical features, histology and associated HPV types. Br J Dermatol 1994; 130: 158166.