HPV is the most common sexually transmitted infection in the world. HPV 16 and 18 are found to account for 90% of all HPV-related cancers in men. We are reporting a rare case of penile squamous carcinoma associated with HPV infection in a patient with ulcerative colitis and under immunosuppressants (IMMs).
A 72-year-old male patient with a personal history of severe chronic obstructive pulmonary disease was diagnosed with ulcerative colitis in 2007. He achieved initial remission with topical and systemic salicylates. But months later he presented a severe flare that required treatment with corticoids. After that episode he was started on thiopurines for corticoid-dependency. He maintained a long-lasting remission thereafter.
In January 2013 he was diagnosed with penile squamous carcinoma that required glansectomy and urethroplasty. Histological results showed a well-differentiated squamous cell carcinoma, with perineural invasion. Inmunohistochemistry was positive for p16. In situ hybridization confirmed HPV 16/18 positive and HPV 6/11 negative. After the diagnosis of penile cancer he stopped azathioprine and was de-intensified to mesalazine 4g per day, maintaining ulcerative colitis remission. On October 2013, he developed an inguinal lymphadenopathy that was a metastatic squamous cell carcinoma. No adjuvant therapy was decided upon in light of the poor prognosis and comorbidity of the patient. He finally died in June 2014.
HPV is highly prevalent in men and it is related to the development of genital warts, penile intraepithelial neoplasia and invasive penile carcinomas. Approximately 40% of invasive penile carcinomas are attributable to HPV 16/18. There are no previous reports of HPV-related cancers in male patients with IBD taking IMMs. Although a cause–effect relationship with IMMs cannot be demonstrated in this case, long-lasting immunosuppression has been shown to increase the number and persistence of HPV-induced lesions.1
Recently, the Second European evidence-based consensus on the prevention, diagnosis, and management of opportunistic infections in IBD recommended HPV vaccination for females aged 11–14 years before onset of sexual activity. They also recommend routine vaccination of males according to national guidelines.2 Vaccinating boys improves cervical cancer eradication, reduces virus transmission, and contributes to the prevention of HPV-associated diseases in both genders3; therefore more substantial incremental benefits are expected from adding males to vaccination programs.
In the present case we have shown an HPV-positive penile cancer developed under therapeutic immunosuppression with thiopurines. To the best of our knowledge, this association has not been reported before. Male vaccination has been shown to significantly reduce HPV-associated anogenital infection and related lesions in men. Therefore, we believe that HPV surveillance and vaccination programs in IBD should actively take into account the male population.