Coexistence of condyloma acuminatum and extramammary Paget's disease on penis and scrotum: A rare case report

Abstract

Rationale: Extramammary Paget's disease (EMPD) is a rare skin cancer that commonly occurs in sites rich in apocrine glands, such as perineum, vulva, axilla, scrotum, and penis. On the other hand, condyloma acuminatum (CA; also referred to as anogenital warts) is a common benign neoplasm caused by human papillomavirus. Few cases of coexistent EMPD and CA have been reported because of the rarity of the condition.

Patient concerns and diagnosis: A 72-year-old man with a genital mass, which appeared to be composed of multiple papillomatous masses, was referred for surgical resection. The lesion was first noticed 6 months previously and grew rapidly. CO2 ablative laser therapy was performed twice at a primary clinic, but the mass recurred.

Intervention and outcomes: Excisional biopsy revealed the presence of coexistent EMPD and CA. Additional wide excision was performed, and postoperative biopsy confirmed no residual tumor. Two years after surgery, no recurrence had occurred.

Lessons: CA can co-occur with several types of skin malignancies, and a skin malignancy coexisting with CA is difficult to diagnose visually. Therefore, even if a skin lesion in the genital region is considered to be CA, we recommend punch biopsy before treatment because it can benefit prognosiss.

Figure 1.

Preoperative photograph. 8.0 × 6.0 cm lesion on penis and scrotum with hypopigmented plaque and multiple papillomatous masses.

Figure 2.

Histologic features. (A) A polypoid lesion of condyloma acuminatum was noted (asterisks), and the adjacent surface and follicular epithelium were thickened (arrows) (H&E, ×40). (B) At high magnification, large, atypical cells with abundant cytoplasm (Paget cells) were observed in thickened epithelium (H&E, ×200). (C, D) Immunohistochemical staining for allowed Paget cells, which presented as single cells or nests, to be easily identified at the periphery and base of the condyloma and adjacent thickened epithelium (CK7, ×40/CK7, ×200). CK7 = cytokeratin 7.

Figure 3.

Intraoperative photographs. (A) Wide excision was performed with a margin of 2 cm. Excision was performed until the entire margin was negative, as determined by frozen biopsy. (B, C) Defects in the scrotum and abdomen were covered using a scrotal dartos musculocutaneous flap, and the penile shaft was covered using a full-thickness skin graft.

Figure 4.

Postoperative photographs. (A) No recurrence occurred during 2 yr of postoperative follow-up, and no wound complication was encountered. (B) FTSG reconstruction of the penile shaft provided sufficient elasticity to preserve erection function. FTSG = full-thickness skin graft.