Paratesticular cellular angiofibroma with atypical (bizarre) cells: Case report and literature review

We report the extremely unusual occurrence of a cellular angiofibroma (CAF) with atypical (bizarre) cells in the spermatic cord. We present a 63-year-old man, who was referred to the Urology Service with a six-month history of a slowly growing painless nodule in the right inguino-scrotal area. The clinical impression was that of a lipoma. The mass was locally excised. Gross examination showed a well-circumscribed neoplasm attached to the spermatic cord and measuring 5 cm in the greatest dimension. Microscopic examination of the tumor showed the appearance of CAF with scattered severely atypical (bizarre) cells distributed throughout the lesion. By immunohistochemistry, atypical cells showed diffuse expression of p16, CDK-4, CD34 and vimentin. Keratin AE1/AE3, S-100 protein, p53, and epithelial membrane antigen were negative. The patient is free of disease two months after tumor excision. To the best of our knowledge, this is the third case of CAF with atypical (bizarre) cells occurring in the paratesticular area. Pathologists should be aware of this morphological variation of CAF to avoid misdiagnosis and over-treatment.