Primary high-grade non-Hodgkin lymphoma of the female genital tract is extremely rare. Vaginal bleeding, abdominal pain or urinary complaints might be its most frequent symptoms. We report a 27-year-old multipara who underwent large loop excision of the transformation zone because of the repeated finding of a low-grade squamous intraepithelial lesion identified during routine cancer screening. Incidentally, CD20-positive, primary, diffuse large B-cell lymphoma infiltrating the mucosa of the endocervix was also diagnosed from this specimen. The case is unusual because the patient had no symptoms, specific colposcopic signs or visible mass. R-CHOP 21 immunochemotherapy was introduced and resulted in complete remission without hysterectomy. The patient is without any evidence of disease after 49 months of follow-up. Primary cervical lymphomas are mainly subepithelial initially, and therefore they may be under-recognized due to the inefficiency of smears to diagnose such lesions. Early diagnosis and available targeted treatment allowed a cure in the reported example.

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