*Purpose: Cytomegalovirus (CMV) tissue-invasive disease occurs in higher proportion in CMV-mismatched kidney transplant recipients (KTxR) with no pre-existing immunity and those who are markedly immunologically-impaired. While CMV disease may affect any organ, cervicitis has not been reported in KTxR.

*Methods: A 46 year old woman with end stage kidney disease due to adult polycystic kidney disease underwent a living unrelated donor kidney transplant. She received induction with thymoglobulin and was maintained on belatacept, mycophenolate mofetil (MMF) and prednisone. She was a CMV mismatch and was receiving Valganciclovir 900 mg daily prophylaxis. Patient also had a history of cryotherapy for abnormal Pap smear and endometrial ablation for abnormal uterine bleeding. Post-transplant, she developed antibody mediated rejection which was treated with plasmapheresis, intravenous immunoglobulins and eculuzimab. Eight weeks later, she developed acute cellular rejection and received thymoglobulin and methylprednisone. Serum creatinine settled down at 1.5 mg/dl. Four months post-transplant, she developed watery diarrhea while still being on valgancilovir. CMV DNA level was 123,000 IU/ml plasma. She was switched to IV ganciclovir while MMF was discontinued, and prednisone increased to 10 mg along with CytoGam infusions. CMV titers increased to up to 2 million copies and treatment was changed to IV Foscarnet because of CMV UL97 resistance confirmed by next generation sequencing.

*Results: Surveillance ultrasound (US) of kidney revealed a right adnexal cyst which led to transvaginal US showing increased endometrial fluid collection. Dilation and curettage failed thus leading to an MRI of pelvis which showed multilocular cysts within cervix (Figure 1) suspicious for adenoma malignum. As such, a cervical biopsy was performed which showed viral inclusions consistent with CMV infection (Figure 2). These CMV-induced changes were confirmed on immunohistochemistry.

*Conclusions: CMV occurs commonly in the most immunocompromised KTxR and may be manifested in atypical syndromes. Our case shows the first description of CMV cervicitis in a KTxR. The presence of an endocervical mass during episodes of CMV viremia should prompt a consideration of CMV disease of cervix.

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