Management and sequelae of a local skene's gland adenocarcinoma recurrence

Primary adenocarcinoma of the Skene's gland is exceedingly rare, with only about two dozen well-documented cases in medical literature. Its immunophenotypic similarities to colorectal and prostatic adenocarcinoma create diagnostic challenges, and optimal management remains undefined. Treatment of recurrent disease is individualized and relies on little data to guide management.

A 62-year-old woman presented in 2017 with a periurethral mass diagnosed as moderately differentiated Skene's gland adenocarcinoma, demonstrating a gastrointestinal-type immunophenotype (CK20+, CDX2+, p53+, PSA focal+, partially CK7 and p16+, ER/PR-, PAX8-). She achieved complete clinical response after definitive chemoradiation with cisplatin in 2018. Biopsy-confirmed local recurrence was identified in 2020 with exam suggestive of fixation to the pubic bone, prompting interstitial high-dose-rate brachytherapy. Clinical presentation and subsequent imaging revealed progressive destructive changes of the pubic symphysis, a large pelvic abscess, and septic osteomyelitis. After treatment with antibiotics, subsequent MRI and PET findings suggested chronic osteomyelitis and radiation-induced necrosis. The patient underwent anterior pelvic exenteration with cystectomy, resection of the pubic symphysis, neovagina creation and ileal conduit urinary diversion. Her postoperative course was complicated by small-bowel obstruction, C. difficile colitis, hydronephrosis, nephrolithiasis, chronic kidney disease, and a right lower extremity DVT. At five-year follow-up, she remains alive with no evidence of disease.

This case demonstrates the diagnostic complexity of locally recurrent Skene's gland adenocarcinoma in the context of prior radiation and the curative potential of re-irradiation which ultimately necessitated an anterior exenteration in this case. Multidisciplinary evaluation is essential when managing periurethral malignancies in a previously irradiated pelvis.