Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review

Primary urethral adenocarcinoma in females is an extremely rare malignancy with unclear origin and only a few retrospective cases have been reported. The controversy continues to exist over the origin of primary urethral adenocarcinoma from periurethral glands (which include the Skene’s glands), ure...

Full description

Bibliographic Details
Main Authors: Junjie Tian, Ting Zhu, Zhijie Xu, Xiaoyi Chen, Yunfei Wu, Guanghou Fu, Baiye Jin
Format: Article
Language:English
Published: MDPI AG 2023-01-01
Series:Medicina
Subjects:
Online Access:https://www.mdpi.com/1648-9144/59/1/109
_version_ 1827623373272776704
author Junjie Tian
Ting Zhu
Zhijie Xu
Xiaoyi Chen
Yunfei Wu
Guanghou Fu
Baiye Jin
author_facet Junjie Tian
Ting Zhu
Zhijie Xu
Xiaoyi Chen
Yunfei Wu
Guanghou Fu
Baiye Jin
author_sort Junjie Tian
collection DOAJ
description Primary urethral adenocarcinoma in females is an extremely rare malignancy with unclear origin and only a few retrospective cases have been reported. The controversy continues to exist over the origin of primary urethral adenocarcinoma from periurethral glands (which include the Skene’s glands), urethritis glandularis or intestinal metaplasia. Herein, we report one case of a 49-year-old female with distal urethral adenocarcinoma who presented with obstructive voiding. Abdominal and pelvic CT scans and chest radiology were unremarkable. Biopsy of the mass confirmed urethral adenocarcinoma. The patient underwent partial ureterectomy and was disease-free at the 2-years follow-up period. We also present another extremely rare case of primary urethral adenocarcinoma with mucinous features in a 58-year-old female who initially complained of external urethral orifice itching with painless urethral bleeding and was treated with local excision. The patient has not received any neoadjuvant or adjuvant therapy, and experienced tumor recurrence, inguinal lymph nodes metastasis, and even local iliopsoas metastasis during over 10-years follow-up. In conclusion, our current study emphasizes the importance of imaging studies and biopsy in making an accurate preoperative diagnosis of this rare disease, and further highlights the role of multimodal therapy. A combination of radiotherapy, chemotherapy and surgery is recommended for the optimal local and distant disease control. Moreover, better medical compliance and regular follow-up are required in these patients.
first_indexed 2024-03-09T11:44:46Z
format Article
id doaj.art-b939d90c92b44d81b6debb20e0f4aae2
institution Directory Open Access Journal
issn 1010-660X
1648-9144
language English
last_indexed 2024-03-09T11:44:46Z
publishDate 2023-01-01
publisher MDPI AG
record_format Article
series Medicina
spelling doaj.art-b939d90c92b44d81b6debb20e0f4aae22023-11-30T23:24:28ZengMDPI AGMedicina1010-660X1648-91442023-01-0159110910.3390/medicina59010109Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature ReviewJunjie Tian0Ting Zhu1Zhijie Xu2Xiaoyi Chen3Yunfei Wu4Guanghou Fu5Baiye Jin6Department of Urology, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310024, ChinaDepartment of Urology, Jinyun People’s Hospital, Lishui 321400, ChinaDepartment of Urology, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310024, ChinaDepartment of Urology, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310024, ChinaDepartment of Urology, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310024, ChinaDepartment of Urology, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310024, ChinaDepartment of Urology, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310024, ChinaPrimary urethral adenocarcinoma in females is an extremely rare malignancy with unclear origin and only a few retrospective cases have been reported. The controversy continues to exist over the origin of primary urethral adenocarcinoma from periurethral glands (which include the Skene’s glands), urethritis glandularis or intestinal metaplasia. Herein, we report one case of a 49-year-old female with distal urethral adenocarcinoma who presented with obstructive voiding. Abdominal and pelvic CT scans and chest radiology were unremarkable. Biopsy of the mass confirmed urethral adenocarcinoma. The patient underwent partial ureterectomy and was disease-free at the 2-years follow-up period. We also present another extremely rare case of primary urethral adenocarcinoma with mucinous features in a 58-year-old female who initially complained of external urethral orifice itching with painless urethral bleeding and was treated with local excision. The patient has not received any neoadjuvant or adjuvant therapy, and experienced tumor recurrence, inguinal lymph nodes metastasis, and even local iliopsoas metastasis during over 10-years follow-up. In conclusion, our current study emphasizes the importance of imaging studies and biopsy in making an accurate preoperative diagnosis of this rare disease, and further highlights the role of multimodal therapy. A combination of radiotherapy, chemotherapy and surgery is recommended for the optimal local and distant disease control. Moreover, better medical compliance and regular follow-up are required in these patients.https://www.mdpi.com/1648-9144/59/1/109female urethral adenocarcinomamucinous adenocarcinomamultimodal therapyfollow-up
spellingShingle Junjie Tian
Ting Zhu
Zhijie Xu
Xiaoyi Chen
Yunfei Wu
Guanghou Fu
Baiye Jin
Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review
Medicina
female urethral adenocarcinoma
mucinous adenocarcinoma
multimodal therapy
follow-up
title Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review
title_full Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review
title_fullStr Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review
title_full_unstemmed Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review
title_short Management of Primary Female Urethral Adenocarcinoma: Two Rare Case Reports and Literature Review
title_sort management of primary female urethral adenocarcinoma two rare case reports and literature review
topic female urethral adenocarcinoma
mucinous adenocarcinoma
multimodal therapy
follow-up
url https://www.mdpi.com/1648-9144/59/1/109
work_keys_str_mv AT junjietian managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview
AT tingzhu managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview
AT zhijiexu managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview
AT xiaoyichen managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview
AT yunfeiwu managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview
AT guanghoufu managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview
AT baiyejin managementofprimaryfemaleurethraladenocarcinomatworarecasereportsandliteraturereview