Adenocarcinoma arising in urinary bladder endocervicosis

Abstract

Endocervicosis is a rare benign condition characterized by the presence of endocervical-type mucinous glands. Urinary bladder endocervicosis forms an elevated lesion in the posterior wall of the urinary bladder and is sometimes misdiagnosed as a malignant tumor clinically and pathologically. Herein we describe the first case of adenocarcinoma arising in urinary bladder endocervicosis. The patient, a 58-year-old woman, presented with asymptomatic hematuria. Cystoscopy revealed a nodular mass measuring 4 cm in diameter in the posterior wall, and total cystectomy was performed. Histology revealed that the elevated lesion of the bladder wall was composed of haphazard proliferation of cystic glands lined by benign endocervical-type epithelium. An adenocarcinoma arose at the center of this endocervicosis. Mucin histochemistry revealed the presence of sulfomucin in both the endocervicosis and adenocarcinoma components. Immunohistochemically, the endocervicosis was positive for cytokeratin (CK) 7, AE1/ AE3, CAM5.2, HBME1, CA19-9, and estrogen receptor (ER), and negative for CK20, CDX2, progesterone receptor (PR), MUC5AC, and β-catenin. The adenocarcinoma showed similar immunohistochemical results, except for loss of ER expression and a slight increase in the ratio of Ki-67-positive cells. This case indicates that endocervicosis, known as a benign lesion, harbors the possibility of malignant transformation.

Bladder endocervicosis – A rare diagnosis with a silent presentation

“…This is supported by the fact that more than one type of mullerian tissue can be present and the lesions are predominantly located in the posterior wall of the bladder covered by the posterior peritoneum. [ 2 3 5 ] The posterior location of the lesion was also a feature seen in the present case.…”

“…Bladder endocervicosis is characterized by the presence of endocervical mucinous epithelium in the bladder wall. [ 2 ]…”

“…Definitive diagnosis is established by histopathological analysis, based on cellular architecture and cytological findings. [ 2 3 5 ] Histologically, it is characterized by the presence of endocervical mucin-like epithelium in the bladder wall and may be accompanied by cystic dilatation. [ 1 ] In cystitis glandularis and cystic cystitis, the glands are confined to the muscularis propria layer without extension to the mucosal muscularis layer.…”

“…Bladder endocervicosis is a benign, extremely rare condition, that is defined by the presence of ectopic endocervical tissue in the bladder wall. [ 1 2 ] Usually, it is associated with endometrial or endosalpingiosis ectopic tissue. The simultaneous presence of at least two forms was designated mullerianosis by Young and Clement, in 1996.…”

“…The most commonly affected organs by this disease are urinary bladder, followed by the vagina and uterine cervix. [ 2 ] Clinical manifestation is nonspecific, but suprapubic pain, hematuria, urinary frequency, and urgency are classical symptoms associated with this disease. Dyspareunia and catamenial exacerbation can also be clinical findings in some cases.…”