Primary endometrioid adenocarcinoma developed at urethrovaginal septum has not been reported. A 61-yr-old woman presented with recurrent urinary tract infection. She had received hormone replacement treatment with estrogen and progesterone for 5 yr. A pinpoint ulceration at slightly elevated anterior vaginal wall was found and biopsy revealed endometrioid adenocarcinoma. Magnetic resonance imaging showed the 4.3 cm sized mass in urethrovaginal septum. She has undergone anterior pelvic exenteration, pelvic lymph node dissection, and urostomy with ileal conduit. Microscopic finding of the pathology revealed endometrioid adenocarcinoma. Co-existence of endometriosis was not identified. Tumor at urethrovaginal septum was difficult to be detected till growing to be bulky, because of vaginal axis, misunderstanding of the tumor as symphysis pubis, no definitive symptom, and its rarity. This is the first reported case of extraovarian endometrioid adenocarcinoma developed at the urethrovaginal septum. Understanding normal functional anatomy and meticulous physical examination are essential to detect this rare tumor in the urethrovaginal septum.
Carcinoma, Endometrioid/*diagnosis/pathology/surgery ; Diagnosis, Differential ; Endometrial Neoplasms/*diagnosis/pathology/surgery ; Female ; Humans ; Magnetic Resonance Imaging ; Middle Aged ; Urethral Neoplasms/*diagnosis/pathology/surgery ; Vaginal Neoplasms/*diagnosis/pathology/surgery

OBJECTIVES: Inhalation exposure to humidifier disinfectants has resulted to various types of health damages in Korea. To determine the epidemiological correlation necessary for presuming the legal causation, we aimed to develop a method to synthesize the entire evidence. METHODS: Epidemiological and toxicological studies are systematically reviewed. Target health problems are selected by criteria such as frequent complaints of claimants. Relevant epidemiologic studies are reviewed and the risk of bias and confidence level of the total evidence are evaluated. Toxicological literature reviews are conducted on three lines of evidence including hazard information, animal studies, and mechanistic studies, considering the source-to-exposure-to-outcome continuum. The confidence level of the body of evidence is then translated into the toxicological evidence levels for the causality between humidifier disinfectant exposure and health effects. Finally, the levels of epidemiological and toxicological evidence are synthesized. RESULTS: Under the Special Act revised in 2020, if the history of exposure and the disease occurred/worsened after exposure were approved, and the epidemiological correlation between the exposure and disease was verified, the legal causation is presumed unless the company proves the evidence against it. The epidemiological correlation can be verified through epidemiological investigations, health monitoring, cohort investigations and/or toxicological studies. It is not simply as statistical association as understood in judicial precedents, but a general causation established by the evidence as a whole, i.e., through weight-of-the-evidence approach. CONCLUSIONS The weight-of-the-evidence approach differs from the conclusive single study approach and this systematic evidence integration can be used in presumption of causation.