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OBJECTIVE To perform a systematic review of the literature to estimate the prevalence and outcomes of occult tubal carcinoma in BRCA mutation carriers and high-risk patients undergoing risk-reducing salpingo-oophorectomy. DATA SOURCE A search was done using OVID MEDLINE, EMBASE, and ClinicalTrials.gov between 1946 and March 2019 with keywords and MeSH terms selected by an expert medical librarian and coauthors. METHODS OF STUDY SELECTION Two independent reviewers performed study selection with an initial screen on abstracts and a second on full articles. Articles were rejected if they were irrelevant to the study question, pertained to a different population or did not report occult tubal neoplasia. Quality was assessed using methodologic index for nonrandomized studies criteria. TABULATION, INTEGRATION, AND RESULTS Data were extracted and recorded in an Excel database. Forest plots for the prevalence of occult carcinoma were done using STATA. Among 2,402 studies assessed, 27 met the inclusion criteria for qualitative and quantitative analysis. A total of 6,283 patients underwent risk-reducing salpingo-oophorectomy between 2002 and 2019: 2,894 cases were BRCA1, 1,579 BRCA2, and 1,810 high-risk based on family history. Among these, 75 patients were diagnosed with occult tubal carcinoma at the time of surgery. The pooled prevalence was 1.2% (I=7.1%, P=.363) occurring at a median age of 53.2 years (range 42.4-67). In a subanalysis of 18 studies reporting follow-up data, 10 recurrences (18.7%, 95% CI 7.5-53%) and 24 cases of post-risk-reducing salpingo-oophorectomy peritoneal cancer (0.54%, 95% CI 0.4-1.9%) were reported after a median follow-up of 52.5 months. BRCA1, older age, and previous breast cancer were more often associated with occult malignancy. CONCLUSION Occult tubal carcinomas found at risk-reducing salpingo-oophorectomy in high-risk patients and BRCA mutation carriers have significant potential for recurrence despite the frequent administration of postoperative chemotherapy.

Introduction/Background Fortuitous cases of invasive tubal carcinoma in BRCA patients have been reported at the time of risk reducing salpingo-oophorectomy (RRSO). We performed a systematic review of the literature to determine the incidence of microinvasive tubal carcinoma in BRCA or high-risk patients undergoing RRSO, and evaluated treatment outcomes, recurrence and overall survival. Methodology The systematic review of the literature was done using OVID, EMBASE, Medline and PubMed with keywords selected by an expert medical librarian. Two independent reviewers performed study selection, data extraction, data analysis and quality assessment. Data was extracted,recorded in a database, and synthesized using tables. Quality was assessed using an index for non-randomized studies (MINORS). Statistical analysis was done with SAS. Results Among 2314 studies, 35 met inclusion criteria and were retained for qualitative analysis; 27 were used for quantitative analysis. A total of 6425 patients underwent RRSO between 1990 and 2019; 2963 were BRCA1 and 1605 BRCA2. Among these, 86 patients had occult tubal carcinoma. The pooled incidence of occult tubal carcinoma after RRSO in BRCA mutated or high-risk women was 1.34%, occurring at a median age of 54 years (range 42.4–67). After a median follow-up of 47.5 months, 13 recurrences and 27 cases of post-RRSO peritoneal cancer were reported. BRCA1, older age, previous breast cancer and extensive sampling of the fimbriated end were more often associated with occult malignancy. Additionally, we report a case of a 57 year old BRCA2+ patient with microinvasive tubal carcinoma found at prophylactic surgery. Despite chemotherapy and complete interval debulking, she recurred after 33 months with diffuse carcinomatosis, treated with second line calyx/carboplatin and olaparib maintenance. After 16 months, she developed acute myeloid leukaemia and died from this secondary malignancy. Conclusion Despite early diagnosis and aggressive chemotherapy, microinvasive tubal carcinoma found at the time of RRSO can recur and result in poor outcomes Disclosure Nothing to disclose