title: Outcome of unexpected adnexal neoplasia discovered during risk reduction salpingo-oophorectomy in women with germ-line BRCA1 or BRCA2 mutations.
abstract: OBJECTIVE
  This study computed the risk of clinically silent adnexal neoplasia in women with germ-line BRCA1 or BRCA2 mutations (BRCA(m+)) and determined recurrence risk.
  
  
  METHODS
  We analyzed risk reduction salpingo-oophorectomies (RRSOs) from 349 BRCA(m+) women processed by the SEE-FIM protocol and addressed recurrence rates for 29 neoplasms from three institutions.
  
  
  RESULTS
  Nineteen neoplasms (5.4%) were identified at one institution, 9.2% of BRCA1 and 3.4% of BRCA2 mutation-positive women. Fourteen had a high-grade tubal intraepithelial neoplasm (HGTIN, 74%). Mean age (54.4) was higher than the BRCA(m+) cohort without neoplasia (47.8) and frequency increased with age (p < 0.001). Twenty-nine BRCA(m+) patients with neoplasia from three institutions were followed for a median of 5 years (1-8 years.). One of 11 with HGTIN alone (9%) recurred at 4 years, in contrast to 3 of 18 with invasion or involvement of other sites (16.7%). All but two are currently alive. Among the 29 patients in the three institution cohort, mean ages for HGTIN and advanced disease were 49.2 and 57.7 (p = 0.027).
  
  
  CONCLUSIONS
  Adnexal neoplasia is present in 5-6% of RRSOs, is more common in women with BRCA1 mutations, and recurs in 9% of women with HGTIN alone. The lag in time from diagnosis of the HGTIN to pelvic recurrence (4 years) and differences in mean age between HGTIN and advanced disease (8.5 years) suggest an interval of several years from the onset of HGTIN until pelvic cancer develops. However, some neoplasms occur in the absence of HGTIN.
authors: Division of Women's and Perinatal Pathology, Department of Pathology, Brigham and Women's Hospital, USA.
authors: James R
authors: JR
authors: Conner
authors: Division of Women's and Perinatal Pathology, Department of Pathology, Brigham and Women's Hospital, USA.
authors: Emily
authors: E
authors: Meserve
authors: Cell-Netix, Seattle, USA.
authors: Ellen
authors: E
authors: Pizer
authors: Dana Farber/Brigham and Women's Cancer Center, Boston, MA, USA.
authors: Judy
authors: J
authors: Garber
authors: Department of Pathology, University of Michigan Medical School, Ann Arbor, USA.
authors: Michael
authors: M
authors: Roh
authors: Fred Hutchinson Cancer Research Center, Seattle, WA, USA.
authors: Nicole
authors: N
authors: Urban
authors: University of Washington School of Medicine, Seattle, WA, USA.
authors: Charles
authors: C
authors: Drescher
authors: Division of Women's and Perinatal Pathology, Department of Pathology, Brigham and Women's Hospital, USA.
authors: Bradley J
authors: BJ
authors: Quade
authors: Division of Gynecologic Pathology, Department of Obstetrics and Gynecology, Brigham and Women's Hospital, Boston, USA.
authors: Michael
authors: M
authors: Muto
authors: Division of Women's and Perinatal Pathology, Department of Pathology, Brigham and Women's Hospital, USA.
authors: Brooke E
authors: BE
authors: Howitt
authors: Department of Obstetrics and Gynecology, University of Michigan Medical School, Ann Arbor, USA.
authors: Mark D
authors: MD
authors: Pearlman
authors: Division of Gynecologic Pathology, Department of Obstetrics and Gynecology, Brigham and Women's Hospital, Boston, USA.
authors: Ross S
authors: RS
authors: Berkowitz
authors: Division of Gynecologic Pathology, Department of Obstetrics and Gynecology, Brigham and Women's Hospital, Boston, USA.
authors: Neil
authors: N
authors: Horowitz
authors: Division of Women's and Perinatal Pathology, Department of Pathology, Brigham and Women's Hospital, USA. Electronic address: ccrum@partners.org.
authors: Christopher P
authors: CP
authors: Crum
authors: Division of Gynecologic Pathology, Department of Obstetrics and Gynecology, Brigham and Women's Hospital, Boston, USA.
authors: Colleen
authors: C
authors: Feltmate
mesh_terms: D000328:Adult; D000368:Aged; D005260:Female; D019398:Genes, BRCA1; D024522:Genes, BRCA2; D020022:Genetic Predisposition to Disease; D018095:Germ-Line Mutation; D006801:Humans; D007044:Hysterectomy / Q000379:methods; D008875:Middle Aged; D009364:Neoplasm Recurrence, Local / Q000235:genetics / Q000473:pathology; D010051:Ovarian Neoplasms / Q000235:genetics* / Q000473:pathology / Q000517:prevention & control / Q000601:surgery*; D010052:Ovariectomy / Q000379:methods; D012307:Risk Factors; D016896:Treatment Outcome
keywords: BRCA; Fallopian; Ovarian cancer; Serous cancer