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20 October 2021: Articles

A 46-Year-Old Woman with Primary Infertility and a Diagnosis of Microcystic Stromal Tumor of the Ovary Confirmed by Histology and Gene Sequencing: A Case Report and Review of the Literature

Challenging differential diagnosis, Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)

Maria A. Arafah A* , Layla AlBreacan A , Khalid Akkour B , Suliman Alomar B

DOI: 10.12659/AJCR.933528

Am J Case Rep 2021; 22:e933528

Table 1. A summary of all previously published cases of an ovarian microcystic stromal tumor.

CaseReferenceAgeMolecular findingsManagementFollow-up (months)
1Irving and Young, 2009 []1 62NATAH, BSO, LND, OMNA
245TAH, BSO, peritoneal biopsies
351TAH, BSO, OM, appendectomy, pelvic washings
558TAH, BSO, LND, peritoneal biopsy, pelvic washings
845LSO, endometrial curettage
1145TAH, BSO
1255TAH, BSO
1344TAH, BSO
1537TAH, BSO
1639RSO, pelvic washings
17Maeda et al, 2011 []4 33CTNNB1 mutation1 GnRHa, RSO, enucleation of uterine masses, partial OMNED (14)
1841CTNNB1 mutationRSO, enucleation of uterine leiomyomaNED (4)
19Yang and Bhattacharjee, 2014 []5 45NAOpen ovarian tumor resectionNA
20Niu and Peng, 2014 []6 42NANANA
21Irving et al, 2015 []7 One of the cases was a 39-year-old patient with FAPCTNNB1 mutation in 8/14 patientsNANA
25Kang et al, 2015 []8 41CTNNB1 mutationLaparoscopic LSONA
26Lee et al, 2015 []9 40APC mutationLSO, right ovarian wedge resection, total colectomyNED (9)
27Bi et al, 2015 []10 69CTNNB1 mutationLSONED (60)
2829CTNNB1 wild-typeLO, right ovarian biopsyNED (18)
2940CTNNB1 mutationLONED (7)
3065CTNNB1 mutationTAH, BSONA
3157CTNNB1 mutationTAH, BSONED (59)
3241CTNNB1 wild-typeTAH, BSO, OM, appendectomyNED (2)
33Podduturi et al, 2015 []11 50CTNNB1 mutationHysterectomy, BSO, pelvic and para-aortic LND, OM, staging biopsiesNA
34Chen et al, 2015 []12 47NALSONED (18)
35Gunes et al, 2015 []13 52CTNNB1 mutationTAH, BSO, OMNED (3)
36Lee et al, 2016 []14 24CTNNB1 mutationLaparoscopic LSONED (8)
3731CTNNB1 mutationOpen LSO, para-aortic LND, peritonectomyNED (3)
38Liu, et al, 2016 []15 23APC mutationLaparoscopic hysterectomy, RO, BSNA
39Murakami et al, 2017 []16 26CTNNB1 wild-type4 GnRHa, LSONED (36)
40Na et al, 2017 []17 33CTNNB1 mutationLaparoscopic RSO, partial OMNED (57)
4131CTNNB1 mutationLSO, Para-aortic LNDNED (20)
42Meurgey et al, 2017 []18 46FOXL2 and DICER1 wild-types2 LSO, 1 RSONA
45Qureshi et al, 2017 []19 50NALONA
46Jeong et al, 2018 []20 66NABSO, bilateral pelvic and para-aortic LND, infra-colic OM, peritoneal biopsiesNED (18)
47Zhang et al, 2018 []21 33APC mutationRight laparoscopic cystectomyIntraovarian and extraovarian recurrence (108)
48Hasanzadeh et al, 2019 []23 60NATAH, BSONED (15)
49McCluggage et al, 2019 []24 61CTNNB1 mutationBSONA
5056CTNNB1 mutationHysterectomy, BSO
5145NAHysterectomy, BSO
5271CTNNB1 mutationBSO and endometrial curettage
53Liu et al, 2019 []25 46NALaparoscopic RSONED (54)
5456NALaparoscopic hysterectomy, BSONED (46)
55Deng et al, 2020 []26 25NALaparoscopic resection of ovarian tumorNED (4)
56He et al, 2020 []27 33NALaparoscopic resection of ovarian tumorNED (19)
57Current case46CTNNB1 mutationOpen LSO, infra-colic OM, bilateral peritoneal biopsies, left pelvic LND, appendectomyNED (24)
a Follow-up data were available for 7 patients with no evidence of disease after a mean period of 51 months.
b The study evaluated 15 patients; 11 were included in Irving et al, 2009 and 4 were additional cases.
c In 2017, the cases were re-examined by McCluggage et al for mutations. Two cases were initially reported to have wild-type showed mutations and 3 cases showed mutations including the patient with FAP.
BEMP – benign endometrial polyp; BS – bilateral salpingectomy; BSO – bilateral salpingo-oophorectomy; EEA – endometrioid endometrial adenocarcinoma; FAP – familial adenomatous polyposis; GnRHa – gonadotropic releasing hormone analog; LND – lymph nodes dissection; LO – left oophorectomy; LSO – left salpingo-oophorectomy; NA – not available; NED – no evidence of disease; OM – omentectomy; RO – right oophorectomy; RSO – right salpingo-oophorectomy; TAH – total abdominal hysterectomy.

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923