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

Maria A. Arafah; Layla AlBreacan; Khalid Akkour; Suliman Alomar

doi:10.12659/AJCR.933528

20 October 2021 : Case report Saudi Arabia

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^1ABCDEF*, Layla AlBreacan

^2ABCDEF, Khalid Akkour^3BCD, Suliman Alomar^4BCD

DOI: 10.12659/AJCR.933528

Am J Case Rep 2021; 22:e933528

Authors information Article notes Copyright and License information

Background Case Report Discussion Conclusions References:

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

Case	Reference	Age	Molecular findings	Management	Follow-up (months)
1	Irving and Young, 2009 []¹	62	NA	TAH, BSO, LND, OM	NA
2		45		TAH, BSO, peritoneal biopsies
3		51		TAH, BSO, OM, appendectomy, pelvic washings
4		29		LO
5		58		TAH, BSO, LND, peritoneal biopsy, pelvic washings
6		26		BSO
7		29		RO
8		45		LSO, endometrial curettage
9		63		RO
10		56		BSO
11		45		TAH, BSO
12		55		TAH, BSO
13		44		TAH, BSO
14		36		LSO
15		37		TAH, BSO
16		39		RSO, pelvic washings
17	Maeda et al, 2011 []⁴	33	CTNNB1 mutation	1 GnRHa, RSO, enucleation of uterine masses, partial OM	NED (14)
18	Maeda et al, 2011 []⁴	41	CTNNB1 mutation	RSO, enucleation of uterine leiomyoma	NED (4)
19	Yang and Bhattacharjee, 2014 []⁵	45	NA	Open ovarian tumor resection	NA
20	Niu and Peng, 2014 []⁶	42	NA	NA	NA
21	Irving et al, 2015 []⁷	One of the cases was a 39-year-old patient with FAP	CTNNB1 mutation in 8/14 patients	NA	NA
22
23
24
25	Kang et al, 2015 []⁸	41	CTNNB1 mutation	Laparoscopic LSO	NA
26	Lee et al, 2015 []⁹	40	APC mutation	LSO, right ovarian wedge resection, total colectomy	NED (9)
27	Bi et al, 2015 []¹⁰	69	CTNNB1 mutation	LSO	NED (60)
28		29	CTNNB1 wild-type	LO, right ovarian biopsy	NED (18)
29		40	CTNNB1 mutation	LO	NED (7)
30		65	CTNNB1 mutation	TAH, BSO	NA
31		57	CTNNB1 mutation	TAH, BSO	NED (59)
32		41	CTNNB1 wild-type	TAH, BSO, OM, appendectomy	NED (2)
33	Podduturi et al, 2015 []¹¹	50	CTNNB1 mutation	Hysterectomy, BSO, pelvic and para-aortic LND, OM, staging biopsies	NA
34	Chen et al, 2015 []¹²	47	NA	LSO	NED (18)
35	Gunes et al, 2015 []¹³	52	CTNNB1 mutation	TAH, BSO, OM	NED (3)
36	Lee et al, 2016 []¹⁴	24	CTNNB1 mutation	Laparoscopic LSO	NED (8)
37	Lee et al, 2016 []¹⁴	31	CTNNB1 mutation	Open LSO, para-aortic LND, peritonectomy	NED (3)
38	Liu, et al, 2016 []¹⁵	23	APC mutation	Laparoscopic hysterectomy, RO, BS	NA
39	Murakami et al, 2017 []¹⁶	26	CTNNB1 wild-type	4 GnRHa, LSO	NED (36)
40	Na et al, 2017 []¹⁷	33	CTNNB1 mutation	Laparoscopic RSO, partial OM	NED (57)
41	Na et al, 2017 []¹⁷	31	CTNNB1 mutation	LSO, Para-aortic LND	NED (20)
42	Meurgey et al, 2017 []¹⁸	46	FOXL2 and DICER1 wild-types	2 LSO, 1 RSO	NA
43		37
44		47
45	Qureshi et al, 2017 []¹⁹	50	NA	LO	NA
46	Jeong et al, 2018 []²⁰	66	NA	BSO, bilateral pelvic and para-aortic LND, infra-colic OM, peritoneal biopsies	NED (18)
47	Zhang et al, 2018 []²¹	33	APC mutation	Right laparoscopic cystectomy	Intraovarian and extraovarian recurrence (108)
48	Hasanzadeh et al, 2019 []²³	60	NA	TAH, BSO	NED (15)
49	McCluggage et al, 2019 []²⁴	61	CTNNB1 mutation	BSO	NA
50		56	CTNNB1 mutation	Hysterectomy, BSO
51		45	NA	Hysterectomy, BSO
52		71	CTNNB1 mutation	BSO and endometrial curettage
53	Liu et al, 2019 []²⁵	46	NA	Laparoscopic RSO	NED (54)
54	Liu et al, 2019 []²⁵	56	NA	Laparoscopic hysterectomy, BSO	NED (46)
55	Deng et al, 2020 []²⁶	25	NA	Laparoscopic resection of ovarian tumor	NED (4)
56	He et al, 2020 []²⁷	33	NA	Laparoscopic resection of ovarian tumor	NED (19)
57	Current case	46	CTNNB1 mutation	Open LSO, infra-colic OM, bilateral peritoneal biopsies, left pelvic LND, appendectomy	NED (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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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

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