European Journal of Obstetrics & Gynecology and Reproductive Biology 185 (2015) 78–82

Ovarian-sparing local mass excision for ovarian fibroma/fibrothecomain premenopausal women

Yeon Jean Cho a, Hee Suk Lee a, Joo Myung Kim b, Soo Yoon Lee b, Taejong Song c,Seok Ju Seong d, Mi-La Kim d,*a Department of Obstetrics and Gynecology, Dong-A University Medical Center, Dong-A University, College of Medicine, Busan, Republic of Koreab Department of Obstetrics and Gynecology, Cheil General Hospital and Women’s Healthcare Center, Kwandong University College of Medicine, Seoul,

Republic of Koreac Department of Obstetrics and Gynecology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul, Republic of Koread Department of Obstetrics and Gynecology, CHA Gangnam Medical Center, CHA University, Seoul, Republic of Korea

A R T I C L E I N F O

Article history:

Received 5 October 2014

Received in revised form 19 November 2014

Accepted 27 November 2014

Keywords:

Fibroma

Fibrothecoma

Laparoscopy

Laparotomy

Ovary

Excision

A B S T R A C T

Objectives: To evaluate the recurrence rate of ovarian fibroma/fibrothecoma and reproductive outcomes

following ovarian-sparing local mass excision in premenopausal women.

Study design: A retrospective cohort study was performed at two gynecologic surgery centers using data

collected between January 2005 and December 2011. It included premenopausal patients treated with

ovarian-sparing local mass excision and pathologically proven ovarian fibroma/fibrothecoma who were

followed up for at least 6 months. The recurrence of fibroma/fibrothecoma and pregnancy outcomes in

those who wanted to conceive after local mass excision were collected and analyzed.

Results: The mean age of the patients (n = 50) was 33.3 � 6.9 years (range, 20–50 years), and the mean

follow-up duration was 26.6 � 19.2 months (range, 6–88 months). Fibroma was present in 40 patients,

fibrothecoma in 7, and cellular fibroma in 3. Natural conception occurred in 11 of the 12 patients who became

pregnant during the follow-up period. On follow-up ultrasonography, one patient experienced recurrent

disease, 50 months after initial surgery, resulting in a crude overall recurrence rate of only 2%.

Conclusion: Given the 2% recurrence rate of ovarian fibroma/fibrothecoma following ovarian sparing

local mass excision, local mass excision appears to be an effective surgical option in women of

reproductive age.

� 2014 Elsevier Ireland Ltd. All rights reserved.

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Introduction

Ovarian fibromas/fibrothecomas are solid tumors of the ovarythat account for 1–4.7% of all ovarian neoplasms. They areclassified as sex cord-stromal tumors and are characterized byovergrowth of the ovarian stroma [1–5]. They can occur at any age,but most frequently are identified during middle age (mean,48 years), with a recent study reporting that, of 97 patientsdiagnosed with fibroma/fibrothecoma, 49.5% were <40 years old[6]. However, <10% occur in young women before the age of30 years, and they occur rarely in children: when they do occur,they are associated with genetic syndromes such as Gorlin–Goltz,Maffucci, and Sotos syndromes [4,5,7–11].

* Corresponding author. Tel.: +82 2 3468 3677; fax: +82 2 558 2638.

E-mail address: mila76@naver.com (M.-L. Kim).

http://dx.doi.org/10.1016/j.ejogrb.2014.11.042

0301-2115/� 2014 Elsevier Ireland Ltd. All rights reserved.

With the recent trend of delayed childbearing, there has beenan increased demand for ovarian-sparing surgery to preservereproductive potential or ovarian hormonal function. However,accurate preoperative diagnosis of ovarian fibroma/fibrothecomais challenging: instead, malignancy is often diagnosed, owing tothe presence of ascites or pleural effusions (reported in 10% of theovarian fibroma patients), elevated serum CA125 levels (positiveassociation with tumor size), and the solid nature of the tumor[4,12]. As a consequence, salpingo-oophorectomy or oophorecto-my is commonly planned. It has been shown, however, that theprognosis of ovarian fibroma/fibrothecoma is extremely good andrecurrence is uncommon [12,13]. Favorable clinical outcomesare seen even in mitotically active cellular fibroma (histologiccriteria are dense cellular proliferation of fibroblasts with mild tomoderate nuclear atypia, a mitotic count of �3 mitotic figures(MFs)/10 high power fields (HPFs), and low malignant potential),which is distinct from fibrosarcoma (moderate to severe nuclearatypia, a mitotic counts of �4 MFs/10 HPFs, and clinical

Table 1Clinical characteristics of the patients who were treated by ovarian sparing

tumorectomy.

Characteristics Mean � SD or, median (range),

number (%)

Age (years) 33.3 � 6.9 (31.5, 20–50)

Parity

0 39(78%)

1 4 (8%)

�2 7 (14%)

Body mass index (kg/m2) 21.4 � 3.2 (20.7, 16.8–30.1)

Associated symptoms

Incidentally detected, no symptoms 32 (64%)

Abdominal discomfort or pain 13 (26%)

Palpable abdominal mass 3 (6%)

Vaginal bleeding 2 (4%)

Associated pelvic pathology

Myoma 15 (30%)

Myoma + adenomyosis 2 (4%)

Myoma + ovarian endometrioma 3 (6%)

Ovarian endometrioma 1 (2%)

Ovarian cysts (mucinous/serous

cystadenoma)

2 (4%)

Carcinoma in situ of the cervix 1 (2%)

Final pathologic results

Fibroma 40 (80%)

Fibrothecoma 7 (14%)

Cellular fibroma 3 (6%)

Follow-up durations (months) 26.6 � 19.2 (21, 6–88)

SD, standard deviations.

Y.J. Cho et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 185 (2015) 78–82 79

malignancy) [14,15]. For these reasons, when ovarian fibroma/fibrothecoma is suspected, ovarian-sparing surgery could be afeasible option, especially for young patients of reproductive age.

Little is known, however, about the recurrence rate andreproductive outcomes after ovarian sparing local mass excisionfor treatment of ovarian fibroma/fibrothecoma. Therefore, in thepresent study we aimed to present our experience with ovarian-sparing local mass excision for ovarian fibroma/fibrothecoma anddiscuss the postoperative ovarian fibroma/fibrothecoma recur-rence rate and reproductive outcomes in premenopausal women.

Materials and methods

We performed a retrospective review of patients who hadundergone surgical treatment and were diagnosed pathologicallywith ovarian fibroma or fibrothecoma between January 2005 andDecember 2011 at two gynecologic surgery centers in the Republicof Korea (CHA Gangnam Medical Center, CHA University and CheilGeneral Hospital and Women’s Healthcare Center, KwandongUniversity College of Medicine). Women who were treated withovarian-sparing local mass excision with or without a hysterecto-my and followed up with transvaginal ultrasonography (TVS) atleast 6 months after the initial surgery were included. Patients whowere treated with oophorectomy or salpingo-oophorectomy wereexcluded. This study was approved by the respective InstitutionalReview Boards of both institutions.

Demographic data, including patient age, body mass index(BMI), parity, menopausal status, presenting symptoms, familialhistory of genetic syndromes, and associated gynecologic diseaseswere analyzed. The largest diameter of the ovarian tumor (if theovarian masses were bilateral, the sum of the larger diameters wasrecorded), ovarian tumor location, sonographic characteristics, andassociated ascites were recorded. Operative data were collected,including the operative procedure(s), associated adhesion ortorsion of the mass, intraoperative frozen section, and methodsfor specimen removal. After the surgery, we recommended follow-up visits for transvaginal scanning (TVS), every six months for thefirst year, and every 6–12 months thereafter. All follow-up data,including recurrence of ovarian mass on TVS, reoperation data, andsubsequent pregnancy outcomes were retrospectively collectedand analyzed.

Statistical analyses were performed using SPSS for Windowsversion 20 (IBM Corp., Armonk, NY). The Shapiro–Wilk test wasutilized to test the normality of the data. Descriptive data areexpressed as mean � standard deviation (SD). Skewed data arereported as median (range).

Results

During the study period, a total of 275 patients were surgicallymanaged and pathologically diagnosed with ovarian fibromas/fibrothecomas in the two centers. Of these patients, 106 (38.5%)underwent ovarian sparing local mass excision, and 50 of thesepatients were premenopausal and followed up for at least6 months after the initial surgery; therefore, 50 patients wereincluded.

The baseline characteristics of the patients are shown inTable 1. The mean age was 33.3 � 6.9 years (range, 20–50 years), andthe mean follow-up duration was 26.6 � 19.2 months (range, 6–88months). No-one had a familial history of a genetic syndrome. Themajority of the ovarian masses were detected incidentally; only18 patients (36%) had symptoms such as pain, a palpable abdominalmass, or vaginal bleeding. The majority of the patients (n = 40, 80%)had fibroma. Associated pelvic pathology such as myoma, adeno-myosis, and ovarian cysts was present in 24 patients (48%), and these

conditions were either concurrently addressed during the ovariansparing local mass excision or a cesarean section (n = 2).

The perioperative findings of the ovarian masses are presentedin Table 2. The mean diameter was 4.7 cm (range, 1–9.3 cm), andtwo patients had bilateral tumors. The preoperative diagnosis wasbenign ovarian tumor or fibroma in the majority of patients (n = 28,56%), followed by uterine myoma in 13 (26%). In three (6%)patients, an ovarian solid mass was detected incidentally duringsurgery for other gynecologic conditions. Thirty-six patients (72%)underwent laparoscopic surgery, including one laparoendoscopicsingle-site (LESS) surgery. Of the 14 (28%) patients who underwenta laparotomy, only one was for an ovarian mass alone, while theremaining 13 patients were operated simultaneously with otherconditions, including two cesarean sections. All of the intraoper-ative frozen section (n = 11) results were correlated with the finalpathology. Thirty-three patients (66%) had a mass connected to apedicle or protruding the ovarian surface more than 50% of thesolid mass (Fig. 1). The remaining ovarian masses were surroundedby normal ovarian tissue (Fig. 2). During the laparoscopic surgery,the specimen was removed by electronic morcellation within theendopouch in 34 of 36 (94.4%) patients. The excised mass wasplaced inside the endopouch under direct visualization. Anelectronic morcellator was inserted at the umbilicus or rightlower trocar site after dilating the initial incision up to 15 mm andmorcellated the mass within the endopouch without spreading thefragments of mass beyond the endopouch. One patient needed asmall laparotomy incision because the mass was too hard andmorcellation failed. In the patient who was treated by LESS surgery,the specimen was removed via the umbilical incision withoutmorcellation.

Subsequent pregnancy outcomes are listed in Table 3. Naturalconception occurred in 11 of the 12 patients who became pregnantduring the follow-up period. On the follow-up ultrasonography,only one patient experienced recurrent disease, 50 monthsafter the initial surgery. She underwent a second operation bylaparoscopic right salpingo-oophorectomy, and the pathologicresults revealed a fibrothecoma. Therefore, the crude overallrecurrence rate was only 2%.

Table 2Perioperative findings of the ovarian mass.

Characteristics Mean � SD or, median

(range), number (%)

Largest diameter (cm) 4.7 � 2.2 (4.6, 1–9.3)

Location of mass

Right 21 (42%)

Left 27 (54%)

Bilateral 2 (4%)

Sonographic characteristics

Solid 25 (50%)

Solid + cystic 11 (22%)

Cystic 11 (22%)

Not found in sono (incidentally detected) 3 (6%)

Associated with ascites 2 (4%)

Presence of uterine myomas 20 (40.8%)

Serum CA 125 level, U/mL (n = 24) 38.2 � 51.6

(25.6, 8.3–274.3)

�35 16 (66.7%)

>35 8 (33.3%)

Preoperative diagnosis

Benign ovarian tumor including fibroma 28 (56%)

Myoma 13 (26%)

Fibroma or subserosal myoma 5 (10%)

Incidentally detected during the operation 3 (6%)

Ovarian malignant tumor 1 (2%)

Operation methods

Laparoscopy 36 (72%)

Laparotomy 14 (28%)

Operative procedure

Mass excision only 25 (50%)

Mass excision + cesarean section 2 (4%)

Mass excision + hysterectomy 3 (6%)

Mass excision + myomectomy 16 (32%)

Mass excision + other ovarian cyst enucleation 3 (6%)

Mass excision + LEEP 1 (2%)

Intraoperative frozen section 11 (22%)

Surface adhesion 0 (0%)

Torsion of mass 0 (0%)

Protruding mass from the ovarian surface 32 (64%)

SD, standard deviations; LEEP, loop electrosurgical excision procedure.

Y.J. Cho et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 185 (2015) 78–8280

Comments

To the best of our knowledge, these findings are the first to bereported from follow-up results of ovarian-sparing local massexcision for ovarian fibroma/fibrothecoma. The crude recurrencerate of ovarian fibroma/fibrothecoma was only 2%. Moreover,14 pregnancies were recorded during the follow-up period in12 patients who wanted to conceive; the majority of these were aresult of natural conception, while one patient conceived by invitro fertilization owing to male factor infertility.

Ovarian fibromas/fibrothecomas are the most common benignsolid tumors of the ovary, and are frequently identified duringmiddle age [1]. In the present study, 134 of 275 patients (48.7%)were �40 years old at the time of the surgery. Fifteen patients wereaged in their 20s, and the youngest patient was 20 years old.

While the treatment of choice for ovarian fibroma/fibrothecomais surgical removal, the choice of treatment method depends on thesize and nature of the ovarian mass, age of the patient, andpreoperative diagnosis. Recently, the laparoscopic approach hasbeen considered for the management of ovarian fibromas, and a fewcase series have been reported [6,16,17]. There have also been anumber of small case series and case reports of ovarian-sparing localmass excision for ovarian fibroma/fibrothecoma [5,6,11,13,16,18–24]. However, the concern with the use of ovarian local massexcision for solid ovarian tumors is the possibility of malignantdisease. Irving et al. reported that 6% of cellular fibromas and 10% ofmitotically active cellular fibroma cases showed ovarian surfaceadhesion or rupture and that 11% and 13% of these patients,respectively, demonstrated related extraovarian involvement [14].

The typical imaging findings of fibroma/fibrothecoma are asfollows: (1) sonographic findings show round, oval, or lobulatedsolid tumors and cast striped shadows owing to the cellularbundles and intersecting strips of hyaline-like collagen and fibroustissue; (2) computed tomography (CT) findings appear as a solidmass with delayed contrast enhancement; and (3) magneticresonance imaging (MRI) examinations exhibit low signal intensityon T2-weighted images and weak contrast enhancement. If thepreoperative findings show atypical presentation, and associatedwith elevated levels of CA125 or ascites/pleural effusion, carefulintraoperative ovarian surface evaluation, and frozen biopsyshould be considered before more radical surgery such asoophorectomy or salpingo-oophorectomy in younger patients[6,25,26]. Instead, simple excision of the masses (leaving only thenormal-appearing ovarian tissue) is preferred in younger patients[21]. The decision to perform conservative surgery is justified bythe benign nature of the fibroma/fibrothecoma and the preserva-tion of ovarian function for future fertility. The risk of recurrence offibroma/fibrothecoma is not well known, but it does not justifymore invasive surgery. Of note, regular ultrasound pelvic evalua-tions are required after ovarian-sparing surgery [22].

During the laparoscopic operation, removal of the specimen isdifficult and time-consuming, and requires the use of specificmethods. We employed a technique of electronic morcellation of theexcised mass within the endopouch. The technique allows anyfragments of the tumor dropped during morcellation to be collectedinside the pouch without contaminating the peritoneal cavity.

An exophytic growth pattern is not rare in ovarian fibroma/fibrothecoma. Sivanesaratnam et al. reported a high incidence(50%) of long pedicles off the fibroma, and Oh et al. also reportedexophytic growth patterns with pedicle-like structures in thefibroma by MRI findings in 27.1% of patients [4,26]. Usually, anexophytic ovarian fibroma is associated with the normal-appear-ing oval shape of the affected ovary, whereas an affected ovarywith an endophytic fibroma appears crescent-shaped along theperipheral margin of the tumor [26]. Upon pathologic examinationof the removed ovaries, Oh et al. reported that the majority of theovarian fibromas had remaining normal ovarian tissue of varyingvolumes even though the remaining ipsilateral ovarian tissue wasnot detected on MRI [26]. In the present study, 32 of the 50 patientsexhibited an exophytic pattern, with pedicle-like structures or>50% of the mass protruding from the normal ovarian surface. Theremaining 18 patients exhibited an endophytic pattern, and �50%of the mass surface was covered with normal ovarian tissue. Eventhese endophytic masses were surgically separated from thenormal ovarian tissue with ease.

Genetic syndromes, such as Gorlin–Goltz, Maffucci, and Sotossyndromes, have been associated with ovarian fibroma in youngerwomen in previous studies [4,5,7–11]. Interestingly, all thereported cases of recurrent ovarian fibroma have been related toGorlin–Goltz syndrome [13,21–24]. Until recently, the majority ofsurgical interventions are likely to have been oophorectomy orsalpingo-oophorectomy with or without hysterectomy, and we areunaware of any case reports of recurrent disease followingovarian-sparing local mass excision in the absence of a geneticsyndrome. Despite the fact that none of the patients in the presentstudy had a familial history of genetic syndromes, we experienceda recurrent case, 50 months after the initial surgery. During theinitial surgery, bilateral ovarian masses (right, 1.5 cm; left, 1.7 cm)were incidentally detected during a cesarean section and excised,sparing the normally appearing ovarian tissue. During the routineTVS examination at 50 months, a 7.9 cm sized semisolid masswas detected in the right ovary. A laparoscopic right salpingo-oophorectomy was conducted due to fear of recurrent disease; theleft ovary was grossly normal. Fibrothecoma was confirmedpathologically.

Fig. 1. Operative findings of the right ovarian endophytic fibroma. (A) This solid mass was surrounded by normal ovarian parenchymal tissue. (B) The gross appearance of the

left ovary was normal. (C) The enucleated mass was placed into an endopouch and morcellated using an electronic morcellator within the pouch. Fragments of the

morcellated mass were collected in the pouch while avoiding dissemination in the pelvic cavity. (D) Postoperative findings of ovarian sparing local mass excision of the right

ovary.

Y.J. Cho et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 185 (2015) 78–82 81

This study has certain limitations. First, the data were collectedretrospectively from medical records. Therefore, we evaluated onlythe patients who were followed-up at least 6 months after thesurgery. Moreover, we did not contact all the patients who

Fig. 2. Operative findings of the left ovarian exophytic fibroma. (A) An exophytic solid mas

tissue by a pedicle. (C) After coagulation of the pedicle, the mass was easily resected. Th

Postoperative findings of ovarian sparing local mass excision of the left ovary.

underwent a local mass excision. Therefore, the pregnancyoutcomes and recurrence rate may be underestimated. Neverthe-less, we believe that the benefits of ovarian function preservationtake priority over the possibility of recurrence and reoperation.

s was noted on the left ovary. (B) This solid mass was connected with normal ovarian

e mass was then morcellated using an electronic morcellator within the pouch. (D)

Table 3Pregnancy outcomes subsequent ovarian sparing tumorectomy.

Case

No.

Age at

surgery

G/P at

surgery

Interval of

pregnancy

and surgery

(Mo)

Mode of

conception

Pregnancy

outcome

1 36 2/0 40 Spontaneous Full term C/S

delivery

d/t myomectomy

2 37 0/0 28 Spontaneous Full term NSVD

3 34 0/0 15 Spontaneous Full term NSVD

4 31 0/0 10 Spontaneous Full term NSVD

5 28 2/0 13 Spontaneous IUP 5wks, S/A

25 Spontaneous Full term C/S

delivery d/t

induction

failure

6 27 0/0 63 Spontaneous Ongoing pregnancy

7 24 1/0 32 Spontaneous IUP 6 wks, A/A

8 41 1/0 13 Spontaneous IUP 6 wks, missed

abortion!D/E

18 Spontaneous IUP 5 wks, missed

abortion!D/E

9 29 0/0 18 Spontaneous Full term C/S

delivery d/t

myomectomy

10 31 2/1 14 Spontaneous Full term C/S

delivery d/t

previous C/S

11 30 0/0 5 Thawing-ET IUP 8 wks,

missed

abortion!D/E

12 26 0/0 6 Spontaneous Ongoing pregnancy

No., number; G/P, gravidity/parity; Mo, months; C/S, cesarean section; d/t, due to;

NSVD, normal spontaneous vaginal delivery; S/A, spontaneous abortion; A/A,

artificial abortion; D/E, dilatation and evacuation; ET, embryo transfer.

Y.J. Cho et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 185 (2015) 78–8282

In conclusion, ovarian-sparing local mass excision, particularlyusing a laparoscopic approach, is an effective option in women ofreproductive age for preoperatively or intraoperatively suspectedovarian fibroma/fibrothecoma. Given the low recurrence rate ofovarian fibroma/fibrothecoma following ovarian sparing localmass excision, minimally invasive and fertility-sparing surgeryshould be selected, especially in younger patients for whomfertility and ovarian function can be preserved.

Conflict of interest statement

The authors report no conflict of interest.

Financial disclosure

None.

Condensation

Ovarian-sparing local mass excision appears to be an effectivesurgical option for ovarian fibroma/fibrothecoma in women ofreproductive age, considering the low rate of recurrence.

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