doi:10.1111/jog.14430 J. Obstet. Gynaecol. Res.

2020

Accessory ovary may be a treatment for infertility: Case

report and review of current literatures

Xiao-Ye Zhou, Xue-Ling Tang, Fei-Lei Zhang and Xiao-Jun Yang

Department of Obstetrics and Gynecology, The First Affiliated Hospital of Soochow University, Suzhou, China

Abstract
Accessory ovary is a type of ovarian dysplasia, which is often defined as an ovarian tissue placed near and
directly connected to the normal ovary or one of ovarian ligaments. It is often asymptomatic, mostly is
found or diagnosed at laparotomy, laparoscopy or autopsy. Accessory ovary is often excised during surgery
due to its potential malignant behavior. We report a case of endometriosis cyst occurred simultaneously in
right side of orthotopic and accessory ovaries, together with torsion 180 of accessory ovarian cyst. Consider-
ing that the patient had not given birth and the large size of cysts, exploratory laparotomy was performed.
During laparotomy, both site of ovarian cyst have been removed with orthotopic and accessory ovarian tis-
sues preserved. After surgery, a large number of antral follicles were found both in right side of orthotopic
and accessory ovaries by ultrasonography. Accessory ovary is considered to have physiological function,
and can be preserved as a fertility protection measure for women who have fertility requirements. At pre-
sent, the definition of ectopic ovary, accessory ovary and supernumerary ovary are very vague and rarely
discussed separately. So, we proposed a new way to clarify the concepts of ectopic ovary, accessory ovary
and supernumerary ovary. Moreover, we advocated that they should be discussed separately in terms of
definition and management measures.
Key words: accessory ovary, endometriosis, fertility preservation, torsion.

Introduction ovary. In this way, supernumerary ovary and acces-

Ovarian dysplasia includes single or bilateral ovarian
insufficiency, ovarian hypoplasia, supernumerary
ovaries, accessory ovaries and etc., each one of them
is very rare. Accessory ovaries and supernumerary
ovaries are similar medical terms, and the most com-
monly recognized diagnostic criteria are proposed by
Wharton1 in 1959. The supernumerary ovary is
defined as completely independent of the normally
placed ovaries without any direct connections to any
of ovarian ligaments. The accessory ovary is located
near and connected to the normally placed ovaries,
and this extra ovary may split off from the normal
ovary during development and gain blood supply
through tissue connection with the normally placed
sory ovary can be distinguished mainly according to
their positional relationship with the orthotopic
ovary. However, Lachman and Berman2 insisted that
the definition of supernumerary and accessory ovary
is not accurate and advocated that the word ‘ectopic
ovary’ should be used instead of ‘supernumerary or
accessory ovary’. Therefore, currently, most of the
supernumerary, accessory or ectopic ovary is dis-
cussed together.
Case Report
A 27-year-old unmarried woman with a history of
endometriosis (EMs), gravida 1, para 0, had a history
of spontaneous abortion, was admitted to the hospital

Received: April 5 2020.

Accepted: July 25 2020.
Correspondence: Professor Xiao-Jun Yang, Department of Obstetrics and Gynecology, The First Affiliated Hospital of Soochow
University, 188 Shizi Road, Suzhou 215006, Jiangsu Province, China. Email: yang.xiaojun@hotmail.com

© 2020 Japan Society of Obstetrics and Gynecology 1

X.-Y. Zhou et al.

because of abdominal pain for a week. The patient
has a history of recurrent asymptomatic right acces-
sory cyst (unknown size) 2 years ago, without any
follow-up conducted. In 05 November 2018, under no
obvious inducement, she complained with persistent
right lower abdominal pain, accompanied by nausea
and vomiting. She had a history of laparoscopic sur-
gery for bilateral ovarian EMs cyst 7 years ago, no
obvious accessory ovary was recorded for the right
side of ovary during the surgery.
Laboratory tests showed elevation levels of cancer
antigen 125 (CA125): 282.00 U/mL, carbohydrate anti-
gen 19-9 (CA19-9): 464.47 U/mL, both values were
higher than normal. Other laboratory results including
female sex hormone and ovarian reserve function (anti-
Müllerian hormone [AMH]) were all within normal
ranges. Ultrasound examination demonstrated normal
size and shape of uterus and the left ovary (Fig. 1a). In
addition, in front of the left side of the uterus and in the
right adnexal area, two cyst with rough and thick wall
and undetected blood flow appeared in the pelvic cav-
ity, which is full of strong echo of sticky and floccus
fluid, about 109 × 90 × 143 mm in size for the left and
128 × 111 × 146 mm in size for the right, respectively
(Fig. 1b). Computed tomography (CT) identified a mul-
tiple cystic solid mass in pelvic cavity which considered
originating from the right adnexa (Fig. 1c). During lapa-
rotomy, two smooth, liquid-filled cystic masses were
found from the right ovarian tissue. Further inspection
showed that one cyst at about 12 cm in diameter origi-
nated from the right orthotopic ovary, while the other
cyst, at about 14 cm, originated from the right accessory
ovary. Furthermore, the accessory ovarian cyst was
found to connect to tissue of the right orthotopic ovary,
while it was free from the bondage of surrounding ana-
tomical structures, such as the fallopian tube, ovary
arteries and veins and the utero-ovarian ligament.
Disseminated EMs lesion depositions were found at
the surface of pelvic peritoneum. Nevertheless, it is
worth mentioning that the cyst from the right acces-
sory ovary was rotated 180 . Chocolate-colored fluid
was aspirated from both cysts before ovarian
cystectomy. The final pathologic diagnosis was con-
sistent with intraoperative findings: cystic EMs of

Figure 1 Imaging examination before surgery. (a) A normal ovary of 30 × 19 × 23 mm size can be detected clearly on the
left. (b) The B-ultrasound demonstrated that there is a rough-walled, thick anechoic area in front of the left side of the
uterus, which is filled with floccus echo and no color blood flow signal is detected (the one with the thin arrow). A non-
echo area is also visible in the right adnexal area, with rough and thick wall and slightly enhanced rear echo. A partly
flocculent echo is seen in the inner part, and also no color blood flow signal is detected (the one with the thick arrow),
suggesting ovarian endometriosis cyst. (c) Computed tomography identified multiple cystic or solid mass in pelvic cav-
ity, which was considered to be from the right adnexa.

2 © 2020 Japan Society of Obstetrics and Gynecology

 A case of accessory ovarian EMs cyst

Figure 2 Postoperative pathological results. (a) Endometrial glands and stromal tissue can be observed in the cyst wall of
the ovary, which are consistent with the characteristics of endometriosis cyst. (b) Hemorrhage and necrosis can be
observed in the cyst wall of the accessory ovary, in accordance with torsion of the accessory ovarian cyst observed dur-
ing surgery.

Figure 3 The B-ultrasound results after the surgery. (a) B-ultrasound result 1 month after the surgery: Two hypo-echoic
findings on the right side adnexal area: The size of the right orthotopic ovary was about 25 × 18 × 23 mm (the thick
arrow), and the size of the accessory ovary next to the right orthotopic ovary was about 23 × 19 × 22 mm (the thin
arrow). (b) B-ultrasound result 1 year after the surgery: A large number of antral follicles were found both in right side
of orthotopic and accessory ovaries by ultrasonography: 18 antral follicles in the orthotopic ovary (the thin arrow),
13 antral follicles in the accessory ovary (the thick arrow).

right orthotopic (Fig. 2a) and accessory ovary
(Fig. 2b). In addition, focal hemorrhage and infarction
was observed in cyst wall of the accessory ovary
(Fig. 2b). One month after the surgery, ultrasonogra-
phy showed two low-echo areas of similar size were
found in the right adnexal area (Fig 3a). One year
after the surgery, a large number of antral follicles
were found both in right side of orthotopic and acces-
sory ovaries (Fig 3b).
Discussion
In our case, EMs cysts occurred simultaneously in the
orthotopic and accessory ovary, and especially the
accessory ovary cyst was accompanied by torsion,
which was very rare. Preoperative examination dem-
onstrated normal range of female sex hormone levels
and AMH, the patient complained no clinical symp-
toms except for 1-week pain of the lower right abdo-
men. During laparotomy, the accessory ovary was
found directly connected to tissue of the right
orthotopic ovary, without any associations to anatom-
ical structures of the ovary. Two EMs cysts of similar
size were presented simultaneously in the orthotopic
and accessory ovary respectively, accompanied by
torsion of the accessory ovary cyst. This case is con-
sidered to fulfill the diagnostic criteria of the acces-
sory ovary defined by Wharton.1 The occurrence and
progressive increased volume of EMs cyst and ultra-
sonography showed a large number of antral follicles
in the accessory ovary after surgery demonstrated

© 2020 Japan Society of Obstetrics and Gynecology 3

X.-Y. Zhou et al.
that the accessory ovary can have similar physiologi-
cal functions and pathophysiological changes as
orthotopic ovary.
Based on the previous laparoscopic surgical record-
ing, in which no anatomical abnormalities were found
in the right adnexal area, it was speculated that the
occurrence of accessory ovary in this case was an
acquired source. Secondary to pelvic surgery or
inflammation seems the most likely explanation,
rather than embryonic origin. In this case, torsion of
the accessory ovarian cyst may be due to the greater
mobility of the accessory ovary, which was found to
have little anatomical restraint from the fallopian
tube, ovarian suspending ligament, and the utero-
ovarian ligament.
In the cases that have been reported, on one hand,
the ectopic ovary can be found in a variety of sites,
mainly in the pelvic area.3,4 On the other hand, there
are various theories regarding the origin of ectopic
ovary. Printz et al.5 believed that there are two possi-
ble sources of ectopic ovaries in terms of embryology:
ovarian tissue transplantation or improper migration
of germinal cells. While, Lachman and Berman2
explain the origin of ectopic ovary from another per-
spective, they found that most of the ectopic ovary
were secondary to pelvic surgery, suggested that the
ectopic ovary could be divided into three types: post-
operative transplantation of ectopic ovarian tissues,
inflammatory ectopic ovary and really ectopic ovary
caused by abnormal ovarian decline. Meanwhile, Lim
et al.6 also believed that ectopic ovary have embryo-
logical or acquired origin. The acquired source of
ectopic ovary may be the implantation of ovarian tis-
sue during surgery or the postinflammatory implanta-
tion. Ectopic ovary may result from the accidental
interruption of normal ovary and subsequent tissue
implantation in other pelvic sites during the opera-
tion, or result from adherence to the surrounding tis-
sues due to chronic inflammation after surgery.
Women with ectopic ovary are prone to have concom-
itant congenital urological and gynecological mal-
formations, which are believed to be related to its
embryological origin.
Most ectopic ovary are usually considered non-
functional and asymptomatic,7 while Fei et al.8believe
that ectopic ovarian tissue can have normal endocrine
function and pathological changes, such as various
benign or malignant ovarian cysts or tumors. Tumor
torsion can also occur in the ectopic ovary, just as
they do in the orthotopic ovary. And histologic exam-
ination of the accessory ovary showed that the
4 © 2020 Japan Society of Obstetrics and Gynecology
accessory ovary was well-functioning with normal
follicles.9
Among the benign and malignant tumors that have
been reported in the ectopic ovaries, about 20% of
cases are associated with EMs. Early in 1951, EMs
was found in the ectopic ovary of a rhesus monkey10.
A total of 12 cases of ectopic ovary associated with
EMs have been reported so far (including present
case)11–15 and among them, eight cases of EMs origi-
nated from ectopic ovaries (66.67%), eight cases had a
previous history of gynecologic surgery (66.67%),
11 cases suffered with pelvic pain (91.76%), seven
cases with previous diagnosis of EMs (58.33%), six
cases with infertility (50.00%) and none of them had
any congenital malformations (Table 1).
From these reported cases of EMs in the ectopic
ovary, it can be confirmed that EMs may occur in the
ectopic ovary alone, which is a common benign ovar-
ian cyst frequently located in orthotopic ovary or
implanted in pelvic peritoneum. Furthermore, in these
case series of ectopic ovary, the source of ectopic
ovary is more of an acquired source: eight cases had a
history of previous surgery (66.67%), and there was
no case with congenital gynecological and urological
malformations. On the other hand, two theories have
been proposed regarding the etiology of EMs in
ectopic ovary. First, the wide dissemination of EMs
lesions in the pelvic cavity can also implant or invade
into the excess ovary. Another possibility is lymphatic
spreading, which may explain the development of
distant EMs.16
Complications such as torsion of cyst can also occur
in the accessory ovaries, although it was rarely hap-
pened and only two cases have been reported so far.
Fei et al.8 reported a case of accessory ovary cyst com-
plicated with torsion, presented with acute lower
abdominal pain, nausea and vomiting, simulta-
neously with low fever and leukocytosis, these symp-
toms and signs were similar with cyst torsion in the
orthotopic ovary. In addition, Kamiyama et al.17
reported torsion of a supernumerary ovarian endome-
trial cyst in a 28-year-old pregnant woman (40 weeks
gestation). The cyst that attached to the pregnant
uterus was removed surgically, and postoperative
pathology confirmed hemorrhage and necrosis within
the cyst wall, which was pathologically consistent
with torsion of ovarian endometrial cyst.
We believe that Wharton’s definition is premised
on two normal ovaries and one abnormal ovary,
while Lachman’s classification is very vague. The eti-
ology of ectopic ovary is not always determined, nor
 Table 1 Location and disease characteristics in a series of 12 cases of endometriosis (EMs) in ectopic ovary
Case Authors Reported Patient Location of the Histology of the The history History With pelvic With Infertility With
year age ectopic ovary ectopic ovary of surgery of EMs pain pelvic congenital
EMs malformation
1 Wharton1 1959 37 Pelvic Ovary No No Yes Yes Yes No
2 Burnett7 1961 / Along left ureter, Ovary and EMs Yes Yes Yes Yes No No
retroperitoneal
3 Cruikshank 1982 33 The left pelvic Ovary Yes Yes Yes Yes No No
and Van wall
Drie11
4 Cruikshank 1982 36 The Ovary and No No Yes Yes No No
and Van retroperitoneal mucinous cyst
Drie11 adenoma

© 2020 Japan Society of Obstetrics and Gynecology

5 Lee and Gore12 1984 30 The left fimbria Ovary Yes Yes Yes Yes Yes No
and
rectosigmoid
6 Navarro et al.13 1990 38 Left paracolic Ovary and EMs Yes Yes Yes Yes Yes No
gutter
7 Badawy et al.16 1995 32 Left Ovary and an Yes Yes Yes No Yes No
retroperitoneal endometrioma
and
osseous
metaplasia
8 Kamiyama 2001 28 On the pregnant Ovary and cystic No No No No No No
et al.17 uterus endometrioma
9 Imir 2006 30 On the sigmoid Ovary Yes Yes Yes Yes Yes No
et al.14 colon
10 Ogishima 2017 40 Rectovaginal Ovary and EMs No No Yes No No No
et al.15 pouch with cystic
change
11 Lim 2018 42 on the anterior Ovary and cystic Yes No Yes No Yes No
et al.6 rectosigmoid endometrioma
colon
12 Present case 2018 27 Side of the right Ovary and cystic Yes Yes Yes Yes No No
ovary endometrioma
A case of accessory ovarian EMs cyst

5
X.-Y. Zhou et al.
is it caused by a single factor. We always cannot
determine which one is the real cause, or both. There-
fore, we suggest that ectopic, accessory and supernu-
merary ovary should be discussed separately, and
redefined according to the information of the number
and location of ovaries which can be accurate
determined.
Ectopic ovary is used to refer specifically to a nor-
mal number of ovaries with abnormal position. Ovary
is not found in the normal ovarian position, but found
in other abdominal cavity positions such as groin,
omentum and mesentery. Supernumerary ovary is
defined as the existence of two ovaries with normal
positions, and the additional isolated ovarian tissues
that are completely independent of the normally
placed ovaries without any direct connections to any
of ovarian ligaments are found. Then, according to
the previous surgical history and whether concomi-
tant congenital urological and gynecological mal-
formations, the supernumerary ovary can be further
divided into embryonic or acquired origin. Accessory
ovary is defined as the existence of two ovaries with
normal positions, additional ovarian tissues that
located near the normally placed ovaries and may be
connected to various ligament vascular structures of
normal ovaries are found. Similarly, the accessory
ovary can be further divided into embryonic or
acquired origin according to the previous surgical his-
tory and whether concomitant congenital urological
and gynecological malformations.
And management strategies of supernumerary and
accessory ovary should also be discussed separately
according to the situation of the patient,18 especially
the fertility status. When the patient has no reproduc-
tive desire, orthotopic ovary can meet the hormonal
requirement of a woman. Therefore, supernumerary
or accessory ovary should both be excised, due to
potential malignant transformation.19 However, when
the patient has fertility requirements, supernumerary
ovary and accessory ovary should be managed differ-
ently. For supernumerary ovary, it does not have any
normal affiliated fallopian tube or direct anatomical
connections to the orthotopic ovary and uterus, its
oocytes cannot be collected into the fallopian tube to
fulfill the natural fertilization process, even if it has
the normal function of hormone secretion and ovula-
tion. And in artificial assisted reproductive technol-
ogy, due to the variability and uncertainty of its
location, it is also very difficult to get exact location of
the supernumerary ovary and perform oocytes
retrieval after the surgery. Therefore, the presence of
6 © 2020 Japan Society of Obstetrics and Gynecology
supernumerary ovary does not significantly increase
the chance of conception and it is not recommended
to preserve it during the surgery. On the other hand,
for the accessory ovary, it is connected directly with
orthotopic ovary or linked by certain ovarian liga-
ments. It has relatively normal anatomical structure
and fixed location that could increase the chance of
natural conception or it could be used for oocytes
retrieval in artificial assisted reproductive technology,
so the accessory ovaries should be preserved for treat-
ment of infertility in case of unilateral or bilateral
ovarian hypo-function. Meanwhile, intra-operative
biopsy and pathology is required to confirm the
benign characteristic of accessory ovaries before the
decision of preservation of accessory ovary. During
the surgery, the accessory ovary should be anchored
to reduce the occurrence of complications such as tor-
sion, and some metal clips can be placed near the
accessory ovary to mark its position for postoperative
imaging follow-up and surgical intervention. After
surgery, close follow-up is essential and patients are
advised to have their accessory ovaries removed
promptly after completion of the childbirth.
Diseases such as cysts or tumors and torsion of cyst
can be found in the accessory ovary separately or
simultaneously, the clinical symptoms, imaging mani-
festations, intraoperative founding and pathological
diagnosis of the disease in accessory ovary was simi-
lar with those in orthotopic ovary. Although rarely
happened, clinicians should take into account in dif-
ferential diagnosis and manage it according to the fer-
tility status of patients. The role of accessory ovary in
female reproduction should be fully considered and
tissue preservation is worth to be done in patients
with fertility requirement.
Finally, many unresolved questions remain to be fur-
ther investigated regarding the cysts/tumors that
occur in the accessory ovary alone. According to the
definition1, the accessory ovary is located close to the
orthotopic ovary, and shares certain blood vessel or
ligaments. From the structural aspect, the accessory
ovary is under the similar micro-environments with
the orthotopic ovary. However, the accessory ovary
and orthotopic ovary appear to be incongruous in bio-
logical behavior and functional characteristics. Such as,
the accessory ovary can developed various cysts/
tumors with the co-existence of a normal orthotopic
ovary. So far, very little is known regarding the under-
lying mechanisms. However, it was speculated that,
the accessory ovary might experience certain unknown
inner changes during its development, lead to further

alterations in its structure and biological behavior, and
then subsequently increase the probability of tumori-
genesis. In addition, those factors that facilitate the
development of accessory ovary might also be poten-
tial etiologies that induce the inner changes and further
lead to lesions in accessory ovary.
Ethics Approval and Consent to
Participate
This article does not contain any studies with human
participants or animals performed by any of the
authors and all procedures performed were in accor-
dance with the ethical standards of the institutional
and/or national research committee and with the 1964
Helsinki declaration. This study was approved by the
ethics committee of our institution and the patient pro-
vided informed consent. Written informed consent was
also obtained from the patient for publication of this
case report and all patient-related information and
images. A copy of the written consent is available for
review by the Editor-in-Chief of the journal.
Disclosure
None declared.
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