Liu et al.

BMC Women's Health (2025) 25:6 BMC Women's Health

https://doi.org/10.1186/s12905-024-03533-x

CASE REPORT Open Access

Ovarian adenomyoma: a case report

Weilong Liu1, Tongtong Yao2†, Haiyan Wang3, Wenjing Yu4, Hongtang Shi3, Jiwei Guo5 and Zhiqiang Liu3*

Abstract
Introduction Ovarian adenomyoma is a rare gynecological tumor with a high misdiagnosis rate, leading many
patients to undergo unnecessary surgeries that may affect fertility. Menstrual abdominal pain is the most common
symptom, and auxiliary examinations often cannot clarify its nature. It often relies on intraoperative diagnosis, and
surgical resection can achieve good therapeutic effects.
Case presentation A 50-year-old woman presented with lower abdominal pain during her menstrual period for
the past two months. She had a previous medical history of uterine adenomyomectomy, ovarian cystectomy, and a
cesarean section. Ultrasound revealed a 5.7 × 3.8 × 4.3 cm mass on the posterior wall of the uterus, a 9.9 × 5.6 × 8.2 cm
hypoechoic mass in the right posterior part of the uterus, and a 2.8 × 2.2 × 2.7 cm anechoic mass in the left ovary.
CA125 (Carbohydrate antigen 125) 191.80U/ml (0–30). MRI (magnetic resonance imaging) imaging confirmed a
7.9 × 6.2 × 7.2 cm fibroid on the right posterior wall of the uterus. Consider partial degeneration of multiple uterine
fibroids and benign cystic degeneration in the lower left abdomen. Surgical resection was performed smoothly, and
the diagnosis was confirmed by postoperative pathology.
Conclusion Ovarian adenomyoma is a rare benign gynecologic tumour with a high rate of misdiagnosis. When a
patient presents with recent lower abdominal pain or dysmenorrhea, a history of endometriosis or myomectomy,
and MRI findings showing irregular bleeding patterns in a pelvic mass, the possibility of extrauterine adenomyosis
should be considered. Minimally invasive treatment options, such as single-port laparoscopy or vaginal dissection,
may offer advantages, but caution should be exercised due to the potential for malignant tumors. Preserving fertility
is something worth exploring. We hope to provide warnings to more gynaecologists and reduce misdiagnosis and
unnecessary treatment.
Keywords Ovarian adenomyoma, Uterus-like mass, Extrauterine, Adenomyosis, Case report

Introduction
The prevalence of adenomyosis ranges from 5–70% [1].
†
Tongtong Yao: co-first author This condition can develop into localized nodules or
*Correspondence: masses, known as adenomyomas. The most common
Zhiqiang Liu locations for extrauterine adenomyomas are the para-
[email protected]
1
Binzhou Medical University Hospital, Binzhou, Shandong, China rectal space, ovaries, and broad ligament [2]. However,
2
Obstetrics and Gynecology, Liaocheng Women and Children Health, ovarian adenomyomas are extremely rare. As far as we
Liaocheng, Shandong, China know, only 11 cases of ovarian adenomyoma have been
3
Gynecology, Binzhou Medical University Hospital, Binzhou, Shandong,
China reported, with only one documented case in China [2].
4
Obstetrics and Gynecology, Gansu Provincial Central Hospital, Lanzhou, The first case was reported by McDougal in 1986 [3], and
Gansu, China all cases were diagnosed through postoperative pathol-
5
Medical Research Center, Binzhou Medical University Hospital, Binzhou,
Shandong, China ogy. Notably, five women of reproductive age underwent

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Liu et al. BMC Women's Health (2025) 25:6 Page 2 of 5

unnecessary hysterectomy due to the lack of a pre- with a diameter of about 10 cm was observed internally.
cise preoperative diagnosis. Based on existing reports, A grey-white oval hard mass with a diameter of 10 cm
abdominal and pelvic pain, along with menstrual abnor- was seen on the section, with a clear boundary between
malities, are the most common clinical manifestations the surrounding tissue. The right fallopian tube is elon-
of ovarian adenomyoma [4]. Additional symptoms, such gated, and the left ovary is enlarged, demonstrating a
as abnormal vaginal bleeding and infertility, have also cyst approximately 3.0 cm in diameter with a thin wall
been noted [2]. Given the varying sizes and locations of and clear fluid. The left fallopian tube is unremarkable. A
adenomyomas, patients may also experience symptoms grayish-red hard mass of approximately 1.0 cm in diam-
like rectal compression, leading to altered bowel habits eter was observed in the sacral ligament on the left side.
or stool characteristics [5]. The compression from large Postoperative pathology confirmed adenomyosis of the
pelvic and extrauterine adenomyomas can also affect uterus, an adenomyoma of the right ovary, a mass in the
urination [6]. The lack of specificity in these clinical fea- left uterosacral ligament, and endometriosis. The patient
tures often leads to unnecessary treatments; this article was followed up for one month after surgery without any
discusses the case of a woman who was admitted to the abnormalities (See Figs. 1, 2 and 3).
hospital for 2 months of abdominal pain due to menstru-
ation and was diagnosed with ovarian adenomyoma after Discussion
surgery. We also conducted a literature review discussing Among published cases of ovarian adenomyoma, the
the pathogenesis, MRI characteristics, minimally invasive majority of patients (81.8%, 9/11) were women of child-
treatment approaches, and fertility preservation related bearing age. Lower abdominal pain or menstrual lower
to ovarian adenomas, which holds significant implica- abdominal pain was reported in 72.7% (8/11) of cases,
tions for the future treatment of the disease. This work and excessive menstrual flow in 36.4% (4/11). Other
follows the SCARE 2023 guidelines to ensure quality symptoms included constipation, primary amenorrhea,
reporting [7]. palpable masses, dyspareunia, and infertility [2, 4, 8].
Additionally, 36.4% (4/11) had a history of endometriosis
Case presentation or uterine fibroids. Elevated levels of CA125 and CA19-9
A 50-year-old patient was hospitalized due to lower (Carbohydrate antigen 199) were observed in two cases
abdominal pain during menstruation for 2 months. Sev- (18.2%, 2/11), which returned to normal after surgery.
enteen years ago, the patient underwent a laparoscope Ultrasound examinations in ten patients revealed solid
uterine adenomyomectomy to address menorrhagia or complex cystic masses, and four patients underwent
and urinary frequency, during which a 3 cm right ovar- MRI, with one displaying multiple hyperintense sig-
ian follicular cyst was also removed. Postoperatively, her nals on T1-weighted images and hypointense signals on
menstruation was normal. She had a cesarean Sect. 8 T2-weighted images. At the same time, another showed
years ago and has no history of dysmenorrhea. On physi- low signals on both T1 and T2-weighted images, which
cal examination, the patient weighed 58 kg, with a BMI were not given due attention. Despite these findings,
(Body mass index) of 22.7. The uterus was enlarged unnecessary hysterectomies were performed in 45.5%
to the size of a uterus at 40 days of pregnancy. A hard, (5/11) of childbearing-age women. Only one case showed
solid mass (approximately 9 cm) was palpable behind the malignant transformation [9], while most patients recov-
uterus, with normal mobility and no tenderness. Ultraso- ered well postoperatively. Limited follow-up data suggest
nography revealed a 5.7 × 3.8 × 4.3-cm mixed hypoechoic no recurrence in two patients.
and anechoic mass on the posterior wall of the uterus, The pathogenesis of ovarian adenomyoma remains
with a 9.9 × 5.6 × 8.2-cm hypoechoic area in the right pos- unclear. Two main theories have been proposed: uter-
terior part of the uterus. The right ovary was not visible, ine or Müllerian fusion defects and subluminal stromal
and an anechoic area, measuring 2.8 × 2.2 × 2.7 cm with transformation [9]. The Müllerian fusion defect theory
good internal translucency, was detected in the left ovary. links the anomaly to developmental issues in the repro-
MRI revealed a space-occupying lesion in the right pos- ductive tract [10]. Among the 35 cases of ectopic ade-
terior part of the uterus, measuring 7.9 cm × 6.2 cm × nomyoma analyzed by Viola et al. [11], 22 cases were
7.2 cm, closely related to the adjacent right uterine wall. considered to have developed due to abnormalities in
CA125 191.80 U/mL. Preoperative diagnoses included reproductive tract formation. Alternatively, subluminal
multiple uterine fibroids and a left ovarian cyst. stroma, derived from the urogenital ridge, may undergo
Considering the patient’s lack of desire for fertility and metaplasia and differentiate into smooth muscle cells, as
the possibility of malignant tumours, a total hysterec- initially described by Cozzuto in 1981 [12]. This patient
tomy and right adnexectomy were performed. Intraop- has a history of uterine adenomyoma resection and endo-
eratively, the right ovary appeared enlarged and adherent metriosis, and the occurrence may be related to smooth
to the surrounding tissues, and a smooth oval tumour muscle metaplasia within the endometriotic lesions or
Liu et al. BMC Women's Health (2025) 25:6 Page 3 of 5

Fig. 1 A–D T1WI, the mass showed equal signal intensity, with multiple small round internal areas of high signal intensity (A); T2WI, the mass demon-
strated primarily a slightly high signal intensity, with multiple small internal round areas of high signal intensity and patchy low signal intensity (B); Lipid
suppression on T1WI showed a mixed signal and scattered small round high signal and patchy low signal areas (C); T1WI enhancement shows uneven
moderate enhancement (D)

Fig. 2 The pathological image is HE × 100, with red arrows indicating glands and blue arrows indicating fibrous tissue

cellular implantation following the resection of the uter- bleeding. Pathology reveals the presence of endometrial
ine adenomyoma. glands, specialized endometrial stroma, and well-formed
Some were initially considered malignant ovarian smooth muscle bundles [13]. The patient’s pathologi-
tumours before surgery [9]. Therefore, a preoperative cal images further confirm this, showing the presence of
MRI is necessary. Typical MRI findings for ovarian ade- glands and smooth muscle, with SMA (Smooth muscle
nomyosis include low signal intensity on T2-weighted actin) staining positive, which supports a smooth mus-
images and areas of high signal intensity on fat-sup- cle origin for the lesion and confirms the diagnosis of
pressed T1-weighted images, indicative of internal adenomyoma. Previously, the tumour was described as
Liu et al. BMC Women's Health (2025) 25:6 Page 4 of 5

Fig. 3 Postoperative ultrasound of the uterus and right ovary showed no abnormal echogenicity

Table 1 Key differential points

Disease Prevalence MRI findings Histopathological
classification characteristics
Endometriosis General female (2-10%), T1 high signal multiplicity, T2 screening, the T2 dark spot sign, and Adhe- Confirm ectopic en-
women with infertility sion with surrounding tissues [17–19] dometrial stroma and
(50%) [8] glands [20]
Ovarian 1% of all ovarian tumors T1 and T2-weighted MRI, which reveal low signal intensity, along with The primary components
leiomyoma [21] early uniform enhancement following the administration of a contrast include smooth muscle
agent [22] and spindle cells [22]
Ovarian thecoma 0.15 to 1% of all ovarian A sizable, well-defined solid mass with cystic regions that appears Composed of spindle
tumors [23] isointense or slightly hyperintense on the T2WI and SPAIR sequence in the cells with a moderate
pelvic cavity [24] amount of cytoplasm [24]

a well-defined oval nodular mass, often surrounded by a critical in managing ovarian adenomyoma. Techniques
thick muscle wall and a cavity containing chocolate-like such as single-port laparoscopy or transvaginal tumor
brown, bloody, serous, and mucinous fluids. Blood clots resection promote faster recovery with minimal scarring.
are commonly observed within the masses. CA125 is an It is well established that the administration of GnRH
important tumour marker for early screening of ovarian agonists (gonadotropin-releasing hormone agonists) can
cancer and can also show varying degrees of elevation in delay the progression of adenomyosis; therefore, explor-
benign gynaecological diseases [2]. Elevated CA125 lev- ing the potential application of GnRH agonists to delay
els have been reported in broad ligament and salpingeal disease progression in patients with ovarian adenomyosis
adenomyomas [14, 15], and elevated CA199 levels have who do not require surgery or to prevent recurrence in
been noted in ampullary adenomyomas [16]. Although women of reproductive age postoperatively, is warranted
the CA125 level in this patient was higher than the nor- [25].
mal level, it is not specific to ovarian adenomyosis. Dif- In summary, ovarian adenomyoma is a rare and chal-
ferentiation among endometriosis, ovarian fibromas, and lenging gynecological. Preoperative diagnosis is difficult
ovarian thecomas is essential (See Table 1). and often relies on postoperative pathology. Minimally
Due to the significant impact of unnecessary hyster- invasive surgical approaches and fertility preservation
ectomy on patients’ fertility, preserving reproductive strategies warrant further investigation. If there is recent
function and employing minimally invasive surgery are lower abdominal pain or dysmenorrhea, a history of
Liu et al. BMC Women's Health (2025) 25:6 Page 5 of 5

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