BMC Women’s Health Volume 21, Article Number: 263 (2021) Cite this article

Endometriosis of the uterus can be manifested as diffuse solid lesions or cystic lesions. The former is common, the latter is rare, especially cystic adenomyosis larger than 5 cm.

A 30-year-old woman was admitted to the hospital with severe deterioration of dysmenorrhea. Ultrasonography revealed a rare, well-defined cystic lesion, approximately 5.5 × 4 × 5.0 cm. The level of CA-125 increased slightly. She underwent laparoscopic surgery to remove the uterine muscle tissue. The histopathology of the specimen showed that there were endometrial glands in the wall of the cyst, and a large area of ​​hemorrhage was visible on the inner wall of the cyst. The patient recovered well after the operation, and the symptoms were completely relieved.

This is a rare case of cystic adenomyosis treated by laparoscopic surgery.

Adenomyosis is the presence of endometrial glands and stroma in the background of the myometrium, accompanied by adjacent smooth muscle hyperplasia. It may be diffuse or cystic. Diffuse adenomyosis is more common [1], cystic adenomyosis is a rare disease, more common in young patients [2]. Large adenomyosis cysts are lined with functional endometrioid tissue in place, which is characterized by periodic changes, accompanied by epithelial shedding and hemorrhagic infarction of adjacent smooth muscles [1]. Patients with adenomyosis may have important clinical manifestations such as pelvic pain and severe dysmenorrhea, and may not require any gynecological surgery.

The diagnosis and treatment of these cases have brought huge difficulties, which will be difficult to overcome until well-designed studies are initiated to guide management [3].

Here, we report our experience in the treatment of cystic adenomyosis with laparoscopic surgery in a 30-year-old woman.

A 30-year-old woman was admitted to the hospital with severe dysmenorrhea for about 2 years. She is not pregnant and has not received any surgical treatment. Upon admission, a pelvic examination showed normal appendages and an enlarged uterus. Ultrasound examination revealed a 5.5 × 4 × 5.0 cm cystic lesion with a clear border on the left anterior wall, which was separated from the normal uterine cavity (Figure 1a, b). CA-125 levels increased slightly (76.2 U/mL).

Three-dimensional ultrasound image. a The uterus shows a normal shape of the uterine cavity. b The left anterior wall 4.5 × 4 × 5.0 cm clearly demarcated cystic lesion, well separated from the normal uterine cavity

Minimally invasive surgery is a preferred way to treat these diseases, so laparoscopic surgery is considered the first choice for this situation. During laparoscopy, uterine lesions were found in the left part of the fundus of the uterus near the round ligament (Figure 2a). The ovaries and fallopian tubes look normal. When we use the monopolar hook to open the cyst cavity, we can see the chocolate-like fluid flowing out of the cyst (Figure 2b) and a cyst with brown tissue, which has no borders like normal fibroids (Figure 2c). The uterine musculature was removed from the surrounding myometrium. The operation did not penetrate the uterine cavity. The surgical wound was sutured with two layers of continuous sutures (Figure 2d). The histopathology of the specimen revealed that the wall of the cyst was lined with endometrial glands (Figure 3a), and macrophages were seen on the inner wall of the cyst to engulf hemosiderin (Figure 3b), which was diagnosed as cystic adenomyosis [1, 2]. The patient recovered smoothly after the operation. The patient received a single dose of 3.75 mg of gonadotropin-releasing hormone (GnRH) analog subcutaneous injection for 3 cycles, which is beneficial to improve the surgical effect and relieve postoperative dysmenorrhea symptoms [1]. Her symptoms disappeared completely during the outpatient follow-up for 4 months, the ultrasound examination was normal (Figure 4), and the CA125 dropped to normal (21.0 U/mL).

Under laparoscopy. a Uterine lesions were found on the left part of the fundus of the uterus near the round ligament and on the uterus. b The cysts flow out chocolate-like liquid, there is brown tissue in the cyst cavity, and there is no boundary with normal fibroids

Histological manifestations of adenomyosis cysts. a Endometrial glands lining the cyst wall (H&E × 40). b Large area of ​​hemorrhage on the inner wall of the cyst (H&E × 40)

Ultrasonography 4 months after surgery

Cystic lesions in the uterus are not common, and cystic adenomyosis is rare [4]. Uterine cysts are divided into two categories: congenital and acquired. Acquired cysts include cystic degeneration of uterine leiomyomas, cystic adenomyosis, and serous cysts. Ultrasound is the first choice for diagnosing adenomyosis, but MRI is more helpful for diagnosis. Elevated serum CA-125 levels have been proposed as a diagnostic tool for cystic adenomyosis. These patients usually have elevated serum CA-125 levels. In this case, an extreme increase in serum CA-125 level was observed before surgery and decreased after tumor resection, consistent with previous reports.

Acién et al. [2] The diagnostic criteria for cystic adenomyosis include (1) isolated accessory mass, (2) normal uterus (endometrial cavity), normal fallopian tubes and ovaries, (3) pathological examination of surgically removed mass, (4) vice The cavity is lined with glands and interstitial endometrial epithelium, (5) chocolate brown liquid, (6) no adenomyosis (if the uterus has been removed), although there may be small in the adjacent myometrium Uterine adenomyosis lesions to the accessory cavity. In our case, the patient met all the above criteria because the histopathology of the specimen showed that the cyst wall was lined with endometrial glands, and macrophages could be seen on the inner wall of the cyst to engulf hemosiderin, confirming the diagnosis of cystic adenomyosis .

Because many patients with cystic adenomyosis are young, minimally invasive surgery, such as laparoscopic resection, is considered desirable. Laparoscopic resection can significantly improve related dysmenorrhea and increase the possibility of successful pregnancy [4]. Hormone therapy with GnRH agonists or oral contraceptives is a treatment option for cystic adenomyosis, which has a certain effect, but symptoms may reappear after drug treatment is stopped. The patient was given a single dose of 3.75 mg of gonadotropin releasing hormone (GnRH) analog subcutaneous injection for 3 cycles, which is beneficial to improve the surgical effect and relieve the symptoms of dysmenorrhea.

Cystic adenomyosis is rare. It can be asymptomatic or manifest as progressive dysmenorrhea. Ultrasonography and MRI are complementary diagnostic tools. CA125 can be used as a preoperative diagnostic index and postoperative follow-up. Surgery is the preferred method of treatment. GnRHa can be used as an adjuvant treatment.

All data generated or analyzed during this study is included in this published article.

Cucinella G, Billone V, Pitruzzella I, etc. A 25-year-old woman with adenomyosis cyst: a case report. J minimally invasive gynecology. 2013;20(6):894-8.

Acién P, Acién M, Fernández F, etc. Hollow accessory uterine mass: Müllerian duct abnormalities in women with normal uterus. Obstetrics and Gynecology. 2010;116(5):1101-9.

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Takeuchi H, Kitade M, Kikuchi I, etc. Diagnosis, laparoscopic treatment, and histopathological findings of juvenile cystic adenomyoma: a review of nine cases. Fertilize. 2010;94(3):862-8.

This research was funded by the Chongqing Natural Science Foundation, and the award number is cstc2019jcyj-msxm0877.

Department of Gynecology and Pelvic Floor Oncology, Chongqing Maternal and Child Health Hospital, 120 Longshan Road, Yubei District, Chongqing

Zhao Chengzhi, Lu Shentao, Li Lei

Department of Pathology, Chongqing Maternal and Child Health Hospital, 120 Longshan Road, Yubei District, Chongqing

Department of Ultrasound, Chongqing Maternal and Child Health Hospital, 120 Longshan Road, Yubei District, Chongqing

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CZ and LL handled the case. BW and CZ conducted pathological diagnosis and review of cases and wrote manuscripts. BW, CY, and ST participated in the diagnosis, obtained informed consent, and decided on the treatment of the case. The final manuscript read and approved by all authors.

The approval of the Human Institutional Review Board is not required because the study is a summary of diagnosis and treatment information, which is considered to be the routine management of our hospital. Written informed consent has been obtained and obtained from the patient to publish the case.

The patient’s written consent has been obtained to publish the case.

The authors declare that they have no competing interests.

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Zhao, CZ., Wang, B., Zhong, Cy. Wait. Laparoscopic surgery for the treatment of uterine cystic adenomyosis: a case report. BMC Women's Health 21, 263 (2021). https://doi.org/10.1186/s12905-021-01341-1

DOI: https://doi.org/10.1186/s12905-021-01341-1

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