Ruptured Pedunculated Adnomyoma with Cystic Degeneration in a Postmenopausal Woman: A Case Report and Literature Review

We report a case of a 50-year-old woman, who is Para 1. Her past medical history is unremarkable, except for two previous myomectomies. The patient is menopause for 1 year and is not on hormone replacement therapy. The patient presented complaining of right lower abdominal pain of a few weeks duration. Her physical examination was unremarkable. Ultrasound examination revealed two uterine fibroids of 4 cm in diameter, and a left ovarian cystic mass of 11 cm in diameter [ Figure 1 ], which was suspected by a computed tomography scan to be left ovarian endometrioma. Ultrasound image showing a left side mass 11.3 cm × 7.7 cm Tumor markers were within normal ranges except for a slightly elevated CA-125; 54.2 U/mL. The decision was made to do a total laparoscopic hysterectomy with bilateral salpingo-oophorectomy (TLH + BSO). Intraoperative findings showed diffuse endometriotic lesions on the abdominal wall, colon, bladder, uterus, cul-de-sac, ovaries, tubes, peritoneum, and omentum, in addition to an enlarged uterus, with three pedunculated masses arising from the right side of the uterine body and fundus, appearing like fibroids [ Figure 2 ]. The uterus, three masses, and two small subserosal fibroids, with increased vasculature Surprisingly, the mass that was thought to be originating from the left side by imaging studies, was discovered to be originating from the right side of the uterus. Its appearance is suggestive of a collapsed cystic lesion, which raised the possibility of previous rupture, according to the presence of diffuse spread of endometriotic lesions all over the abdominal and pelvic cavity. TLH + BSO was performed in a classical way using three-dimensional technology, and the specimen was extracted through the vagina. The final pathology report showed a uterus with adenomyoma, fibroids, and endometrial polyp, while the three pedunculated masses arising from the right side of the uterus were found to be adenomyoma with endometriotic cystic degeneration.

Endometriosis is a heterogeneous, complex, benign gynecological disease affecting 10% of women in reproductive age. It is defined as the presence of endometrium-like glands and stroma out of the uterus, primarily on the pelvic peritoneum and ovaries.[ 1 ] Adenomyosis refers to a disorder, in which endometrial glands and stroma are present within the myometrium.[ 2 ] Adenomyoma is a circumscribed nodular aggregate of benign endometrial glands, accompanied by adjacent myometrial hyperplasia, and infiltration by inflammatory cells.[ 3 4 ] A rare form of adenomyosis is cystic adenomyosis, according to the patient’s age, it is divided into juvenile and adult cystic adenomyosis.[ 5 ] It is characterized by more extensive hemorrhage within myometrial cysts. Adult type is more common in menopausal women, as junctional zone disruption is suggested for pathogenesis.[ 6 7 ] Endometriotic cystic degeneration is a very rare finding in leiomyoma, reported few times in literature,[ 8 9 ] while data about cystic degeneration of adenomyoma are scarce,[ 10 ] reported for the first time by Parulekar in 1990;[ 11 ] rupture of such cysts is reported only a handful of times in premenopausal patients.[ 12 13 ] Adenomyosis and adenomyoma are common estrogen-dependent gynecologic disorders,[ 14 ] hinting that they are predominantly a disease of reproductive age, the prevalence of which largely varies between 9% and 66%.[ 2 ] In postmenopausal women, adenomyosis and adenomyoma are rare in the absence of hormonal replacement therapy,[ 15 ] while endometriosis is found in about 2%–4% of patients.[ 16 ] The most common symptoms of adenomyosis are heavy menstrual bleeding and dysmenorrhea. In more than 30% of cases, dysmenorrhea is secondary and progressive.[ 4 ] Less commonly, chronic pelvic pain and dyspareunia, with some sources reporting an association of 11%–12% with infertility.[ 6 17 ] Around 4.5% of patients are asymptomatic.[ 2 ] Here, we present a postmenopausal woman with a ruptured adenomyoma with a review of the literature.

Cystic adenomyosis or maybe cystic degeneration of adenomyoma is a form of adenomyosis which may present as a pedunculated mass lesion, mimicking ovarian endometrioma clinically and on imaging studies. It is a very rare presentation, especially a ruptured adenomyotic cyst in a menopausal woman. The incidence of rupture adenomyotic cyst remains unknown but is possible. Diagnosis is challenging, and management requires knowledge and confident surgical skills. This study was conducted in accordance with the ethical principles outlined in the Declaration of Helsinki and its amendments. The authors certify that they have obtained all appropriate patient consent form. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. All authors were involved in the drafting, editing and approval of the manuscript for publication. In addition to this, each author contributed the following: Mohammad Zeiter: collected data, reviewed articles, wrote and edited manuscript. Chuan-Chi Kao: reviewed articles, edited manuscript. Kuan-Gen Huang: reviewed and edited manuscript. Chyi-Long Lee: reviewed and edited manuscript. Luay Abu Atileh: reviewed and edited manuscript. All authors have read and agreed to the final version of the manuscript. The data underlying this article will be shared on reasonable request to the corresponding author. Prof. Chyi-Long Lee and Dr. Kuan-Gen Huang an editorial board member at Gynecology and Minimally Invasive Therapy , had no role in the peer review process of or decision to publish this article. The other authors declared no conflicts of interest in writing this paper.

The mechanism and pathophysiology of endometriosis and adenomyosis is not fully understood.[ 2 18 ] Endometriosis and adenomyosis are hormonal dependent. They are mainly diseases of reproductive age[ 1 18 ] with their presence in a menopausal woman being rare[ 15 16 ] in the absence of exogenous hormones. Higher incidence of adenomyosis has been reported in women treated with tamoxifen for breast cancer.[ 19 ] The fact that adenomyosis is mainly found in old multiparous women, increases the possibility of uterine muscle “trauma” from expansion and placentation to be involved in the pathogenesis. Disruption of the junctional zone allows for endometrial tissue to “sneaks” into the myometrium. That also explains the increased risk in patients with a history of uterine surgeries such as dilatation and curettage, cesarean section, and myomectomy. Four distinct types of adenomyosis are identified; diffuse sclerotic, nodular, and cystic, occurring in 54.5%, 13%, 28%, and 4.5% of cases, respectively,[ 20 ] it can coexist with leiomyoma in 50%, endometriosis in 11%,[ 17 ] and endometrial polyps in 7% of cases.[ 6 ] Cystic adenomyosis is a rare form, that is often misdiagnosed as uterine malformation and is usually found within the uterine body, causing a deformity in its shape.[ 10 ] Pedunculated subserosal adenomyotic cysts are very rare, and being associated with extensive endometriotic lesions in a postmenopausal woman, as in our case, is extremely rare. The rupture of a large adenomyotic cyst is reported to be possible,[ 7 ] with a few cases reported in premenopausal patients.[ 12 13 ] The diagnosis is made by ultrasound and magnetic resonance imaging (MRI) studies. Cystic adenomyosis may be misdiagnosed as adnexal mass, which makes the diagnosis challenging and may lead to unnecessary investigations and procedures. Ultrasound findings are endometrial infiltration, smooth muscle proliferation, and increased vascularity.[ 6 ] Characteristic MRI findings for adenomyosis are uterine enlargement, which is characterized by ill-defined regions within the junctional zone, as junctional zone thicker than 12 mm is diagnostic; in addition to hyperintense myometrial cysts reflecting regions of ectopic endometrial tissue,[ 6 ] while in cystic adenomyosis lesions show high signal intensity on the T1-weighted image and significantly low signal intensity on the T2-weighted image.[ 5 ] Bhusal et al . reported the diagnosis of cystic endometriosis within a degenerated myoma poses challenges, as it may mimic other cystic pelvic masses on imaging studies. Therefore, careful interpretation of imaging findings and consideration of clinical history are essential for accurate diagnosis and appropriate management. Treatment strategies for such cases may require a multidisciplinary approach, addressing both the endometriosis and myoma-related subfertility. Their case underscores the importance of recognizing rare presentations of cystic degeneration within myomas and the potential coexistence of other pathologies, such as endometriosis.[ 8 ] No laboratory tests for the diagnosis of adenomyosis.[ 21 ] Nevertheless, CA-125 can be used as preoperative diagnostic indicator and one of the dynamic monitoring indicators for postoperative follow-up.[ 5 ] Pistofidis et al . highlighted the histopathology as adenomyosis with cystic changes is characterized by the presence of cystic spaces lined by endometrial-like epithelium and surrounded by myometrial tissue containing ectopic endometrial glands and stroma. This distinct histological appearance distinguishes it from other subtypes of adenomyosis.[ 20 ] Treatment is challenging and the desire for future fertility should be considered when treating adenomyosis, as it can be treated medically (nonsteroidal anti-inflammatory drug, oral contraceptive pills, Mirena, etc.), with MRI/ultrasound-guided high-intensity ultrasound thermal ablation in focal disease, uterine artery embolization or surgical excision when future fertility is desired. Hysterectomy is the definitive cure.[ 6 ] Medical treatment is appropriate for patients with mild symptoms and small lesions and is similar to adenomyosis, however, surgery is considered the most effective method of treatment.[ 5 ] Li et al . reported a case of laparoscopic treatment of a large cystic adenomyosis of the uterus. In the case described, laparoscopic excision of a large cystic adenomyosis resulted in the resolution of symptoms and preservation of fertility, highlighting the effectiveness of this approach in achieving successful outcomes. The ability to perform precise dissection and excision of cystic lesions while preserving the integrity of the uterus is particularly advantageous for patients desiring future childbearing. Furthermore, laparoscopic surgery for cystic adenomyosis requires meticulous preoperative evaluation, including comprehensive imaging studies to delineate the extent of the disease and assess for any associated pathology. Intraoperative techniques such as cystic wall identification and careful excision of cystic contents are crucial to ensure complete removal of the adenomyotic lesions. Overall, this case report underscores the role of laparoscopic surgery as a safe and effective treatment option for large cystic adenomyosis of the uterus.[ 7 ] The fact that our patient had no history of chronic symptoms, and her presentation was relatively acute, with the presence of extensive endometriotic lesions, a “collapsed” looking adenomyotic cyst; and the normal-looking ovaries without any features of endometriosis, with the absence of extensive adhesions that would be expected in such an extent of the disease, makes the possibility of a ruptured adenomyotic cyst to be a justifiable cause for spread of the lesions. Hatirnaz et al . highlight the importance of considering rare manifestations of endometriosis, such as cystic endometriosis within degenerated leiomyomas, in the differential diagnosis of pelvic masses in infertile women with diminished ovarian reserve.[ 9 ] Parulekar reported that conservative management may suffice for asymptomatic cases, while surgical intervention may be warranted for symptomatic or significantly enlarged cystic adenomyomas.[ 11 ] In Yasuda et al . report, this case highlights the importance of considering ruptured cystic adenomyosis in the differential diagnosis of acute abdomen in women of reproductive age, particularly those with a history of dysmenorrhea or infertility. Laparoscopic enucleation represents a minimally invasive and effective treatment option for managing this rare condition, emphasizing the importance of interdisciplinary collaboration and individualized patient care.[ 12 ] Dr. Keating et al . highlight the importance of considering ruptured adenomyotic cysts in the differential diagnosis of acute pelvic pain, particularly in women of reproductive age with a history of dysmenorrhea or infertility. Further research is needed to better understand the pathogenesis, optimal management strategies, and long-term outcomes of this rare condition.[ 13 ] To our knowledge, this is the first case reported in the literature with the combination of pedunculated adenomyotic multiple cysts in a menopausal woman with features suggestive of the ruptured adenomyotic cyst. We also would like to highlight the cystic degeneration of adenomyoma rather than the primary development of adenomyotic cyst.