23 December 2024: Articles
Multifaceted Cervical Endometriosis: A Post-Surgical Diagnosis in a Complex Gynecological Case
Challenging differential diagnosis, Rare disease
Qingyuan Wang ABCDEF 1, Wenjun Shan BCDEF 1, Qiyin Zhu CDE 1, Wenyan Wang ABCDEF 1*DOI: 10.12659/AJCR.945246
Am J Case Rep 2024; 25:e945246
Abstract
BACKGROUND: Endometriosis is a condition where uterine lining tissue grows outside the uterine cavity, commonly on the ovaries and pelvic peritoneum, but can also occur in rare locations such as the cervix, lungs or pleura. Cervical endometriosis is typically diagnosed retrospectively through post-surgical pathology as it was in this case. This article presents a case of cervical endometriosis with recurrent vaginal bleeding, reviews recent literature to aid in clinical diagnosis and treatment.
CASE REPORT: A 42-year-old woman presented with a 5-year history of recurrent vaginal bloody discharge. Clinical evaluations revealed a cyst on the posterior wall of the cervix, suspected to be an endometriotic cyst, along with a uterine diverticulum and an endometrial polyp. The patient underwent surgical excision of the cyst, repair of the uterine diverticulum, and removal of the endometrial polyp. Postoperative pathology confirmed the diagnosis of an endometriotic cyst of the cervix. The patient was subsequently treated with oral Dienogest. An 8-month follow-up showed no complaints of discomfort, with gynecological examinations indicating good recovery and no abnormalities detected on ultrasound.
CONCLUSIONS: Cervical endometriotic cysts are rare but can lead to serious complications if not properly diagnosed and managed. Clinicians should remain vigilant for abnormal symptoms such as recurrent vaginal bleeding and pelvic pain. Timely and appropriate treatment measures, including surgical intervention and hormonal therapy, should be implemented to reduce the risk of complications and monitor for potential malignant transformation. Regular follow-ups are crucial to ensure early detection of any recurrence or progression.
Keywords: Endometriosis, Gynecology, Uterine Cervical Diseases, Humans, Female, adult, Uterine Hemorrhage
Introduction
Endometriosis is a condition in which endometrial tissue grows outside the uterine cavity; it is a common clinical disease with a prevalence of approximately 5+−10% among women of reproductive age [1]. Its clinical symptoms mainly include pain and infertility, with a risk of further malignant transformation [2]. Endometriosis commonly affects the ovaries and pelvic peritoneum but can also occur in the bladder, rectum, surgical scars, and, very rarely, in the lungs or pleura. Cervical endometriosis is also rare, and due to the lack of typical clinical symptoms, its diagnosis is often made retrospectively through postoperative pathology. A study from 1987 estimated its incidence at about 0.11–2.40% among people undergoing colposcopy [3], and a retrospective study from China reported a rate of 0.24% [4], highlighting its rarity. Symptoms of cervical endometriosis can include abnormal vaginal bleeding, with some reported cases leading to significant hemorrhage due to cyst rupture. This article shares a case of cervical endometriosis with recurrent vaginal bleeding, reviews the related literature from the past 5 years, and provides a summary analysis aimed at aiding the clinical diagnosis and treatment of cervical endometriosis.
Case Report
PATIENT INFORMATION:
A 42-year-old woman presented with “recurrent vaginal bloody discharge for 5 years”. She first experienced abnormal vaginal bloody discharge 5 years ago, occurring before and after menstruation, without seeking clinical treatment. Repeated gynecological ultrasound suggested: 1) uterine fibroids; 2) a cystic mass on the posterior wall of the cervix. In the past 3 months, the patient experienced increased vaginal bloody discharge accompanied by lower abdominal pain. A re-examination at our hospital suggested: 1) uterine fibroids; 2) uterine diverticulum formation; 3) intrauterine device; 4) a cystic mass on the posterior wall of the cervix (34×32×21 mm) with dense punctate echoes inside.
Menstrual and reproductive history: The patient had regular menstruation, moderate flow, and no symptoms of dysmenorrhea. She had had 3 pregnancies and 1 childbirth, and underwent a lower-segment cesarean section 19 years ago. She underwent 2 induced abortions. She has no current desire for more children. She had an IUD in place for birth control.
Surgical history: Four years ago, she underwent laparoscopic removal of a mature cystic teratoma of the left ovary and myomectomy for “left ovarian mature cystic teratoma and uterine fibroids.” She had no history of hypertension, diabetes, or other underlying diseases.
CLINICAL PRESENTATION:
The patient’s main clinical symptom was recurrent vaginal bloody discharge before and after menstruation and throughout the middle of the menstrual cycle. Gynecological examination showed the cyst could be reached behind the cervix, and the area was 3 cm.Biochemical examination: Routine blood, liver and kidney function, coagulation function, and electrolytes were normal. Serum β-HCG testing was negative. Serum carbohydrate antigen 125 was 61.00 U/ml (reference range: 0–35 U/ml).Ultrasound examination: Vide supra.Pelvic MRI examination (Figure 1) suggested (after removal of the intrauterine device): The posterior wall of the cervical part showed a round-like short T1, mixed T2 signal, with clear boundaries, 27.5×27.6mm in size, with uneven signals inside, and another nodule-like short T1, short T2 signal on its left edge, with a diameter of 10.7 mm.Electrocardiogram and chest CT scan were normal.Cervical HPV testing was normal, and liquid-based cytology examination showed no epithelial cell abnormalities or malignant cells.
DIAGNOSIS AND TREATMENT:
Based on the medical history and auxiliary examinations, the clinical diagnosis was: 1) cyst on the posterior wall of the cervix (possible endometriotic cyst), 2) uterine diverticulum, 3) endometrial polyp. After completing preoperative preparations and obtaining informed consent, the patient underwent surgical treatment, specifically: excision of the cyst on the posterior wall of the cervix + uterine diverticulum repair surgery + endometrial polyp excision surgery.
The surgical process involved separating the uterocervical vaginal gap, where a cystic protrusion with a diameter of 3 cm was visible on the posterior wall of the cervix. Opening the cervical stroma released a large amount of coffee-colored viscous fluid, followed by stripping the cyst wall and sending it for pathological examination.
Postoperative pathology (Figure 2) showed a cervical endometriotic cyst. The patient was treated postoperatively with oral Dienogest.
FOLLOW-UP:
The patient has been closely followed for 8 months, with no discomfort. Gynecological examination showed good recovery of the cervical wound, and gynecological ultrasound examination found no abnormalities.
Discussion
Cervical endometriosis is a relatively rare disease, with most patients with cervical endometriosis showing no significant clinical symptoms and unable to obtain a definitive diagnosis clinically, often discovered postoperatively through pathology. Among these, the formation of a cervical endometriotic cyst is even rarer. In our case, the patient developed a 3-cm cervical endometriotic cyst over several years without any serious complications such as rupture or bleeding. The cervical cyst was successfully removed. Although we suspected the possibility of a cervical endometriotic cyst prior to surgery, the final diagnosis was dependent on the postoperative pathology examination. Therefore, its pathophysiological mechanisms and clinical manifestations remain a focus of current research. In our study, we reviewed related case reports from the past 5 years (Table 1), which were very few but had already revealed the severe complications that cervical endometriotic cysts could cause, including massive bleeding and potential malignant transformation. These cases emphasize the importance of timely, accurate diagnosis, and effective treatment strategies.
In this case, at first, we considered the diagnosis of cervical endometriosis due to the elevation of the patient’s CA-125 and the ultrasonography indicating the cyst with dense punctate echoes inside. We are thankful that the results of the cervical HPV test and the liquid-based cytology examination were clear, allowing us to provisionally exclude malignancy at the initial stage of diagnosis. If the results of these 2 tests are problematic, we prefer the diagnosis of malignancy before surgery. Therefore, we still think that it is difficult to determine the diagnosis of cervical endometriosis before surgery.
In the confirmed case reports from the past 5 years, 1 patient sought treatment for recurrent chronic pelvic pain, and after multiple surgeries, was finally diagnosed, with the final pathology report indicating adenomyosis and a cervical endometriotic cyst. Previous studies have shown that patients with adenomyosis often have coexisting pelvic endometriosis, so we consider the occurrence of cervical endometriosis in this patient may be due to the progression of adenomyosis [6]. Another case report described 2 patients who experienced symptoms of massive bleeding due to rupture of a cervical endometriotic cyst. One patient underwent uterine artery embolization, conservative treatment after blood transfusion, and took oral Dienogest for 9 months, without recurrence [7]. Our reported patient also received oral Dienogest treatment, with no recurrence, which can provide a reference for future treatment of patients with cervical endometriotic cysts. Another patient underwent emergency abdominal surgery and hysterectomy after blood transfusion, with postoperative pathology confirming a cervical endometriotic cyst [7]. The present case proves the importance of early identification and management of potentially dangerous symptoms in the treatment strategy of cervical endometriosis. This not only requires a high degree of clinical vigilance from doctors but also the use of modern imaging technology and histopathological examination to make timely and correct diagnoses and treatment decisions. Baoxuan Li and others reported a patient with endometrial-like cancer invading the cervix, with clinical symptoms of abdominal pain, diarrhea, and intermittent fever, who was postoperatively confirmed to have a family history of Lynch syndrome [5]. This case reminds us that for patients considered for diagnosis of cervical endometriotic cysts, further detailed evaluation combining biochemical tests, imaging examinations, and necessary pathological examinations is still needed to rule out malignant lesions, especially in patients with a family history of Lynch syndrome.
Additionally, we found a case report of primary cervical lymphoma in a 47-year-old female patient with symptoms of abdominal pain and renal failure [8]. Imaging suggested soft-tissue compression of the ureter in the uterine region. Eventually, she underwent surgery for suspected deep endometriosis, with the final pathology result being primary cervical lymphoma. This reminds us that in the diagnostic process for patients with cervical endometriotic cysts, differentiation from primary cervical lymphoma is necessary to avoid misdiagnosis.
Conclusions
In summary, although cases of cervical endometriosis cyst are relatively rare, the potential for severe complications and treatment challenges require clinicians to remain highly vigilant in their daily work. For patients presenting with abnormal symptoms, especially those with a family history of Lynch syndrome, thorough examinations and detailed family history investigations should be actively conducted to provide appropriate treatment measures, reduce the risk of severe complications, and effectively monitor for potential malignant transformation.
References:
1.. Taylor HS, Kotlyar AM, Flores VA, Endometriosis is a chronic systemic disease: Clinical challenges and novel innovations: Lancet, 2021; 397(10276); 839-52
2.. Vercellini P, Viganò P, Somigliana E, Fedele L, Endometriosis: pathogenesis and treatment: Nat Rev Endocrinol, 2014; 10(5); 261-75
3.. Veiga-Ferreira MM, Leiman G, Dunbar F, Margolius KA, Cervical endometriosis: Facilitated diagnosis by fine needle aspiration cytologic testing: Am J Obstet Gynecol, 1987; 157(4 Pt 1); 849-56
4.. Wang S, Li XC, Lang JH, Cervical endometriosis: Clinical character and management experience in a 27-year span: Am J Obstet Gynecol, 2011; 205(5); 452.e1-5
5.. Li B, Wang Y, Wang Y, Deep infiltrating endometriosis malignant invasion of cervical wall and rectal wall with lynch syndrome: A rare case report and review of literature: Front Oncol, 2022; 12; 832228
6.. Atalay Mert S, Dilbaz B, Ercan EE, Sahin Duran F, A rare case of the cervical endometriosis and overview of the literature: Reprod Sci, 2023; 30(9); 2882-86
7.. Matoba Y, Tsuji K, Morisada T, Massive hemorrhage due to cervical endometriotic cyst rupture: Two case reports and a literature review: Am J Case Rep, 2021; 22; e934120
8.. Ha K, Sheets K, Kedar R, Primary cervical lymphoma mimicking deep endometriosis: J Minim Invasive Gynecol, 2024; 31(5); 357-59
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