A rare case of chronic complete uterine inversion in a postmenopausal woman: a case report and literature review

Abstract

Uterine inversion is a rare condition that refers to the collapse of the fundus into the uterine cavity and occurs in puerperal and non-puerperal conditions. Non-puerperal uterine inversion is particularly infrequent. Diagnosing non-puerperal uterine inversion is often challenging because it resembles vaginal or cervical tumors and pelvic organ prolapse. Furthermore, this condition alters the anatomical structure of pelvic organs, thereby complicating diagnosis and treatment, and potentially leading to misdiagnosis with grave consequences. We report a case of a postmenopausal woman who presented with irregular vaginal bleeding and dysuria for 2 months. Preoperative enhanced pelvic magnetic resonance imaging suggested a benign tumor combined with uterine inversion, which was subsequently confirmed during surgery. A laparoscopic attempt to reposition the uterus failed, leading to successful repositioning via an abdominal incision and subsequent total abdominal hysterectomy with bilateral salpingo-oophorectomy. A histopathological examination showed a submucosal leiomyoma, which was smaller than that typically reported in other cases. We also conducted a review of previous cases to offer empirical guidance for the diagnosis and treatment of this rare condition.

Introduction

Uterine inversion (UI) is a rare phenomenon characterized by the inward sinking of the uterine fundus towards the uterine cavity or its protrusion from the cervix. UI typically presents as a severe complication during the third stage of labor, with an incidence rate of approximately 1 in 15,000. ¹ Notably, non-puerperal uterine inversion (NPUI), although extremely rare, is predominantly associated with uterine tumors. ² Interestingly, NPUI has also been reported in women with no sexual experience. ³ The diagnosis and management of NPUI poses challenges. We report a case of chronic complete UI in a postmenopausal woman, and provide a detailed account of the diagnostic process, surgical management, and subsequent prognosis of this condition.

Case presentation

Our patient was a 55-year-old woman with a history of two pregnancies and one live birth. Her medical history included type 2 diabetes and chronic bronchitis. She presented to our hospital with a 2-month history of irregular vaginal bleeding and dysuria. A physical examination showed a stable general condition with steady vital signs. Complete uterine prolapse was observed, along with the presence of a small mass at the uterine fundus. A routine blood test showed a white blood cell count of 19.1 × 10⁹/L, a neutrophil percentage of 90.9%, a hemoglobin concentration of 77 g/L, and a platelet count of 441 × 10⁹/L. Tumor markers, such as cancer antigen 125 (CA125), CA199, carcinoembryonic antigen, CA724, and alpha fetoprotein, were within the normal range. Ultrasound initially suggested uterine prolapse. Enhanced pelvic magnetic resonance imaging (MRI) showed uterine prolapse into the vagina, sinking of the uterine fundus into the uterine cavity, and protrusion through the cervix to reach the vaginal orifice. A low-signal shadow measuring approximately 4 × 3 × 3 cm was observed beyond the vaginal orifice (Figure 1(a)). Axial imaging showed a characteristic “bull’s-eye” configuration (Figure 1(b)). Contrast medium injection showed considerable enhancement of the uterine body, while minimal enhancement was observed in a low-signal shadow at the uterine fundus (Figure 1(c)). Furthermore, the post-void residual urine volume measured approximately 941 mL. This finding in conjunction with the patient’s clinical manifestations and examination findings led to a diagnosis of UI accompanied by infection and urinary retention.

Figure 1.

Magnetic resonance imaging and intraoperative observations of the patient. (a) A sagittal T2-weighted magnetic resonance image of the pelvis shows a U-shaped uterus (red arrow) and a low-signal shadow of the fundus (red star) of the uterus, measuring approximately 4 × 3 × 3 cm and protruding beyond the vaginal orifice. (b) A T2-weighted axial image of the pelvis shows a bulls-eye appearance (red arrow). (c) The uterus on a contrast-enhanced T1-weighted magnetic resonance image shows heterogeneous enhancement (red arrow). A myoma attached to the uterus can also be seen (red star). (d) A laparoscopic image shows dilatation of the original cervical dome with an inwardly plunging uterine body. Only the fallopian tubes and ovaries are visible. (e) Under anesthesia, the entire uterus and a mass at the base of the uterus prolapsed from the vaginal orifice, and two tubal openings appeared at the base of the uterus and (f) while the assistant continuously pushed the tumor upward in the vagina, the surgeon clamped and pulled the cervix, opened the vesical peritoneal reflection and pushed the bladder downward. The surgeon then incised the lower portion of the anterior wall of the uterus and used the index finger to apply force through the incision to reposition the uterine body.

The primary cause of the inversion may have been closely related to a necrotic and infected mass at the vaginal introitus. We decided to perform a pathological examination of the mass at the vaginal orifice because of the patient’s postmenopausal status and the nature of the mass. This examination was followed by a total abdominal hysterectomy with bilateral salpingo-oophorectomy. The patient consented to the above-mentioned procedures and underwent surgery after infection control.

Subsequent laparoscopic exploration showed dilation of the initial cervical dome with an inwardly plunging uterine body, in which only the fallopian tubes and ovaries were discernible (Figure 1(d)), thereby confirming the diagnosis of UI. During anesthesia, complete prolapse of the entire uterus and a mass at its base were found to protrude from the vaginal orifice, accompanied by the apparent presence of two tubal openings at the base of the uterus (Figure 1(e)). After an unsuccessful attempt at transvaginal manual repositioning of the uterus under laparoscopic surveillance, conversion to open surgery was performed. Following the incision of the anterior uterine wall, successful repositioning of the uterus was achieved (Figure 1(f)), followed by a hysterosalpingo-oophorectomy. The surgical procedure proceeded uneventfully.

A histopathological examination showed a submucosal leiomyoma of the fundus with erosions (approximately 3.0 cm in diameter) (Figure 2). Inflammatory infiltration was observed in the endometrium, cervix, and bilateral tubal mucosa, but no abnormalities were detected in bilateral ovarian tissue.

Figure 2.

Histology of the patient. Paraffin section pathology shows that the mass in the uterine fundus is a submucosal fundal leiomyoma with erosions.

After receiving postoperative symptomatic treatment, which included anti-infection therapy, bladder function exercises, and management of urinary retention, the patient recovered well and was satisfied with the treatment outcomes. During a 3-year follow-up, no complications were observed. The reporting of this case report conformed to the CARE guidelines. ⁴

This study received approval for publication from the Institutional Review Board of the Affiliated People’s Hospital of Ningbo University (approval number: 2024-N-006). The patient provided signed informed consent for publication.

Discussion

The prevalence of NPUI is exceedingly low. Existing literature on NPUI primarily consists of case reports; therefore its actual prevalence is unknown. In addition, the pathogenesis of NPUI remains inconclusive, with current understanding suggesting a multifactorial etiology. In a comprehensive literature review of articles published from 1940 to 2018, Herath et al. ² found that NPUI was primarily associated with tumors, and only a small proportion (9.2%) of cases were idiopathic. Most masses were benign lesions, predominantly leiomyomas (56.2%), while the minority consisted of malignancies (32.02%), such as carcinosarcoma and endometrial or cervical carcinoma.

Twenty-two articles published within the last 10 years were included in the analysis, with a focus on cases of NPUI associated with leiomyomas (Table 1).^3,5–25 The leiomyoma in our patient was smaller than those in previously reported. This finding suggests that even small fibroids may contribute to NPUI, although other factors, such as cervical insufficiency and increased abdominal pressure, may also play a role. Further studies are required to confirm these findings because of the rarity of this condition, although large-scale research may be difficult because of the limited number of cases.

Table 1.

Case review of non-puerperal uterine inversion associated with leiomyomas.

Reference	Age (years)	Parity	Mass size (cm)	Type of surgery
Our case	55	G2P1	4 × 3 × 3	Abdominal hysterectomy with bilateral salpingo-oophorectomy
Kesrouani A, et al. 5	47	G4P4	11 × 7.9 × 6.6	Total abdominal hysterectomy
Song YJ, et al. 6	42	0	22 × 11.5 × 5.8	Emergent explorative laparotomy
Moshayedi F, et al. 7	30	0	6–7	Myomectomy
Zhang X, et al. 8	34	0	6 × 5 × 5	Transvaginal myomectomy
Benjelloun AT, et al. 9	70	5	7	Vaginal approach with laparotomy
Dadgar S, et al. 10	51	3	5.5 × 6.2	Abdominal hysterectomy
Teimoori B, et al. 11	32	0	7.7 × 6.5	Haultain’s procedure
Lima T, et al. 12	53	G4P2	6.1	Laparotomy
Guerrouj I, et al. 13	43	G1P1	10.5 × 7.8	Vaginal hysterectomy
Datta MR, et al. 14	42	4	7.5 × 6 × 5.4	Abdominal hysterectomy
Al Qahtani NH 15	31	0	6 × 7 × 12	Vaginal myomectomy
Della Corte L, et al. 3	52	0	4	Combined abdominovaginal approach
Chandrasiri D, et al. 16	48	2	30 × 9 × 1	Urgent vaginal myomectomy and abdominal hysterectomy
Sakinci M, et al. 17	39	G2P1	4 × 2, 3 × 2, 2 × 2	Laparotomy
Uomoto M, et al. 18	47	0	10	Total hysterectomy
Kurniawati E, et al. 19	65	3	20 × 15	Vaginal hysterectomy
Kouame A, et al. 20	28	G2P1	15	Abdominal hysterectomy
De A, et al. 21	38	G4P4	7 × 6	Abdominal hysterectomy
Al-Shukri M, et al. 22	36	0	10 × 11	Vaginal myomectomy
Aniebue U, et al. 23	45	Grand multiparous	12 × 10	Vaginal myomectomy
Modares M, et al. 24	48	G2P2	7 × 7	Abdominal hysterectomy
Deka G, et al. 25	48	G5P4	10 × 15	Vaginal myomectomy

UI is categorized into three stages. ³ Stage 1 refers to the inverted uterus remaining within the uterine cavity. Stage 2 is a complete inversion of the fundus through the cervix (as observed in this case). Stage 3 refers to a complete inversion of the uterus and vagina. The symptoms of NPUI are nonspecific and may include irregular vaginal bleeding,^5,8 a protruding intravaginal mass,^6,12 pelvic pain,^10,20 increased vaginal discharge, ²³ and urinary obstruction.^19,21 Preoperative diagnosis of NPUI is particularly challenging, with imaging having a crucial role. MRI is the preferred diagnostic modality because of its examiner-independent advantages. ²⁶ Transvaginal ultrasound has limited diagnostic value, particularly when there are cases involving a sizeable vaginal mass that impedes probe insertion. In this case, an ultrasound scan suggested a uterine prolapse. Typical MRI findings of NPUI include a sagittal image displaying an incomplete Y-shaped uterine cavity or a complete U-shaped configuration (as observed in this case), along with a thickened and inverted uterine fundus, and a distinctive bull’s-eye pattern on axial imaging. Furthermore, enhanced MRI can aid in diagnosing malignant tumors through contrast visualization and in facilitating lymph node identification. Although computed tomography scanning is not commonly used for diagnosing NPUI, it may be considered if MRI is unavailable. Additionally, vigilance towards potential malignancies should be maintained, necessitating measuring tumor markers. A preoperative biopsy or intraoperative frozen sections can also be used when malignancy is highly suspected. However, a final diagnosis should rely on routine paraffin pathology.

Surgery is the primary treatment approach for NPUI. The surgical approach is tailored according to the patient’s age, fertility considerations, and preoperative diagnosis. A review of the recent literature (Table 1) showed that uterine repositioning may be attempted in young women with fertility preservation needs, provided malignancy is excluded, although its success rate is low. This repositioning may also lead to complications, such as uterine perforation and severe hemorrhage. ¹⁸ In most patients with NPUI, particularly those without reproductive concerns, total hysterectomy remains the preferred surgical approach. These findings are in agreement with the conclusions of a review published in 2018. ²⁷ Nevertheless, there are alterations in the anatomical position of the ureter, urethra, and bladder resulting from UI, which subsequently increase surgical complexity. Therefore, initially performing uterine repositioning to restore a normal anatomical structure before proceeding with uterine resection is usually performed. This sequential approach aims to minimize the risk of intraoperative damage posed to surrounding organs. Nonetheless, caution should be exercised when the complete exclusion of malignancy cannot be guaranteed to avoid potential dissemination of tumor cells caused by the incision during repositioning of the uterus. Various methods are used for uterine repositioning, such as non-invasive techniques performed under anesthesia and transabdominal surgery. Transabdominal surgery includes Huntington’s technique involving the use of Alice’s forceps to extract the inverted portion of the uterus or Haultain’s approach, which entails vertically incising the cerclage at the posterior aspect of the cervix followed by gradual traction on the uterine fundus. Another method is transvaginal surgery involving anterior wall opening using Spinelli’s method or posterior wall opening using Kushner’s technique. Additionally, laparoscopic and robotic surgeries have emerged as novel alternatives. ²⁸ A successful case of laparoscopic hysterectomy without uterine repositioning for the treatment of nonpuerperal UI was reported in 2021. ²⁹ In cases involving concomitant malignancies, more comprehensive radical surgery is necessary. Perioperative management is also indispensable in correcting anemia, anti-infection measures, improving urinary retention issues, and promoting bladder function recovery.

Conclusion

We present a rare case of chronic complete UI in a postmenopausal woman, potentially associated with a small submucosal leiomyoma. Awareness of NPUI can assist with the identification of people with this rare disease. The appropriate use of imaging modalities, such as MRI, is crucial to differentiate NPUI from other conditions, such as uterine prolapse and cervical tumors, thereby reducing the risk of misdiagnosis. Additionally, vigilance against the presence of malignant tumors is essential, and a biopsy and pathological examination should be used when necessary for an accurate diagnosis. Surgical intervention remains the primary treatment approach for NPUI, requiring individualized plans based on factors, such as the patient’s age, preoperative diagnosis, and fertility considerations. Perioperative management should encompass various aspects, such as infection control, correction of anemia, and improvement of urinary retention.