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Abstract
Uterine incarceration (UI) is a rare complication in non-pregnant women. A woman in her 50s presented to the emergency department with acute urinary retention and paresis of the right inferior limb. A neurological exam suggested a decrease in the right lower limb strength. On gynaecological exam, the cervix was displaced anteriorly and the cul de sac was obliterated with a pelvic mass. CA 19.9 and CA 125 levels were increased. The MRI of the pelvis confirmed an elongated and anteriorly displaced cervix compressing the urethra and the vesical neck due to a uterine mass. A diagnosis of UI was made and an abdominal hysterectomy with adhesiolysis was suggested as the treatment option. Six months after surgery the patient had no urinary complaints and the neurological exam was normal. UI should be considered in women with urinary and neurological symptoms. A delay in diagnosis may lead to significant morbidity.
- Surgery
- Obstetrics and gynaecology
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Background
Uterine incarceration (UI) is a rare complication that occurs when the uterus becomes trapped between the sacral hollow and the pubic symphysis.1 2 Patients present with non-specific symptoms, mainly with lower abdominal pain, constipation, urinary frequency, retention or incontinence.1 3 UI is mostly recognised as an obstetric complication, described in 1:3000 pregnancies.4 However, there are only a few case reports in the literature regarding UI in non-pregnant women and the incidence of UI in this population is unknown.2 We thereby present a rare acute presentation of an incarcerated uterus in a non-pregnant woman and discuss management options.
Case presentation
A woman in her 50s presented to the emergency department with acute urinary retention and paresis of the right inferior limb. In the last 3 days, she referred to an increased abdominal perimeter, anorexia and nausea. She had a previous episode of urinary retention, for 12 hours, 2 weeks earlier, solved spontaneously with micturition in dorsal decubitus followed by polyuria and pollakiuria on subsequent days. Since then, she mentioned voiding symptoms and that she had not been able to empty her bladder completely in a sitting position, but only in dorsal decubitus, requiring the use of diapers. At the emergency department, she was in anuria for 3 hours. A foley catheter was placed, and 1.5 litres of urine was drained, with no macroscopic findings. Her medical and surgical history was notable for regular menses and no previous deliveries. On neurological examination, a decrease in the right lower limb strength was observed. The patient stayed in the neurological department for a complementary investigation and a gynaecological examination was requested. The cervix could not be visualised during speculum examination. At bimanual evaluation, the cervix was displaced anteriorly and the cul de sac was obliterated with a painful not mobile pelvic mass compatible with a uterine myoma.
Investigations
Her blood tests showed a BHCG negative, haemoglobin of 10.3 g/dL, increased C -reactive protein (13.3 mg/dL), cancer antigen (CA 19.9 level of 94.5 U/mL (normal range <37 U/mL) and CA 125 level of 817 U/mL (normal range <35 U/mL). Renal function, ionogram, urinalysis and microbiologic urine test were normal. A lumbar puncture was performed with cerebrospinal fluid without evidence of bacterial or mycological infection, glucose 67 mg/dL; proteins 30.1 mg/dL. Brain and spine MRI were normal. Complementary electromyography suggested a right sub-acute radicular lesion of L5. The thoraco-abdominal-pelvic CT documented an increased uterus and a right uretero-hydronephrosis with 21 mm (figure 1). No gastrointestinal lesions were identified. A pelvic MRI was performed to characterise this gynaecological condition and confirmed UI caused by significant hypertrophy of the posterior wall of the fundus and body, suggestive of adenomyosis causing a right anterolateral displacement of the bladder (figure 2) and an elongated anteriorly displaced cervix compressing the urethra and the vesical neck. The ovaries were described as normal. The diagnosis of UI was hypothesised.
Abdominal-pelvic CT showing right uretero-hydronephrosis caused by the pelvic mass.
Pelvic MRI (T2 weighted)-right anterolateral displacement of the bladder by the uterine posterior wall mass suggestive of adenomyosis.
Differential diagnosis
In this case, the differential diagnosis of UI that presents with acute urinary retention and right limb paresis includes neurological causes such as stroke multiple sclerosis, spinal injuries and nervous system infections; pharmacological causes, postoperative aetiology, anatomic distortion, including pelvic masses and pelvic organ prolapse. Pharmacological and postoperative causes were excluded immediately as the patient’s antecedents were unremarkable. The absence of pathological findings in the lumbar puncture and brain and spine imaging helped to exclude neurological aetiology. The presence of a non-mobile pelvic mass in the cul de sac on gynaecological exam supported by an increased uterus on pelvic imaging favoured a compressive aetiology.
Treatment
The possible methods of resolution of this condition were discussed with the patient-manual reduction or laparotomy and operative reduction. After being well informed, she signed consent for surgery with abdominal hysterectomy since she was concerned about the recurrence of symptoms, and she was not planning a future pregnancy. During surgery, a Pfannenstiel incision was performed, and we found an enlarged retroverted uterus displaced to the right side of the pelvis, incarcerated in the cul de sac with multiple adhesions of the uterine body and left adnexa to the bowel. The cervix and the vesical neck were deviated in an anterior and cephalic position. The ovaries were macroscopically normal. Before the hysterectomy, the surgical team performed adhesiolysis followed by manual anterior and superior traction of the uterine fundus to free the uterus from the cul de sac.
Outcome and follow-up
The surgical procedure and the postoperative period were uneventful. The bladder catheter was removed on day 2 after surgery, and the patient was able to empty her bladder completely and spontaneously with minimal postvoid residual urine. She was discharged on the 4th day. Six months after surgery the patient had no urinary complaints nor deficits at the neurological exam. Her surgical pathology was suggestive of a 10 cm adenomyoma of the posterior wall of the uterus.
Discussion
According to some studies, predisposing factors to UI are uterine retroflexion, pelvic adhesions, uterine malformations, endometriosis, adenomyosis and fibroids, especially in the posterior location.1 5–7 UI symptoms are non-specific and usually related to mechanical compression of the surrounding organs, such as acute urinary retention (AUR).1 Obstructive urinary retention in women of reproductive age is a rare event reported in 7:100 000 individuals per year.8 The most common causes of obstructive retention are gynaecological surgery and pelvic masses. It can also occur in pregnant women with an incarcerated uterus (1:3000 pregnancies).9 In this clinical context, a detailed gynaecological history and pelvic examination should be obtained. A complementary pelvic imaging study should be performed and is helpful in patients with non-specific symptoms.3 8 According to some authors, MRI is the method of choice to diagnose UI.6 10 As described in our case, Fernandes et al mentioned UI characteristic findings: marked uterine retroversion and anterior/superior displacement of an elongated cervix.3 10
During the investigation of the pelvic mass, the neurological department found increased CA 125 and CA 19–9 levels. According to the literature, it was seen that the CA 125 levels not only increased in ovarian epithelial carcinoma but also under benign conditions such as myomas, endometriosis and adenomyosis. CA 19–9 is a highly sensitive tumour marker for pancreatic and bile duct cancers and can also be elevated in gynaecological tumours such as endometrial carcinoma or ovarian cancer. Recently some studies showed that CA 19–9 can be increased in patients with endometriosis and adenomyosis but rarely exceeds 1000 U/mL.11 12 In our case, the CA 19–9 level was only slightly elevated at 94.5 U/mL (normal range <37 U/mL), the thoraco-abdominal-pelvic CT excluded suspicious gastrointestinal pathology and the ovaries were normal in MRI and intraoperatively. Therefore, we assumed that the large adenomyoma caused the elevation of the CA 19–9 and the CA 125 levels.
The mechanism of urinary retention is unknown but some theories were proposed like the presence of a posterior uterine mass. In our case, similar to other reports, the adenomyoma of the posterior uterine wall in a retroverted uterus leads to the enlargement of the uterus which impacted against the sacrum.1 7 13–16 Further growth of the lesion displaced the cervix to an anterior and cephalic position, under the symphysis pubis, and ultimately compressed the urethra and bladder neck resulting in AUR. Initially, our patient had voiding symptoms and was only able to empty her bladder in decubitus dorsalis as this position allowed the partial release of cervical compression of the urethra and bladder neck. The additional growth of the adenomyoma was responsible for the failure of this mechanism. The mechanical compression of the right L5 root by the uterus was responsible for the neurological motor deficit of the right lower limb. We found another case report of an AUR due to a uterine myoma associated with a chronic motor polyradiculopathy in an electromyogram.16
The first step to manage UI is bladder decompression with the insertion of a Foley catheter.17 Urinary retention should be seen as an emergency given the risk of bladder rupture or chronic neuromuscular dysfunction.18
Conservative measures to correct the retroverted uterus include positional manoeuvres knee–chest or all-fours position.1 In case of failure, manual reduction through vaginal examination should be attempted, with the patient awake or under general or regional anaesthesia. The operator should apply digital firm pressure, in a cephalad direction, in the posterior cul de sac. The patient could be in a dorsal lithotomy position, trendelenburg or knee-chest position.4 6 17 The use of a tenaculum in the posterior cervical lip for countertraction could be useful to increase uterine traction.6 Fernandes et al described a laparoscopy approach complementary to digital uterine traction. In this procedure, the round ligaments can be grasped for additional traction.3 19 Colonoscopic insufflation of the rectosigmoid was also reported as an alternative conservative measure.6 20 To prevent UI recurrence, some authors advise for pessary use for 1 week.3 19 21 Operative reduction should be considered when all interventions mentioned above have failed.6 In our case, the patient opted for surgical treatment.
Surgical treatment of the UI has been described in non-gravid patients and in most cases includes abdominal hysterectomy. The first step is the attempt to antevert the uterus before hysterotomy. As we did in our case, the manual anterior and superior traction of the uterus preceded by adhesiolysis can be a safe option. If it is not technically possible, other approaches should be considered such as traction sutures evolving the myometrium, the use of a Doyen tumour screw or myomectomy.2
Patient’s perspective
First, I was not able to empty my bladder in the toilet. After a few days, I was not able to empty my bladder in any position, I started to use diapers, I felt pelvic discomfort and I lost strength in my right leg. I was very scared, and I went to the emergency department. When the doctors found out that I had a uterine mass compressing my bladder I opted for a hysterectomy because I was afraid of recurrence, and I didn’t want a future pregnancy. I was able to urinate spontaneously 2 days after surgery and I gradually recovered my right leg strength. I had fantastic support from the medical team, and I am very grateful to them.
Learning points
Clinicians should be aware to include uterine incarceration (UI) in the differential diagnosis of acute urinary retention and neurological deficits in non-pregnant women, because a delay in diagnosis may lead to significant morbidity.
When UI is clinically suspected, gynaecological examination and imaging methods, such as gynaecological ultrasound or pelvic MRI, can confirm the diagnosis.
Management of UI in the non-gravid female has generally been approached surgically; however, conservative measures to correct the retroverted uterus can be attempted first.
Ethics statements
Patient consent for publication
References
Footnotes
Contributors LTC participated in the diagnosis and treatment of the patient and wrote the case report. CRdC and ALR also participated in the diagnosis and treatment and verified the case report manuscript. All authors discussed the manuscript and contributed to the final manuscript.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.