Un caz rar de torsiune uterină cu torsiune ovariană bilaterală: o provocare diagnostică

Introduction

Uterine torsion can be described as the rotation of the uterus greater than 45° around its main axis⁽¹⁾. Uterine torsion is an uncommon disorder that has been seen in women of all ages, from premenarchal to postmenopausal. Torsion of a non-gravid uterus is quite rare, and the majority of cases that are recorded involve pregnant women⁽²⁾. Uterine torsion is still a rare type of pelvic organ torsion in humans, as opposed to the more prevalent incidence in cattle and buffaloes⁽³⁾. It is dextrorotatory, or clockwise, in two-thirds of instances that are identified⁽⁴⁾. During the reproductive phase, uterine torsion has been found in a variety of age groups, but it has also been observed in young girls, initially misdiagnosed as an “unusual congenital anomaly”⁽⁵⁾.

Due to its low incidence, lack of distinct symptoms and limited laboratory findings, uterine torsion can be difficult to diagnose, potentially leading to fatal outcomes and infertility⁽⁶⁾. Although the precise cause of uterine torsion is still unknown, there are several important risk factors, including congenital developmental defects in other organs and increased adnexal mobility during pregnancy. Timely surgical intervention is required to prevent ischemia changes from progressing to gangrene.

We present a case of uterine torsion with bilateral ovarian torsion, associated with a large pedunculated subserosal fibroid, in a non-gravid patient who underwent a caesarean section 11 months before.

Case presentation

A 49-year-old, female, known to have a scarred uterus following a caesarean section 11 months before, with a pregnancy achieved through in vitro fertilization (IVF), using donated oocytes, presented to the emergency department with pelvic-abdominal pain that had started approximately one week before, worsening over the past two days, accompanied by vomiting and lower back pain. The day before, the patient sought care at another hospital where a contrast-enhanced abdominopelvic CT scan was conducted, which showed a cystic mass measuring approximately 21/15/19 cm, with thick walls, most likely of adnexal origin, an intensely heterogeneous uterus, fibromatous, with a fluid layer in the Douglas pouch and the right iliac fossa, a slightly dilated left ureter proximally (3-4 mm), partially opacified by contrast medium, with the lumbar ureters compressed. The patient had been known to have a large uterine fibroid for approximately 10 years, a condition that had been neglected over time.

On physical examination, the abdomen seemed enlarged up to the xiphoid process, much like in a full-term pregnancy. A well-defined, painful, tense pelvic-abdominal mass was palpable upon vaginal examination, and the fornices were completely obliterated. A cystic fluid-filled structure with intracystic vegetations was seen on an abdominal ultrasound. It was extensively vascularized on Doppler examination and, based on its ultrasound appearance, it was thought to be of ovarian origin. Laboratory tests revealed hemoglobin 12.8 g/dl and mild leukocytosis (leukocytes = 13.27 m/mm³), but no other significant abnormalities in the coagulation profile, urine analysis, or biochemistry.

An analgesic treatment was administered, but the pain intensified without relief, which led to the decision for urgent laparotomy. A median pubo-supraumbilical incision was made, and upon opening, a tumor formation of approximately 25 cm was found, firm and adherent to the omentum (viscerolysis was performed), with uterine attachment (left uterine horn, uterus twisted approximately 180 degrees, both adnexa adherent and twisted) – Figures 1 and 2. Considering the patient’s age and the extent of the torsion, with both adnexa involved, a total hysterectomy with bilateral adnexectomy was decided. The histopathological result revealed a pedunculated subserosal uterine leiomyoma, extensively necrotic (ischemic necrosis), with cystic transformation following infarction.

Postoperatively, the patient had a favorable recovery, under antibiotic treatment analgesics, anticoagulants and antiemetics. She experienced a hypertensive episode, for which a cardiology consultation was performed, ruling out any acute pathology. She was discharged from the hospital four days postoperatively.

Discussion

A large, heavy fibroid may rotate due to its weight, placing traction on the uterus. When the leiomyoma is sessile, the uterine torsion occurs simultaneously with the torsion of the leiomyoma⁽⁶⁾. The site of uterine torsion typically lies at the uterine isthmus. In our case, the leiomyoma was pedunculated, though large in size.

A single-center retrospective investigation on complex subserosal fibroids that required surgery found that torsion of a uterine leiomyoma is a rare cause of acute abdominal pain, with an incidence below 0.25%. The greatest risk of torsion is linked to pediculated subserosal fibroids, particularly in cases when the pedicle is narrow and lengthy, as in the example⁽⁷⁾.

Inadequate isthmus healing can lead to a less-than-ideal recovery of the normal cervical length, according to a study that used magnetic resonance imaging (MRI) to evaluate women following a lower uterine segment caesarean operation. This could put the uterus at risk for torsion by causing an extended cervix with structural weakness and aberrant angulation in the isthmic area⁽⁸⁾. In our case, the patient gave birth by caesarean section 11 months before, following an IVF procedure.

Large degenerating cystic fibroids and pedunculated subserosal fibroids are two examples of uterine leiomyomas that can closely resemble ovarian tumors, making the diagnosis and treatment extremely difficult. The main imaging technique used to diagnose suspected leiomyomas is ultrasound. Depending on the proportion of fibrous tissue to smooth muscle and if calcification or degenerative changes are present, these tumors usually show up on ultrasonography as homogenous or heterogeneous hypoechoic masses. However, using abdominal or transvaginal ultrasonography to identify the origin might be extremely difficult due to the high size of pelvic tumors^(9,10). In the case we presented, the differential diagnosis was challenging. On ultrasound, a giant pelvic-abdominal mass was visualized, cystic in nature, with liquid content, intrachystic vegetation, and vascularization present on Doppler examination. This was due to necrosis and cystic transformation following infarction of the leiomyoma.

Conclusions

Uterine torsion, though a rare condition, should be considered in the differential diagnosis when a patient presents with unexplained pelvic-abdominal pain, especially in the presence of large uterine fibroids or adnexal masses. Early recognition and timely surgical intervention are crucial to prevent complications such as ischemia, necrosis and subsequent loss of fertility. The case we presented highlights the complexity of diagnosing uterine torsion, particularly when it is accompanied by ovarian torsion and a large pedunculated fibroid. Diagnostic imaging plays an essential role in identifying such conditions, but the final diagnosis often relies on surgical exploration. This case emphasizes the importance of considering uterine torsion, even in non-gravid patients, and the need for a thorough and comprehensive approach to diagnosis and treatment.   

Corresponding author: Alina Potorac E-mail: alinapotorac05@gmail.com

Conflict of interest: none declared.

Financial support: none declared.

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