An Unusual Presentation of Recurrent Ovarian Torsion in an Adolescent Female

Introduction

Ovarian torsion is a gynecologic emergency and a is fairly common cause of acute abdominal pain in women of reproductive age.^1,2 This torsion is a complete or partial rotation of the ovary around the adnexal organs that can result in ischemia. ³ Ovarian torsion usually presents with acute-onset lower-abdominal pain that can be accompanied by nausea and vomiting. ³ Although the annual prevalence of ovarian torsion in females ages 1–20 is reported to be 4.9 per 100,000, the diagnosis should be considered in all girls with acute-onset abdominal or pelvic pain. ⁴

According to Bertozzi et al., roughly one-half of torsions in children are associated with masses that prevent the adnexa from returning to their normal positions after twisting. ⁵ Although patients with normal ovaries account for ∼16%–49% of cases, the cause of torsion in ovaries without masses is of unclear etiology. ⁵

According to Fee et al., the pediatric population is more prone to ovarian torsion and recurrence due to “hypermobile structures, naturally longer utero-ovarian ligaments, adnexal venous congestion, constipation and jarring movements.” ⁶ Torsion is also thought to occur more commonly on the right side due to the cushioning of the sigmoid colon on the left and possibly because the right infundibulopelvic ligament is longer. ¹

The mainstay diagnostic tool for detecting torsion is an ultrasound (US) with Doppler. ³ Doppler may detect the absence of blood flow; but when blood flow is present, this does not exclude the diagnosis. ³ The only definitive way to confirm an ovarian torsion is through visualization during surgery. ³

In the absence of an adnexal mass causing torsion, treatment is typically detorsion or oophoropexy but this carries a risk of the patient having a recurrence in the future. While the incidence of ipsilateral recurrent ovarian torsion is not completely known, it is thought to be between 2% and 5%. ⁴

Case

The patient was a 15-year-old, gravida 0, who presented to the clinic in 2019 for pain associated with her menstrual cycle that also occurred intermittently when she was not menstruating. The pain was described as diffuse in the right lower quadrant, left lower quadrant, and suprapubic area with radiation down her legs. She said that she was unable to do sports or regular activities on some days due to this pain. Her monthly cycles lasted 5–6 days with changing pads every 3 hours. She had been taking ibuprofen as needed for pain without obtaining relief. She was not sexually active. She denied having fever, chills, nausea, vomiting, diarrhea, constipation, and breast pain.

This patient's abdomen was soft, nontender, and nondistended on physical examination. Her blood pressure was 113/79, her pulse was 91 beats per minute, and she was afebrile. A Pelvic examination was deferred, because she was not sexually active. The remainder of the physical examination was unremarkable.

The only past medical history included a right ovarian torsion in 2017 that was surgically reduced, with a normal US showing resolution afterward. During that exploratory laparoscopy in 2017, the right ovary was found to be dark purple and twisted 4–5 times; this torsion was subsequently reduced. After 30 minutes, that ovary was reexamined and was noted to have improved to an injected pinkish red color. It was at this point that the decision was made to leave the ovary in situ. The operative notes did not mention any anatomical anomalies or masses of the ovary.

The patient reported, however, that she had chronic pelvic pain since that time.

The visit diagnosis was menstrual-cycle disorder. This patient's presentation and age were consistent with this diagnosis. Given the history of a previous ovarian torsion, a pelvic US was discussed to reassure the patient and her mother. The first-line treatment considered was hormonal control with oral contraceptive pills.

The patient and her mother decided to have the patient undergo a pelvic US 2 days later due to the increased pain and vomiting that she had experienced since those 2 days. The US showed that she had a left ovarian enlargement (5.4 × 4.7 × 5.3 cm) with an approximated volume of 70 cc. There was a complex cyst occupying a large portion of the left ovary. The impression from the radiologist was “normal flow is seen but given enlargement and history of previous torsion, recent or intermittent torsion remains a consideration. A follow-up ultrasound is recommended in 6 weeks for re-evaluation of complex cyst occupying large portion of the ovary. Right ovary not visualized.”

The US reading was communicated to the patient's mother, who was provided with precautions of when to present to the emergency department (ED). The patient experienced nocturnal, unremitting pain and subsequently went to the ED. This occurred 2 days after her initial presentation in the clinic and several hours after the pelvic US was performed.

A pelvic US was conducted in the ED just several hours after the first pelvic US, and both were performed transabdominally. However, the second US showed absent blood flow in the right ovary that was highly concerning, suggesting ovarian torsion, and a 5.8-cm, left, ovarian cystic lesion that was most likely a hemorrhagic cyst.

Exploratory laparoscopy was conducted in accordance with the US findings and the patient's past medical history. The operative findings were consistent with a recurrent right ovarian torsion; the right ovary and fallopian tube were twisted seven times with apparent anomalous attachment, an unidentifiable infundibulopelvic ligament, and a free-floating right tube and ovary. A large 4 cm × 4 cm, left ovarian cyst was also seen and was believed to be hemorrhagic. The ovary was watched for more than 30 minutes; however the right tube remained firm, friable, necrotic, and deeply purple, with a permanent stricture proximally. Ultimately, after consent from the patient's mother, a right salpingo-oophorectomy was performed.

Results

A follow-up transvaginal pelvic ultrasound was performed 1 month after this patient's surgery. She had continued, mild, left ovarian enlargement (28 mL versus 85 mL) and the previously noted hemorrhagic cyst was not identified. Her postoperative course was uneventful.

Discussion

The current patient's presentation and age were consistent with a diagnosis of primary dysmenorrhea. According to Parker et al., pain with menstruation is seen in 70%–91% of teenagers. ⁷ In addition, menstrual pain and associated symptoms have caused absences from school in 14%–51% of teenage girls and interference with life activities for 15%–59% of these teenagers. ⁷ The current patient's past medical history was the only indication to suspect a recurrent torsion, although it seemed unlikely with her symptoms. She had chronic, cyclic pain in contrast to the typical presentation of ovarian torsion, which is sudden-onset, acute pain.

In a retrospective case review done by Tsafrir et al., 5 of 22 cases (22.7%) of ovarian torsion presented with a gradual onset of abdominal pain. ⁴ In addition, Hartley et al. reported that half of patients with torsion had experienced similar episodes of abdominal pain in the past. ¹ Bertozzi et al. conducted a pediatric literature review and found 4 cases of ipsilateral ovarian torsion recurrence and reported that the median time from the first episode of torsion and recurrence was 12.2 months, with the shortest time being 6 months and longest time being 24 months. ⁵ The current patient went 24+ months between the first torsion and the recurrence.

Intermittent chronic pain is an abnormal presentation of ovarian torsion, which makes the diagnosis difficult, especially due to the prevalence of primary dysmenorrhea in adolescent females. Therefore, the variability of presentation and pain profile of a woman with abdominal or pelvic pain should not preclude a broad differential.

Conservative management of ovarian torsion involves detorsion, which is simply just untwisting of ovary to allow ovarian conservation and fertility preservation in patients desiring to have children. Another management option is oophoropexy. Oophoropexy is “fixation of the ovary to neighboring structures such as pelvic lateral sidewall, ovaric fossa, or posterior wall of the uterus.” ⁵ This method is thought to help prevent recurrence of torsion in patients who do not have an ovarian masses or cysts. As previously stated, the incidence of recurrent ipsilateral ovarian torsion is not completely known and, consequently, the recurrence rate of detorsion versus oophoropexy is also unkown. ⁴

This case also highlights the reason why US is a good diagnostic tool for helping detect torsions but why US is not the “gold standard.” The first US was a transabdominal pelvic US and the impression was that the right ovary could not be seen and there was normal Doppler venous waveform seen. The second US was conducted several hours after the first one, and it was also a transabdominal pelvic US. During the second US, the right ovary was detected and noted to have absent blood flow. There are technical limitations to using US, as well as variability in provider proficiency, which should always be kept in mind.

The chronic diffuse lower abdominal and pelvic pain that this patient experienced might have been attributable to her ovary and fallopian tube gradually twisting over time. It is also probable that between the time the 2 US were done the ovary and fallopian tube had twisted again, completely cutting off all blood flow, thus, explaining the difference in the Dopplers. While US is the mainstay diagnostic tool for detecting ovarian torsion, there are limitations that must always be considered—and US should never replace a thorough history and physical examination.

As the field of minimally invasive gynecologic surgery becomes more pervasive, more research will be done on options for treating ovarian torsion. Currently, research needs to be conducted to determine recurrence rates, as well as comparison of reducing torsion with and without oophoropexy to refine the treatment paradigm further. Fee et al pointed out that there is not a lot known about the complications of oophoropexy and that most of the studies done were in regard to the fertility risks of altering normal anatomy. ⁶ Research must also be conducted to determine potential complications as well as the downstream effects oophoropexy might have on fertility.

Conclusions

If research shows that oophoropexy not only reduces recurrence but also helps to preserve fertility in patients who desire future pregnancy, it follows that oophoropexy should be first line conservative management for ovarian torsion.