Ovarian torsion during ovarian stimulation prior to trigger followed by successful oocyte retrieval: A case report

Abstract

Objective

To report a case of ovarian torsion during ovarian stimulation prior to trigger followed by laparoscopic detorsion, trigger, and subsequent successful oocyte retrieval, as well as to review outcomes from the cycle.

Case Presentation

A 32-year-old woman with a history of recurrent ovarian torsion presented with ovarian torsion during ovarian stimulation for in vitro fertilization prior to trigger injection. She underwent laparoscopic ovarian detorsion followed by trigger and oocyte retrieval.

The rate of fertilization and blastocyst conversion of oocytes retrieved from the ovary affected by torsion was lower than from the contralateral ovary. Viable oocytes and an embryo were produced by the affected ovary.

Conclusion

Ovarian torsion during ovarian hyperstimulation for in vitro fertilization is a rare occurrence, particularly prior to trigger. Clinicians can consider proceeding with trigger and oocyte retrieval after treating ovarian torsion in appropriately counseled patients.

Highlights

• •Ovarian torsion can occur during ovarian stimulation prior to trigger.
• •Detorsion prior to trigger with subsequent trigger may be an option.
• •Oocytes retrieved from a detorsed ovary may have decreased fertilization.
• •Viable oocytes and embryos may result from a detorsed ovary.

1. Introduction

Ovarian torsion is a serious condition that can result in loss of ovarian function. Risk factors include ovarian hyperstimulation syndrome (OHSS), history of ovarian torsion, endometriosis, and ovarian cysts [1]. Patients undergoing assisted reproductive technologies with ovarian stimulation are at greater risk of ovarian torsion due to increased ovarian size [[2], [3], [4]].

Ovarian torsion in patients undergoing in vitro fertilization (IVF) most commonly occurs following oocyte retrieval, with an incidence of 0.024%–0.2% [[5], [6], [7], [8], [9]]. Few cases of torsion have been reported during the period after the administration of hCG (human chorionic gonadotropin) or gonadotropin-releasing agonist (GnRHa) for oocyte maturation (trigger) and before retrieval [[10], [11], [12]]. This report presents a case of ovarian torsion during ovarian stimulation for IVF prior to trigger followed by laparoscopic detorsion and subsequent oocyte retrieval, as well as to review outcomes from this cycle.

2. Case Presentation

The patient was a 32-year-old nulliparous woman with primary infertility related to polycystic ovarian syndrome. She presented to clinic with 12 months of infertility and a history of right ovarian torsion during a cycle of clomiphene citrate (CC) and timed intercourse. Her antimullerian hormone level was 9.3 ng/mL and antral follicle count was >20 follicles bilaterally. Pelvic ultrasound was consistent with polycystic ovarian morphology and remaining workup was unremarkable.

She was treated with CC and intrauterine insemination (IUI). During her second cycle, three days after an 11 mm follicle was noted, she developed severe right lower quadrant pain and vomiting. She underwent laparoscopy and was noted to have a dusky right ovary with torsion of the infundibulopelvic ligament and fallopian tube x 2 (Fig. 1, Fig. 2). Detorsion was performed and IUI was deferred.

Fig. 1–2.

Intraoperative surgical findings which demonstrate the enlarged, edematous right ovary.

She was counseled on options for prevention of recurrent torsion including expectant management, utero-ovarian ligament plication, or oophoropexy. She underwent laparoscopic bilateral utero-ovarian ligament plication with permanent suture.

She then completed three cycles of CC + IUI with 1 or 2 dominant follicles each cycle, but without pregnancy. Next, she proceeded to IVF using a long-leuprolide acetate protocol with follicle stimulating hormone (FSH) 75 international units (IU), human menopausal gonadotropin (hMG) 75 IU, and hCG trigger with conventional insemination. She had 28 oocytes retrieved, resulting in 5 blastocysts, which were cryopreserved due to development of moderate OHSS. She subsequently underwent a single frozen embryo transfer resulting in a biochemical pregnancy, and then three additional frozen embryo transfers, all with negative hCG results.

Next, she proceeded with a second IVF cycle using an antagonist protocol with 75 IU FSH and 75 IU hMG, with a plan for a leuprolide acetate trigger, intracytoplasmic sperm injection, and freezing of all blastocysts due to the history of OHSS. On cycle day (CD) 12, estradiol (E2) was 1400 pg/mL, and ultrasound showed five 11–14 mm follicles in the right ovary, six in the left ovary, and numerous smaller follicles. She was instructed to continue medications and return in two days.

The following day, she reported acute right lower quadrant pain, nausea, and vomiting. She presented to clinic within two hours and was found to have right lower quadrant tenderness with rebound and guarding. Ultrasound showed an enlarged right ovary (4.9 cm × 4.9 cm × 4.4 cm) with multiple follicles and diminished blood flow. She was taken to the operating room for laparoscopic right ovarian detorsion. The infundibulopelvic ligament was torsed x 4. The ovary was noted to be enlarged without grossly visible ischemia. A detorsion was performed. The permanent suture from her prior utero-ovarian ligament plication was intact and the ligaments appeared shortened. She was discharged and continued the gonadotropin-releasing hormone antagonist.

She was feeling well in clinic the next day. Her E2 had risen to 1790 pg/mL and ultrasound showed one follicle >16 mm in the right ovary and five in the left ovary, with normal flow bilaterally. Given the rise in E2 and appropriately sized follicles, options including cycle cancellation or proceeding with trigger and retrieval of oocytes were discussed. The decision was made to proceed with a leuprolide acetate trigger that evening and retrieval 36 h after trigger (72 h after surgery). From the right, torsion-affected ovary, three mature eggs were retrieved, resulting in two day 6 blastocysts, with one high-quality blastocyst for cryopreservation. Eight mature eggs were retrieved from the left, yielding five high-quality blastocysts, which were cryopreserved on day 5. Six weeks later, she had a frozen embryo transfer using a modified natural cycle with FSH. Per center guidelines, accounting for prior unsuccessful transfers, two day 5 blastocysts (4BB, 4BB) originating from the unaffected ovary were transferred. Selection of embryos was based on morphology and day of cryopreservation, not ovary of origin. She had an appropriate rise in hCG and ultrasound at 6 weeks of gestation demonstrated a singleton intrauterine gestation. The pregnancy was uncomplicated, and she delivered at 41 weeks of gestation.

3. Discussion

The patient experienced ovarian torsion during ovarian stimulation prior to trigger and underwent laparoscopic detorsion, trigger, and oocyte retrieval. To our knowledge, this is the first case report of a surgically confirmed case of ovarian torsion prior to trigger during an IVF cycle.

There has been one reported case of suspected ovarian torsion before trigger, but torsion was not confirmed on ultrasound or managed surgically [13]. In that case, a 34-year-old patient presented with symptoms suggestive of torsion and they proceeded with aspiration of follicular fluid for volume reduction of the right ovary, followed by a GnRHa trigger and oocyte retrieval 35 h later (see Table 1 for fertilization rates). This case is limited in its application to clinical management as ovarian torsion was not confirmed on either ultrasound or surgically. Additionally, the management of torsion with volume reduction through follicular aspiration is not a widely accepted method of management.

Table 1.

Fertilization outcomes from our patient and published case reports.

Case Report	Timing of Torsion	Fertilization Rate: Unaffected Ovary	Fertilization Rate: Ovary with Torsion	Notes
Robson et al. 2000	Post trigger	86%	33%	Detorsion after retrieval
Smith et al. 2010	Post trigger	93%	40%	Detorsion before retrieval
Stefanidis et al. 2002	Post trigger	22%	No eggs retrieved	Detorsion after retrieval
Inoue et al. 2020	Pre trigger	100%	83%	Pain resolved after follicle aspiration prior to trigger; torsion was never confirmed with surgery or imaging
Naert et al. 2023	Pre trigger	100%	66%	Detorsion before trigger and retrieval

There have been three prior case reports of ovarian torsion after trigger and prior to retrieval [[10], [11], [12]] (Table 1). One case describes a 27-year-old who took a recombinant hCG trigger on CD 10 and presented to clinic on CD 11 with right lower quadrant pain, nausea, and emesis [10]. She underwent laparoscopic right ovary detorsion eight hours after the onset of pain. One day later, she proceeded with oocyte retrieval.

In the other two cases oocyte retrieval was done prior to laparoscopic detorsion. One case features a 37-year-old who administered the hCG trigger injection and presented with symptoms of left ovarian torsion on the morning of her planned retrieval [11]. Retrieval was performed, hoping that volume reduction of the ovaries would alleviate the ovarian torsion. The patient required subsequent laparoscopic detorsion two hours after retrieval.

The final case [12] describes a 33-year-old who presented with symptoms of left ovarian torsion on the day of her planned retrieval. She underwent transvaginal oocyte retrieval. Pain persisted, and she underwent laparoscopic detorsion six hours after retrieval.

These aforementioned cases where torsion occurs prior to retrieval are rare, as most cases of ovarian torsion in IVF patients occur after retrieval, with presentation any time from 5 days after retrieval to 5 weeks after embryo transfer [14,15]. Torsion after retrieval may be more common due to persistence of corpus luteal cysts, especially in a setting of ongoing pregnancy [18].

There is limited data regarding the management of ovarian torsion during an IVF cycle, particularly when it occurs prior to trigger, as in this case. Given the lack of data, shared decision-making is critical, as is considering the risks and benefits of proceeding with the cycle. Expected impact on oocyte quantity at retrieval and fertilization outcomes post-torsion would contribute to this discussion, but data is limited. Table 1 contains a summary of data from the patient and cases discussed above. All had reduced fertilization rates among the oocytes retrieved from the affected ovary relative to the unaffected ovary, and in one case no oocytes were retrieved from the affected ovary.

Several factors likely contribute to decreased fertilization rates of oocytes from torsed ovaries. One mechanism is decreased arterial flow to the ovary [10], which may lead to a reduced concentration of hCG or luteinizing hormone in the ovary and result in dysfunctional folliculogenesis [12]. Torsion also causes venous and lymphatic stasis, which can also lead to further dysfunctional folliculogenesis [12]. Torsion may cause ischemic injury to oocytes. However, it is unclear how ischemia might impact oocyte viability, fertilization, and maturation [10]. There have been successful pregnancies from ovarian tissue cryopreservation and transplantation, suggesting that although there is likely ischemic injury to follicles due to oophorectomy, ischemia does not cause irreversible damage to follicles [16,17]. Overall duration of the ischemic event may also impact outcomes.

4. Conclusions

In sum, the outcomes of this patient and the other patients discussed above suggest decreased fertilization of oocytes from the ovary affected by torsion. Although torsion may compromise oocyte quality in some capacity, it is still possible to obtain viable oocytes and produce embryos from the affected ovary. Therefore, for patients with ovarian torsion prior to trigger, laparoscopic detorsion followed by trigger and oocyte retrieval is an acceptable option for an appropriately counseled patient.