{"paper_id":"4588bf3a-ca1e-4d30-a17d-a7ada811449d","body_text":"Ovarian torsion in patients undergoing in vitro fertilization (IVF) is a rare (0.024–0.2%) but serious gynecologic diagnosis requiring emergent surgical intervention to salvage ovarian function  [1] . Risk factors for ovarian torsion include ovarian mass, increased ovarian volume, polycystic ovary syndrome (PCOS), ovarian hyperstimulation syndrome (OHSS), and pregnancy  [2] ,  [3] . Patients with a history of PCOS or those classified as high responders to ovarian stimulation are at an elevated risk of ovarian torsion due to the enlarged size of their ovaries compared with typical IVF patients.\nIn patients undergoing controlled ovarian hyperstimulation (COH), clinicians should maintain heightened clinical suspicion for ovarian torsion. Some case reports explore ovarian torsion in the setting of COH, but the authors could find none highlighting the delayed nature of torsion.\nOvarian torsion should be treated emergently with laparoscopic adnexal detorsion. Scant attention has been given to the development of vulvar hematoma in the context of laparoscopic adnexal procedures  [4] . Vulvar hematoma can be high risk, often complicated by infection, hemorrhage, or hypovolemic shock  [5] . One report attributed a case of vulvar hematoma in laparoscopic adnexal surgery to trocar insertion or bleeding from epigastric vessels injured during the procedure  [6] . No literature could be found documenting vulvar hematoma as a post-operative complication of laparoscopic ovarian detorsion and ways to predict, plan, or manage in this setting.\nThe present report concerns a patient with PCOS who experienced two unique complications: rare, delayed torsion after period following COH, and vulvar hematoma after laparoscopic detorsion. The purpose of this case report is to identify modifiable risk factors and to improve clinical care.\n\nThe patient was a 37-year-old nulliparous Caucasian woman who presented with 6 months of infertility in the setting of known PCOS. She was managed on metformin 1000 mg daily with q40 day menstrual cycles. She had a BMI of 24.6 kg/m 2 ; her anti-Mullerian hormone level was 14.3 ng/ml and total antral follicle count on transvaginal ultrasound was 38 without adnexal pathology. Hysterosalpingogram showed bilateral tubal patency and normal uterine contour, but with filling defect consistent with a possible endometrial polyp, for which she underwent an uncomplicated hysteroscopic polypectomy.\nGiven her age, PCOS and desire for two or three children, she pursued a cycle of IVF with preimplantation genetic testing for aneuploidy. She underwent ten days of COH with an antagonist protocol. Her ovarian response was monitored with daily visits from day 5 of stimulation through oocyte retrieval day. Gonadotropin doses were carefully tapered in response to estradiol levels and follicular measurements to minimize risk of ovarian hyperstimulation. Throughout stimulation, the patient denied any adnexal discomfort. She underwent meiotic trigger with leuprolide acetate after 10 days of stimulation when lead follicles measured 19.1 mm and peak estradiol was 3909 pg/ml.\nShe underwent an uncomplicated oocyte retrieval and had 47 oocytes retrieved. 36 oocytes fertilized to 2PN zygotes with conventional insemination, with 23 blastocysts sent for -A, ultimately with 11 euploid embryos. She met post-operative milestones on post-operative day 1 and had her menses on post-operative day 6, when she reported feeling back to normal, with minimal need for pain medication. She requested to pursue frozen embryo transfer with this subsequent menses, and presented seven days after her retrieval with menses, for the start of a frozen embryo transfer cycle. However, her ovaries were persistently enlarged, with a stimulated appearance. Despite the patient feeling asymptomatic, the decision was made to postpone her embryo transfer cycle to allow for complete resolution following COH.\nThe next day, post-retrieval day 8, she presented to the emergency room with sudden onset of left lower abdominal pain, nausea and vomiting. Emergent pelvic imaging revealed bilaterally enlarged ovaries, with the right ovary measuring 10.6 × 6.5 × 6.0 cm and the left ovary 9.6 × 6.9 × 8.6 cm and moderate pelvic free fluid with preserved arterial and venous flow. However, given her clinical presentation consistent with ovarian torsion the decision was made to proceed to diagnostic laparoscopy.\nLaparoscopic entry was uncomplicated, with placement of a 10 mm supra-umbilical Hasson port for open entry. Two additional 5 mm assistant ports in the right and left lower quadrants were then placed approximately 2–3 finger-breadths higher than normal, given the enlarged ovaries and anticipated need for better visualization and laparoscopic mobility. The left ovary appeared swollen and dusky, and was twisted 540 degrees around the utero-ovarian ligament. Successful detorsion was performed. Closure of the umbilical incision and port sites concluded the procedure. Estimated blood loss was 200 cc.\nThe patient was voiding and ambulating with pain well controlled on post-laparoscopy day 0 but the decision was made to keep her in hospital for an extra night, for observation.\nOn post-laparoscopy day 1, she felt a painless swelling on the left labia. Over the next 24 h, the swelling progressed to her mons, with transition to ecchymosis over a 6x2cm area (see  Fig. 1 ). Her hemoglobin down-trended to 9.6 g/dl, from 12.1 g/dl pre-laparoscopy, with a 6-h repeat CBC which was stable. Given clinical stability, the team elected for expectant management. The vulvar hematoma began to resolve on post-laparoscopy day 2, and the patient met criteria for hospital discharge. Close outpatient follow-up was continued. The patient endorsed non painful, non-traumatic bruising along the left upper flank starting on post-laparoscopy day 5 ( Fig. 2 ), which resolved without complication. Fig. 1 Progression of vulvar hematoma on day 1 after laparoscopy (right-hand image 5½ hours after left-hand image). Fig. 1 Fig. 2 Post-laparoscopy complication: left upper lateral abdominal bruising post-laparoscopic adnexal detorsion, day 4. Fig. 2\nProgression of vulvar hematoma on day 1 after laparoscopy (right-hand image 5½ hours after left-hand image).\nPost-laparoscopy complication: left upper lateral abdominal bruising post-laparoscopic adnexal detorsion, day 4.\nAfter successful return to baseline one month post-laparoscopy and hematoma resolution, the patient initiated a frozen embryo transfer cycle with a single euploid embryo which resulted in a viable singleton pregnancy. She had an uncomplicated pregnancy and underwent induction of labor at 41 weeks and 1 day with a spontaneous vaginal delivery of a healthy baby boy weighing 6 lbs. and 12 oz.\n\nThe incidence of ovarian torsion in patients pursuing COH remains uncertain. Studies highlight an elevated risk during and after ovarian stimulation in the setting of ovarian enlargement  [7] . Patients with a high ovarian reserve are often classified as high responders due to an increased baseline number of follicles compared with an age-matched patient with normal ovarian reserve  [8] . Individuals with PCOS have a higher risk of ovarian hyperstimulation syndrome. Though the delayed resolution of ovarian volume after trigger in high responders is not well quantified in current studies, PCOS patients are at an overall increased risk for ovarian torsion given the higher baseline ovarian volume from IVF  [9] ,  [10] .\nThis patient's risk factors included her history of PCOS, as well as the prolonged enlargement of the ovaries in the setting of COH. Her clinical presentation, coupled with her risk factors, led to a high suspicion for ovarian torsion and prompt diagnosis through laparoscopy. A large yield on oocyte aspiration coupled with a shortened luteal phase after leuprolide acetate only trigger should lengthen the restrictions for a longer period, perhaps after menses in this population. Typically, the onset of menses marks a “safety zone” for patients; however, patients with PCOS will have higher egg counts and may require more time to heal prior to initiating embryo transfer.\nSurgical management with laparoscopic detorsion surgery remains the gold standard treatment for ovarian torsion. Even when the ovary appears ischemic, detorsion can effectively restore blood flow, potentially salvaging future ovarian function  [11] .\nVulvar hematomas most commonly occur in the context of obstetric trauma. Vulvar hematomas in non-obstetric settings are infrequent, accounting for up to 0.8% of all gynecologic hospitalizations  [12] . Vulvar hematoma resulting from laparoscopic surgery is rare, possibly because the vasculature connections are more distant. The vulva is primarily supplied by the internal pudendal artery, with labial veins forming the venous drainage system that empties into the external and internal pudendal veins  [13] ,  [14] .\nWhile vulvar hematoma is infrequently encountered as a complication of laparoscopic surgery, it is crucial to recognize the risk factors associated with IVF patients. Injury to the branches of the pudendal artery can cause vulvar hematoma – either from spontaneous rupture or direct damage  [15] . Ovarian vein damage can contribute to vulvar hematoma, as the ovarian vein establishes anastomoses with venous plexuses, including the paraovarian, uterine, vesical, rectal, and vulvar  [16] . Few case reports document vulvar hematoma after laparoscopic surgery. One instance was attributed to injured epigastric vessels during laparoscopic cystectomy  [17] . Tokmak et al. reported a case of vulvar hematoma following laparoscopic endometrioma excision and explored potential etiologies, citing injury to the round ligament or damage to the superficial epigastric vessels during trocar insertion, which communicate with the superficial perineal pouch  [6] . Reducing vessel injury during trocar entry is challenging, though proper laparoscopic surgical technique reduces this risk.\nThe process of IVF may contribute to the risk of vulvar hematoma. Ovarian stimulation upregulates estradiol and ovulation with trigger medication increases the release of VEGF, thereby promoting angiogenesis and increasing blood flow as well as capillary fragility  [18] ,  [19] . Individuals with PCOS exhibit a greater likelihood of having anomalies in ovarian angiogenesis at baseline  [20] . It is plausible that this patient carried an elevated risk for post-operative bleeding due to variations in pelvic vasculature. Ovarian stimulation may have contributed to vessel swelling and friability, and manipulation during detorsion may have caused bleeding from the ovarian veins, subsequently resulting in vulvar expansion.\nNo obvious bleeding was noted during trocar insertion or the detorsion process. However, occult bleeding may be difficult to recognize during laparoscopy. Wong et al. documented three cases of post-operative bleeding during laparoscopic adnexal surgery, proposing that delayed bleeding may arise due to the tamponade effect induced by pneumoperitoneum  [21] .\nHigher port placement was utilized to accommodate the bilaterally enlarged ovaries and to ensure optimal visualization. Four days following the detorsion, the patient exhibited abdominal bruising, as depicted in  Fig. 2 , prompting suspicion of injury to a branch of the epigastric vessel. While increased capillary fragility associated with ovarian stimulation may have contributed to a delayed presentation, the anatomical tracking of the hematoma suggests that damage to an arteriolar branch during trocar placement is a more probable etiology; however, the definitive cause of the hematoma formation remains speculative.  Fig. 3  outlines the typical and actual placement of laparoscopic ports in this patient's case, and the potential vessels in the abdominal wall that could have been injured. Physicians managing patients with risk factors such as enlarged ovaries must balance the advantages of enhanced visualization against the risk of injuring abdominal wall vessels during entry. Fig. 3 (A) Green dots represent typical laparoscopic port placement. Pink brackets represent ovary location in a typical patient. (B) Red dots represent port placement in the present case, with enlarged ovary. Purple brackets represent ovary location in the present case, with stimulated, enlarged ovaries. (Created with BioRender). (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.) Fig. 3\n(A) Green dots represent typical laparoscopic port placement. Pink brackets represent ovary location in a typical patient. (B) Red dots represent port placement in the present case, with enlarged ovary. Purple brackets represent ovary location in the present case, with stimulated, enlarged ovaries. (Created with BioRender). (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.)\n\nThis patient had two rare complications of routine procedures. She had had several risk factors for torsion, including enlarged ovaries due to COH and a history of PCOS. By analyzing these events as a linked clinical sequence, it becomes evident that the persistent ovarian enlargement and anatomical distortion likely predisposed this patient to the development of the ipsilateral vulvar hematoma.\nVulvar hematoma is a rare complication of laparoscopic surgery. Risk factors may include: abdominal vessel injury, pelvic vessel damage during the procedure, and preexisting factors such as altered vascular anatomy or, in the case of IVF, angiogenesis or capillary fragility. This case highlights that careful consideration of port placement is essential to minimize the risk of intra-operative and post-operative bleeding.\n\nAll authors contributed to patient care, conception of the case report, acquiring and interpreting the data, drafting the manuscript, undertaking the literature review and revising the article critically for important intellectual content.\nAll authors approved the final submitted manuscript.\n\nInformed consent was obtained from the patient for publication of this case report and accompanying images. The patient was informed that no identifying information would be published.\n\nThis article was not commissioned and was peer reviewed.\n\nNo such technologies were used during the preparation of this work.\n\nThis work did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.\n\nThe authors declare that they have no competing interest regarding the publication of this case report.","source_license":"CC0","license_restricted":false}